1
MASARYKOVA UNIVERZITA
Nové poznatky v oblasti patogeneze a následk edoušního
u v
MUDr. Milan Urík, Ph.D.
Brno 2021
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za
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Obsah
1 .................................................................................................................................... 4
2 ........................................ 5
2.1 ............................................................................................. 5
2.2 .......................................................................................... 11
2.3 ...................................................................................... 14
2.4 .......................................................................................... 14
3 ...................................................................................... 17
3.1.1 ............................................................................... 17
3.1.2 .................................................................................................... 17
3.1.3 .......................................................................................................... 17
3.1.4 ....................................................................... 18
3.1.5 ....................................... 19
3.1.6 ............................................................. 20
3.1.7 ....................................................................... 23
3.1.8 ..................................................... 24
3.1.9 ............................................ 26
3.1.10 ..................................................... 26
3.1.11 .............................................................. 27
3.1.12 ....................................................................... 28
3.1.13 ....................................................................... 29
3.2 ............................................................. 30
3.2.1 ........................................................................... 32
3.2.2 ......................................................... 34
3.2.3 ....................................................................... 38
3.2.4 ................................................... 46
3.2.5 ....................................................................... 47
3.2.6 ....................................................................... 49
4 ......... 51
4.1.1 ....................................................................... 54
5 .................................................................................... 56
6 .................................................................................................. 60
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20
PŮVODNÍ PRÁCEPŮVODNÍ PRÁCE
SOUHRN
Úvod: Článek prezentuje výsledky studie, která zkoumala, jak se změnilo spektrum bakteriálních patogenů způsobujících
akutní otitidy, meningitidy otogenního původu a epiglotitidy v dětském věku po zavedení povinného plošného očkování
vakcínou proti Haemophilus influenzae b (dále Hib) v České republice.
Materiál a metodika: V rámci studie jsme sledovali 2 soubory pacientů. Kontrolní soubor tvořily děti narozené před rokem
2001, které nebyly očkované vakcínou proti Hib. Sledovaný soubor tvořily děti narozené po roce 2001, které byly očkované
vakcínou proti Hib. Studie probíhala v letech 2002–2012. V každém souboru byly sledovány 3 skupiny dětí: 1. s akutní
otitidou, 2. s otogenní meningitidou a 3. s epiglotitidou. Ve všech případech jsme provedli kultivaci odebraného materiálu
s cílem identifikovat Hib jako původce onemocnění.
Výsledky: V kontrolním souboru bylo odebráno celkem 205 stěrů ze zevního zvukovodu ke kultivaci u dětí s akutní otitidou
po paracentéze. Hib byl nalezen jako původce v 26,3 %. U meningitid otogenního původu byl Hib jako původce nalezen
pouze v 1 případě. V tomto souboru se vyskytla epiglotitida u 8 dětí a ve všech případech byl původcem Hib. Ve zkoumaném
souboru bylo odebráno celkem 201 stěrů ze zevního zvukovodu u dětí s akutní otitidou po paracentéze. Hib byl zachycen
pouze ve 4,4 %. U meningitid otogenního původu nebyl Hib vykultivován ani v jednom případě. V souboru očkovaných dětí
jsme epiglotitidu vůbec nezaznamenali.
Závěr: Z výsledků této studie je patrný významný pokles výskytu Hib jako původce u akutních otitid, otogenních
meningitid a epiglotitid ve skupině očkovaných dětí vakcínou proti Hib. V souboru očkovaných dětí nebyl zaznamenán
žádný případ meningitidy ani epiglotitidy způsobené Hib. Očkování zaručuje téměř kompletní ochranu před závažnými
invazivními onemocněními, v našem případě před meningitidou a epiglotitidou a snižuje počet akutních otitid vyvolaných
Hib.
KLÍČOVÁ SLOVA
Haemophilus influenzae b, akutní otitida, meningitida, epiglotitida, dětský věk, očkování
SUMMARY
Haemophilus diseases in ENT in children before and after the introduction of Haemophilus influenzae
type b vaccine
Introduction: The article presents the results of a study aiming to find out the change in the spectrum of bacterial pathogens
causing acute otitis media, otogenic meningitis and paediatric epiglottitis after the introduction of compulsory vaccination
against H. influenzae b (Hib) at a national level in the Czech Republic.
Material and methods: Two patient groups were observed within the study. The control group consisted of children born
before 2001 who were not vaccinated by Hib vaccine. The observed group consisted of children born after 2001 who were
vaccinated by Hib vaccine. The study was performed during 2002–2012. Three subgroups of children were observed within
each group: 1. Suffering from acute otitis media, 2. Suffering from otogenic meningitis, 3. Suffering from epiglottitis. In all
cases the samples were cultivated in order to identify Hib as the cause of the infection.
Results: In the control group, a total of 205 smears from external ear canal were taken for cultivation in children with
acute otitis after paracentesis. Hib was identified as a cause in 26.3 %. In otogenic meningitis subgroup only one case was
caused by Hib. Eight children suffered from epiglottitis and all cases were caused by Hib. In the observed group, a total of
201 smears from external ear canal were taken in children with acute otitis after paracentesis. Hib was identified in only
4.4 % of cases. In the subgroup of otogenic meningitis Hib was not cultivated in any single case. There were no cases of
epiglottitis in the control group.
Hemofilové infekce v ORL oblasti
u dětí před a po zavedení očkování
antihemofilovou vakcínou
Forstová G.1
, Urík M.1
, Mikolášek P.2
, Pavlovská D.3
, Ševčíková A.4
, Šlapák I.1
1
Klinika dětské otorinolaryngologie FN a LF MU, Brno
2
Klinika dětských infekčních nemocí FN a LF MU, Brno
3
Klinika dětské radiologie FN a LF MU, Brno
4
Oddělení klinické mikrobiologie FN, Brno
Čes-slov Pediat 2017; 72 (1): 20–24
Příloha 1: Forstová G, Urík M, Mikolášek P, Pavlovská D, Ševčíková A, Šlapák I. Hemofilové infekce v ORL
oblasti u dětí před a po zavedení očkování antihemofilovou vakcínou. . Československá Pediatrie.
2017;18(1):20-24.
212017, 72, Č. 1 ČESKO - SLOVENSKÁ PEDIATRIE
PŮVODNÍ PRÁCE
Conslusion: The study results show a significant decrease in occurrence of Hib as a cause of acute otitis media, otogenic
meningitis and epiglottitis in the group of children vaccinated by Hib vaccine. There were no cases of meningitis or epiglottitis
cases caused by Hib in the group of vaccinated children. Vaccination provides a complete protection against serious
invasive diseases, in our case meningitis and epiglottitis, and decreases the occurrence of acute otitis media caused by Hib.
KEY WORDS
Haemophilus influenzae b, acute otitis, meningitis, epiglotitis, pediatric age, vaccination
ÚVOD
Infekce respiračního traktu tvoří téměř polovinu všech
onemocnění v dětském věku. Příznaky i etiologie nákazy
závisí na lokalizaci v dýchacím traktu. Záněty v dětském
věku postihují často sliznici nosních dutin a nosohltanu
a odtud se může zánět rozšířit do středouší [1].
Mezi nejčastější původce akutních zánětů středouší
u dětí patří Streptococcus pneumoniae, Haemophilus influenzae
a Moraxella catarrhalis [1]. Na Dětské otorinolaryngologické
klinice FN v Brně byla provedena studie, zda a jak se
změní spektrum patogenů akutních otitid a meningitid
otogenního původu po zavedení povinného očkování proti
H.influenzaeb. Dále jsme sledovali také výskyt epiglotitidy
v obou souborech. Vakcína byla do očkovacího kalendáře
dětí zařazena od 1. 7. 2001.
H. influenzae je gramnegativní tyčinka, která se podle
charakteru polysacharidů pouzdra třídí do 6 antigenních
typů označovaných písmeny a–f [2].
Přenos infekce se děje vzdušnou cestou. Virulence
kmene je podmíněna tvorbou pouzdra. Prudké infekce
jsou vyvolány typem b [3].
V laboratorní diagnostice se využívá kultivace na krevním
agaru se stafylokokovou čárou (satelitismus) a sérologické
vyšetření – průkaz polysacharidového pouzderného
antigenu v krvi (latex aglutinace, ELISA) [2].
Závažná celková hemofilová onemocnění způsobuje
zejména H. influenzae b. Celková invazivní onemocnění
probíhají většinou jako meningitida, epiglotitida či sepse.
Predisponovány jsou zejména děti ve věku od 3 měsíců do
5 let [1]. Hemofilová meningitida u dětí zanechává až ve
30 % neurologické poruchy a poškození sluchu.
Meningitidy dělíme podle způsobu vzniku na primární,
kdy k infekci dojde hematogenní cestou, a sekundární,
kdy k zánětu dojde přestupem infekce z okolního ložiska
(středouší, pneumatický systém spánkové kosti, vedlejší
dutiny nosní).
V diagnostice sekundárních meningitid je důležitá
anamnéza s výskytem opakovaných zánětů středouší
či dutin. Diagnóza se stanoví na základě přítomnosti
zánětlivého likvoru při lumbální punkci. Biochemické
vyšetření likvoru u bakteriálních zánětů ukazuje zvýšenou
bílkovinu a laktát, sníženou glukózu, cytologický
nález vykazuje pleocytózu s nálezem polymorfonukleárních
leukocytů. Cytologické vyšetření se provádí nejlépe
do jedné hodiny po odběru, později dochází u buněk
k destruktivním a deformačním změnám. Pokud nelze
cytologické vyšetření provést do 45 minut, doporučuje se
vitalitu buněk prodloužit přidáním kultivačního média
nebo inaktivovaného AB séra [4].
Cílem vyšetření odebraného moku je průkaz infekčního
agens přímo či kultivací nebo průkazem bakteriálního
antigenu pomocí PCR a nasazení optimální léčby podle
citlivosti. Vhodná antibiotika jsou zvolena podle věku,
závažnosti zánětu a výsledků odběrů.
Sekundární meningitidy mají zpravidla protrahovanější
průběh než primární. Komplikace jsou četné, akutní
stadium může být provázeno edémem mozku. Bývá
zrakové či sluchové postižení, centrální obrny. U dětí se
po prodělané meningitidě může objevit psychomotorická
retardace či hydrocefalus.
Při sekundární meningitidě je nutno provést sanaci
primárního ložiska. Dále se koriguje vnitřní prostředí,
případný edém mozku, jsou podávána antibiotika intravenózně,
dexametazon jako prevence neurologických
následků a hluchoty. Mezinárodní doporučení upřednostňují
podávání dexametazonu ještě před podáním první
dávky antibiotika a v jeho podávání pokračovat po dobu
čtyř dnů. Lékem volby jsou cefalosporiny III. generace,
popř. aminopeniciliny v kombinaci s inhibitory betalaktamáz
[5].
Akutní záněty středního ucha jsou frekventovaným
onemocněním dětského věku. Uvádí se, že až 40 % dětí
překoná alespoň jednou otitidu. Nejvyšší procento výskytu
je u dětí do jednoho roku. Akutní zánět středního
ucha u dětí vzniká většinou jako komplikace infekce
horních cest dýchacích.
V terapii akutních otitid způsobených H. influenzae b
zůstává lékem volby aminopenicilin, popř. s inhibitory
beta-laktamáz. Při přecitlivělosti k penicilinu lze podat
stabilní cefalosporiny nebo makrolidy [1].
Epiglotitida je akutní onemocnění s vysokými horečkami.
Před zavedením očkování proti Hib se vyskytovala
v každém ročním období u dětí od 2 do 7 let. Nemoc
začíná náhle vysokou horečkou, dítě je dyspnoické,
má stridor, občas kašel, zastřený hlas. Dolní čelist má
mírně vysunutou vpřed, otevřená ústa, někdy i s protruzí
jazyka. Dítě zaujímá sedící polohu s předklonem
vpřed. Tato poloha umožnuje stabilizovat epiglotis tak,
aby úplně neobturovala vchod do hrtanu. Každá změna
polohy může způsobit ucpání hrtanového vchodu edematózní
epiglotis a udušení dítěte. Intenzivní bolest
hrdla vede k poruchám polykání, které se projevují
dysfagií, sliněním a odmítáním potravy. V krevním
obraze je leukocytóza s posunem doleva, etiologickou
diagnózu potvrdí mikrobiologické vyšetření z dýchacích
cest a hemokultura.
Terapie – v  lehčích případech zabezpečíme rychlý
transport pacienta na specializované oddělení, v těžších
případech je potřebná okamžitá inhalace kyslíku, při
zastavení dýchání intubace. Dítě hospitalizujeme a ve
většině případů epiglotitida vyžaduje nazotracheální
intubaci, která obvykle trvá 24 hodin. Vzhledem k bakteriální
etiologii onemocnění jsou indikována širokospektrá
antibiotika intravenózně, převážně cefalosporiny
III. generace, ampicilin nebo amoxicilin, u kmenů pro-
22
PŮVODNÍ PRÁCE
dukujících β-laktamázu je vhodný amoxicilin s kyselinou
klavulanovou, ampicilin se sulbaktamem [1, 6].
Od roku 2001 bylo v ČR zavedeno povinné očkování
proti H. influenzae b dle vyhlášky č. 439/2000 Sb. jako
prevence závažných Hib infekcí. Nejprve byla dětem
podávána od 1. 7. 2001 tetravakcína ve čtyřech dávkách
podle očkovacího kalendáře, poté od 1. 1. 2007 hexavakcína
ve čtyřech dávkách podle očkovacího kalendáře.
Hexavakcína obsahuje antigen připravený z polysacharidu
pouzdra Hib [7, 8].
MATERIÁL A METODIKA
V rámci desetileté studie (2002–2012) byly odebrány
výtěry ze zevního zvukovodu ke kultivaci u pacientů
s akutní otitidou po paracentéze narozených před rokem
2001 a neočkovaných proti Hib, a dále u pacientů s akutní
otitidou po paracentéze povinně očkovaných proti Hib
a narozených od roku 2001. Sledovali jsme také počet
meningitid otogenního původu, epiglotitid a spektrum
původců v obou souborech. K průkazu Hib byla prováděna
kultivace na krevním agaru se stafylokokovou čárou na
mikrobiologii ve FN Brno. Transportní Amiesova půda
zaručuje přežití odebraných mikrobů po dobu nejméně
48 hodin.
Kritéria zařazení do studie:
1. Kontrolní soubor tvořily děti narozené před rokem
2001, neočkované proti Hib s akutní otitidou ošetřenou
paracentézou, sekundární otogenní meningitidou nebo
epiglotitidou.
2. Zkoumaný soubor tvořily děti narozené od roku
2001, očkované proti Hib s akutní otitidou ošetřenou
paracentézou, sekundární otogenní meningitidou nebo
epiglotitidou.
VÝSLEDKY
1. V kontrolním souboru neočkovaných dětí bylo odebráno
celkem 205 stěrů ze zevního zvukovodu. Stěry byly
odeslány na mikrobiologický rozbor. V souboru bylo 110
chlapců, 95 dívek ve věkovém rozpětí 0–7 let, průměrný
věk byl 2,9 let. Mikrobiologické vyšetření určilo původce
onemocnění, tyto výsledky byly dále vyhodnoceny.
U Hib neočkovaných pacientů s diagnózou akutní otitidy
byl vykultivován z mikrobiologických stěrů nejvíce
S.pneumoniae(45,5%),H.influenzaeb(26,3%),S.pyogenes(9,2%),
M. catarrhalis (6,8 %). U meningitid otogenního původu
byl zaznamenán u neočkovaných dětí ve 3 případech
S. pneumoniae, ve 2 případech S. pyogenes, 2x S. aureus,
v jednom případě byl vykultivován Hib a v jednom případě
H. influenzae non b. Sekundární meningitida vznikla
přestupem ložiska infekce z okolí při akutní mastoiditidě.
Ve všech případech byla provedena antromastoidektomie,
sanace primárního infekčního ložiska, v žádném případě
nedošlo k trvalému poškození sluchu. Epiglotitidu jsme
u neočkovaných dětí zaznamenali celkem v 8 případech.
Vždy byl původcem Hib. Bylo provedeno mikrobiologické
vyšetření, odebrána hemokultura.
2. Ve zkoumaném souboru očkovaných dětí bylo odebráno
celkem 201 stěrů ze zevního zvukovodu ke kultivaci.
V souboru bylo 106 chlapců a 95 dívek, ve věkovém rozpětí
0–7 let, průměrný věk byl 2,6 let.
U Hib očkovaných dětí u akutní otitidy byl vykultivován
nejvíce S. pneumoniae (52,2 %), S. pyogenes (10,4 %), S. aureus
Tab. 1. Výsledky testů pro otitis media acuta u očkovaných a neočkovaných dětí.
Patogen OD ND r. č. OD r. č. ND t0
P
1 S. pyogenes 21 19 0,1045 0,0927 -0,3987 0,6901
2 H. influenzae b 9 54 0,0448 0,2634 6,0834 0
3 S. pneumoniae 105 94 0,5224 0,4585 -1,2868 0,1982
4 M. catarrhalis 15 14 0,0746 0,0683 -0,2478 0,8043
5 S. aureus 18 6 0,0896 0,0293 -2,5751 0,01
6 P. aeruginosa 9 5 0,0448 0,0244 -1,1255 0,2604
7 S. agalactiae 0 2 0 0,0098 1,4038 0,1604
8 N. meningitidis 0 0 0 0
9 S. non AB 0 0 0 0
10 S. sk. C 3 1 0,0149 0,0049 -1,0248 0,3055
11 S. sk. F 0 0 0 0
12 S. sk. G 0 2 0 0,0098 1,4038 0,1604
13 H. parainfluenzae 9 3 0,0448 0,0146 -1,793 0,073
14 H. influenzae non b 12 5 0,0597 0,0244 -1,776 0,0757
15 H. species 0 0 0 0
Vysvětlivky použitých symbolů:
OD – počet očkovaných dětí
ND – počet neočkovaných dětí
r. č. OD, r. č. ND – relativní četnost očkovaných resp. neočkovaných dětí
t0
– hodnota testové statistiky
p – p-hodnota (porovnáváme ji se zvolenou hladinou významnosti , je-li p , nulovou hypotézu zamítáme na hladině významnosti )
232017, 72, Č. 1 ČESKO - SLOVENSKÁ PEDIATRIE
PŮVODNÍ PRÁCE
(8,9 %), M. catarrhalis (7,4 %), H. influenzae b byl u akutní
otitidy u očkovaných dětí v mikrobiologických výtěrech
zachycen pouze ve 4,4 %.
U meningitid otogenního původu u očkovaných dětí
byl vykultivován 4x S. pneumoniae, 2x S. aureus, 2x S. koaguláza
negativní, 2x S. agalactiae, 1x H. influenzae non b. Opět
byla provedena vždy sanace primárního ložiska – antromastoidektomie,
v žádném případě nedošlo k trvalému
poškození sluchu. Epiglotitidu jsme ve zkoumaném
souboru nezaznamenali.
Výsledky testů pro otitis media acuta u očkovaných
a neočkovaných dětí uvádíme v tabulce 1. Náš komentář
k tabulce 1: Hypotézu o shodné pravděpodobnosti výskytu
patogenu u očkovaných a neočkovaných dětí zamítáme
na hladině významnosti 0,05 pro H. influenzae b a S. aureus.
U meningitidy a epiglotitidy test nelze provést, protože
se u očkovaných dětí vůbec nevyskytly.
Statistické zpracování dat
Rozdílnost ve výskytu jednotlivých chorob způsobených
zkoumanými patogeny u  skupiny očkovaných
a neočkovaných pacientů byla zkoumána pomocí testu
homogenity binomických rozložení. Nulová hypotéza
tvrdí, že pravděpodobnost výskytu patogenu je u obou
skupin stejná, zatímco alternativní hypotéza tvrdí, že je
rozdílná. Hladina významnosti byla zvolena 0,05. Testy
byly provedeny pomocí systému STATISTICA Cz 12 (citace:
StatSoft, Inc. (2013). STATISTICA (data analysis software
system), version 12. www.statsoft.com).
DISKUSE
Vakcinace Hib změnila četnost invazivních hemofilových
onemocnění. Ve vyspělých zemích dosahovala
četnost onemocnění před vakcinací 20–60/100 000 dětí
ve věkové skupině do pěti let věku [10]. Po zavedení
imunizace konjugovanými Hib vakcínami došlo k poklesu
počtu invazivních hemofilových onemocnění o 70–95 %
[8, 9, 10]. Z našich výsledků vyplývá taktéž pokles počtu
hemofilových meningitid a epiglotitid u očkovaných
dětí. V  zemích, kde se vakcinace neprovádí, představuje
uvedený původce spolu s pneumokoky a meningokoky
klasickou triádu, která vyvolává většinu komunitně
získaných purulentních meningitid. U dětí do pěti let
věku byl Hib před očkováním původcem přibližně 60 –70 %
těchto onemocnění. Smrtnost hemofilové meningitidy
ve vyspělých zemích je poměrně nízká, závažné jsou ale
následky jako např. hluchota či jiné neurologické komplikace,
které se vyskytují přibližně u čtvrtiny nemocných.
Zavedená vakcinace od roku 2001 snížila celkovou
nemocnost v průběhu pěti let na pětinu, nejvýrazněji
klesala ve věkové skupině 0–11 měsíců a 1–4 roky. V roce
2004 bylo hlášeno pouze 5 meningitid s úmrtím jednoho
čtyřletého neočkovaného dítěte [4, 11].
V našem souboru u očkovaných dětí nebyl zaznamenán
žádný případ meningitidy ani epiglotitidy způsobené
Hib. U neočkovaných dětí byl zaznamenán jeden případ
Hib meningitidy a 8 případů epiglotitidy ve sledovaném
období.
Již od roku 1992 byla v České republice dostupná konjugovaná
Hibvakcína. Individuální vakcinace nebyla příliš
využívána. Podstatný zlom nastal v červenci 2001, kdy
byla zahájena vakcinace kojenců proti Hib tetravakcínou
TETRAct-Hib (záškrt, tetanus, dávivý kašel, Hib). Od roku
2007 se používá hexavakcína (záškrt, tetanus, dávivý
kašel, Hib, přenosná dětská obrna, virová hepatitida B).
Hib do té doby byl původcem přibližně 90 % všech invazivních
hemofilových onemocnění [11, 15]. Přibližně 90 %
těchto invazivních onemocnění se objevuje u dětí do pěti
let věku. Děti do dvou měsíců věku jsou chráněny pasivním
přenosem mateřských protilátek. Věková distribuce
onemocnění souvisí s rychlou kolonizací nosohltanu dětí
uvedeným mikrobem, ve věku dvou let je již kolonizováno
přibližně 80 % dětí [12]. Z těchto důvodů vakcinace proti
Hib musí být časná, už před spontánní kolonizací dětí.
Po zahájení vakcinace je pozorován výraznější pokles
počtu invazivních hemofilových onemocnění až při 90%
proočkovanosti dětí ohrožených věkových skupin [12]. Ve
všech zemích byl během 2–3 let po zahájení vakcinace
pozorován strmý pokles počtu invazivních hemofilových
onemocnění, většinou až na 5 % původně hlášených
onemocnění.
Nejdelší zkušenosti s vakcinací mají ve Finsku, kde bylo
zahájeno očkování od roku 1988 až 1989, a ve Švýcarsku,
kde byla vakcinace zahájena v roce 1990 [13]. V průběhu
devadesátých let byla Hib vakcinace dětí postupně zavedena
ve většině rozvinutých zemí. Česká republika se mezi
tyto země zařadila v roce 2001. Imunita je podmíněna
přítomností baktericidních antikapsulárních protilátek,
které jsou získány transplacentárně, po prodělaném
onemocnění a po imunizaci [3].
ZÁVĚR
Na Dětské otorinolaryngologické klinice FN v Brně
bylo během desetiletého období (2002–2012) zhodnoceno
spektrum původců způsobujících akutní otitidy, sekundární
meningitidy otogenního původu a epiglotitidy
u dvou skupin pacientů. U dětí neočkovaných a u dětí
očkovaných proti Hib.
Z výsledků studie je patrný pokles výskytu Hib v mikrobiologických
nálezech u akutních otitid u očkovaných dětí
proti mikrobiologickým nálezům u dětí neočkovaných.
V souboru očkovaných dětí nebyl zaznamenán případ
meningitidy ani epiglotitidy způsobené Hib.
Očkování zaručuje téměř kompletní ochranu před závažnými
invazivními onemocněními – meningitidami
a epiglotitidami, a snižuje počet komplikací při běžných
ORL infektech.
Podpora FN Brno, podpořeno MZ ČR – RVO (FNB,
65269705).
LITERATURA
1. Máchalová M, Forstová G. Hrtan a průdušnice. Akutní zánět středouší.
In: Šlapák I, a kol. Dětská otorinolaryngologie. Praha: Mladá fronta, 2013:
54–56, 193–200.
2. Votava M. Hemofilové infekce. In: Votava M. Lékařská mikrobiologie speciální.
Brno: Neptun, 2003: 1–53.
3. Krbková L. Hemofilové infekce. In: Beneš J, a kol. Infekční lékařství. I. vyd.
Praha: Galén, 2009: 225–227.
4. Blechová Z. Hnisavé meningitidy nejmladších věkových skupin. Neurol
pro praxi 2006; 3: 131–133.
5. van de Beek D. Adjunctive dexamethasone in bacterial meningitis: a meta-analysis
of individual patient data. Lancet Neurol 2010; 3: 245–263.
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6. Šašinka M, Šagát T, a spol. Pediatria. Košice: Status s.r.o., 1998: 462–465.
7. Křížová P. Hemofilové vakcíny a jejich použití v České republice. Pediatr
praxi 2001; 2: 13–15.
8. Dagan R, Fraser D, Roitman M, et al. Effectivenes of a nationwide infant
immunization program agains H. influenzae b. Vaccine 1999; 17: 134–141.
9. Hobstová J. Hnisavé meningitidy v dětském věku. Praha: Galén, 1999.
10. Hobstová J, Petráš M, Kostřicová E, et al. Problematika meningitid
vyvolaných H. influenzae b v souvislosti s plošnou vakcinací proti
H. influenzae b. Klin mikrobiol inf lék 1998; 2: 70–73.
11. Křížová P, Lebedová V. Sledování selhání vakcíny proti H. influenzae b.
Praha: Zprávy CEM (SZÚ) 2003;12 (2): 76–77.
12. Křížová P, Lebedová V, Beneš Č. Vliv rutinní vakcinace v České republice
na výskyt invazivních onemocnění způsobených H. influenzae b. Klin
mikrobiol inf lék 2004; 3: 118–123.
13. McVernon J, Johnson PDR, Pollard AJ, et al. Imunologic memory in
H. influenzae b conjugate vaccine failure. Arch Dis Child 2003; 4: 379–383.
14. Peltola H. H. influenzae b disease and vaccination i Europe:leasons learned.
Pediatr Infect Dis 1998; 17: 126–132.
15. Zjevíková A, Rožnovský L, Křížová P, Lebedová V. Hemofilová meningitida
u řádně očkovaného dítěte. Klin mikrobiol inf lék 2004; 1: 22–24.
16. Chaudhuri A. EFNS guideline on the management of community-acquired
bacterial meningitis: report of an EFNS Task Force on acute bacterial
meningitis in older children and adults. Eur J Neurol 2008; 15 (7):
649–659.
Došlo: 14. 6. 2016
Přijato. 20. 12. 2016
MUDr. Gabriela Forstová
Dětská otorinolaryngologická klinika FN
Černopolní 9
613 00 Brno
e-mail: gpeskova@email.cz
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology
journal homepage: www.elsevier.com/locate/ijporl
Case Report
Post-myringotomy oto-liquorrhea in children - A case study and literature
review
Milan Uríka,b,∗
, Ivo Šlapáka,b
, David Laštovičkab,c
, Jana Jančíkováa,b
, Denisa Pavlovskáb,d
,
Dagmar Hošnováa,b
, Eliška Žarošskáb,e
, Michaela Ťoukálkováb,f
a
Department of Paediatric Otorhinolaryngology, Faculty of Medicine, Masaryk University Brno, Czech Republic
b
Faculty Hospital Brno, Czech Republic
c
Department of Paediatric Surgery, Orthopedics and Traumatology, Faculty of Medicine, Masaryk University Brno, Czech Republic
d
Department of Pediatric Radiology, Faculty of Medicine, Masaryk University Brno, Czech Republic
e
Department of Pediatric Infectious Disease, Faculty of Medicine, Masaryk University Brno, Czech Republic
f
Department of Paediatric Anaesthesiology and Intensive Care Medicine, Masaryk University Brno, Czech Republic
A R T I C L E I N F O
Keywords:
Myringotomy
Tympanic membrane
Complications
Oto-liquorrhea
Children
A B S T R A C T
Myringotomy is a surgical incision of tympanic membrane used mainly as therapy for acute otitis media. It is a
safe and simple procedure; however possible complications must be considered. In this article is described a case
study of a 2-and-a-half-year-old girl with a massive oto-liquorrhea following myringotomy.
1. Introduction
Tympanic membrane paracenthesis, or myringotomy, is defined as a
surgical incision of tympanic membrane. It is used in patients with
acute otitis media (AOM) with bulging tympanic membrane to immediately
drain the inflammatory content of tympanic cavity [1,3–5]. It
is also used in children with otitis media with effusion (OME) when
myringotomy is performed to drain liquid from tympanic cavity and
insert ventilation tube into tympanic membrane [2]. Myringotomy
seems to be a simple intervention; however it has its risks and several
possible complications have been described in literature. This paper
describes a case study of a two-and-a-half-year-old girl who underwent
myringotomy for AOM. A massive oto-liquorrhea occurred, the following
surgical revision revealed luxation of stapes with a profuse flow
of inner-ear liquid from oval window.
2. A case study
The two-and-a-half-year-old girl was admitted to out-patient ENT
office at 6 a.m., parents informed about several hours lasting pain of
right ear, not responding to ibuprofen. The girl was subfebrile (temperature
between 37,5 and 38 °C) and had signs of upper airway inflammation.
ENT specialist on call performed otoscopy which revealed
red, edematous and bulging right tympanic membrane, therefore he
decided to perform myringotomy at a site of its most prominent bulge posterior
inferior quadrant. Serous secretion drained from tympanic
cavity following the myringotomy. Myringotomy was performed standardly
(as is usual in our country) with no signs of complications. A
father firmly holds a child sitting on his lap, fixes child's arms, a nurse
fixes head (this is the standard fixation procedure used in our country)
and a doctor (ENT specialist) performs otoscopy and myringotomy,
using a myringotomy knife. He used the standard otoscope with magnifying
glass. The girl was discharged, the parents were informed about
the necessity of regular lavage of auditory canal, snot sucking, administration
of analgetics and nose drops. The follow-up was scheduled
after two days. However, the parents returned with the girl six hours
later, stating the girl had fever and was vomiting, did not want to eat
nor drink and was sleepy. The ENT specialist decided the girl had to be
hospitalised, blood tests were performed (they were normal) and
treatment with antibiotics was initiated (Ceftriaxon 1.5 g á 24 hours).
The girl underwent neurological, ophthalmic and infectological examinations
including lumbar puncture, all without significant findings.
Computed tomography (CT) examination of brain was performed revealing
pneumocephalus on a right side near tentorium, the air in the
vestibulum and in the internal meatus acusticus and liquid content in
middle ear cavity on right, the rest without pathology (Figs. 1 and 2).
https://doi.org/10.1016/j.ijporl.2018.06.013
Received 23 May 2018; Received in revised form 8 June 2018; Accepted 9 June 2018
∗
Corresponding author. Department of Pediatric Otorhinolaryngology, Černopolní 9, 61300, Brno, Czech Republic.
E-mail address: doctor.urik@gmail.com (M. Urík).
Příloha 2: Urík M, Šlapák I, Laštovička D, et al. Post-myringotomy oto-liquorrhea in children - A case study
and literature review. Int J Pediatr Otorhinolaryngol. Dec 2018;115:153-155. doi:10.1016/j.ijporl.2018.06.013.
Because of the pathological liquid content in the tympani cavity, it was
not possible to sufficiently describe the ossicular chain and the position
of the stapes. The girl also suffered from a congenital impairment of
external ear - bilateral preauricular appendices, therefore a combined
developmental abnormality is expected. Profuse secretion of clear liquid
was observed from right ear, with suspicion on oto-liquorrhea
(Fig. 3). A sample was taken and examined on presence of beta-2transferin,
with a positive result. It is known that perilymph has a very
similar composition to that of cerebrospinal fluid. On day 5 of hospitalisation,
oto-liquorrhea did not tend to stop, therefore we decided to
perform explorative tympanotomy. Under general anesthesia tympanotomy
was performed. We observed the fluid leakage from the place
after myringotomy (posterior inferior quadrant) and revealing luxation
of stapes whose footplate was pulled into inner ear accompanied by
profuse flux of inner ear liquid (perilymph) through oval window.
Spontaneous closure of the perforation was not possible due to fragments
of stapes footplate. Extraction of stapes including footplate was
performed, oval window was closed with fat from auricle which was
compressed by placing tympanic membrane into its initial position,
malleus and incus were left at their positions (Fig. 4). Tympanic
membrane was compressed using tamponade of external ear canal by
Gelitaspon spheres. Immediately after compression, the flux of perilymph
stopped. During the surgery, neurosurgeon performed lumbar
puncture with drain insertion. During the following days of hospitalisation,
10 ml of cerebrospinal fluid every 4 hours was removed daily.
Extraction of Gelitaspon from external ear canal and extraction of
lumbar drain was performed on 7th day after the surgery. Hearing
examination using BERA (Brainstem Evoked Response Audiometry) was
performed with the result of severe hypacusis of right ear (response on
90dB). Oto-liquorrhea did not appear after the surgery. On tenth day
after the surgery, the girl was discharged, she has been coming for
regular visits, subjectively with no complaints, without nausea or ver-
tigo.
Fig. 1. CT scan of the temporal bone (coronar projection), air in the vestibulum
(blue), air in the internal meatus acusticus (green), communication between
vestibulum and internal meatus acusticus (orange). (For interpretation of the
references to colour in this figure legend, the reader is referred to the Web
version of this article.)
Fig. 2. CT scan of the temporal bone (axial projection): red – cochlea, yellow – stapes (on the right sight is the stapes much more anteriorly), green – oval window,
blue – handle of maleus. (For interpretation of the references to colour in this figure legend, the reader is referred to the Web version of this article.)
Fig. 3. Oto – liquorrhea, clear liquid in the external ear canal.
Fig. 4. Insertion of the fat to the oval window, oto -microscopy.
M. Urík et al.
3. Discussion
Myringotomy is a procedure with a long history in otology and is
used daily in patients all over the world. ENT books describe possible
complications of this seemingly simple procedure. These include injury
of jugular vein leading to massive bleeding [6], injury of internal carotid
artery [7], injury of the dura mater [8] and injury of ossicular
chain [9,10]. The most frequent injury is disruption of incudostapedial
joint [10]. Malleus and stapes are more stable than incus and therefore
less frequently injured [11]. According to available data, the most
frequent cause of ossicle bone injury is head trauma with fracture of
temporal bone [12]. No described case of luxation of stapes caused by
myringotomy was found (searched in Pubmed). In the case described in
this paper, standard myringotomy caused luxation of stapes with its
footplate being pressed towards inner ear and therefore blocked spontaneous
closure of perforation. Several methods of solution are described
for case of lesion of stapes and its footplate [13,14]. In our case,
extent of injury and intensity of perilymph flux made reconstruction
and fixation of stapes and its footplate impossible. Therefore, extraction
of stapes and closure of oval window by fat was performed. If we have
suspicion for cerebrospinal fluid or perilymph, we take a sample to
detection of beta-2-transferin. In our department, myringotomy is a
standard procedure used daily for treatment of AOM. Indications of
myringotomy include bulging tympanic membrane, otalgia and signs of
upper airways infection. Parents are informed of the procedure and
must agree with its performation. We standardly used the otoscope with
magnifying glass or microscope. In some countries myringotomy is
performed under general anesthesia to avoid this kind of complication,
but the procedure of fixation described above is the standard and safe
fixation procedure used in our country for many years. The typical
place for myringotomy should exclude the postro-superior quadrant to
avoid ossicles trauma. No similar complication is recorded in the history
of our department. The girl also suffered from a congenital impairment
of external ear - bilateral preauricular appendices, therefore a
combined developmental abnormality is expected, but no other abnormalities
of the middle or inner ear are visible on CT scans. The girl
had no other visible malformation or diseases. Pediatric examination
was normal.
4. Conclusion
Myringotomy remains one of the treatment options in acute otitis
media. The procedure leads to immediate pain relief, evacuation of
inflammatory content from tympanic cavity and decreases the risk of
development of extra and intracranial complications of AOM. An
important factor is that myringotomy may eliminate useless administration
of antibiotics. Although myringotomy is a quite safe and simple
procedure, possible complications must be thought of and myringotomy
should be performed carefully. A child with post-myringotomy complications
should be treated at a department with experience with
middle ear surgery.
Conflicts of interest
There is no conflicts of interest.
Acknowledgements
Supported by a project: MUNI/A/0814/2016 and SUp 9/16.
References
[1] P.H. Kaleida, M.L. Casselbrant, H.E. Rockette, et al., Amoxicillin or myringotomy or
both for acute otitis media: results of a randomized clinical trial, Pediatrics 87
(1991) 466–474.
[2] Richard M. Rosenfeld, Seth R. Schwartz, Melissa A. Pynnonen, et al., Clinical
practice guideline, Otolaryngology-Head Neck Surg. 0194-5998, 149 (2013),
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[3] Mark S. Grubb, David C. Spaugh, Treatment failure, recurrence, and antibiotic
prescription rates for different acute otitis media treatment methods, Clin. Pediatr.
49 (2010) 970–975.
[4] Clinical practice guidelines for the diagnosis and management of acute otitis media
(AOM) in children in Japan, Auris Nasus Larynx 39 (2012) 1–8.
[5] Morten Lindbaek, Norwegian consensus is that only children with recurrent episodes
of otitis media need antibiotics, BMJ 320 (2000) 182.
[6] R. Subotić, The high position of the jugular bulb, Acta Otolaryngol. 87 (2009)
340–344.
[7] Jochen P. Windfuhr, Aberrant internal carotid artery in the middle ear, Ann. Otol.
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Lek. List 9 (1954) 510–512.
[9] Jan Christoffer Luers, Karl-Bernd Hüttenbrink, Surgical anatomy and pathology of
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[10] A.N. Hasso, J.A. Ledington, Traumatic injuries of the temporal bone, Otolaryngol.
Clin. North Am. 21 (1988) 295–316.
[11] Produl Hazarika, Seema Elina Punnoose, John Victor, Rajeev Chaturvedi, Avulsion
of malleoincudal complex with dislocation: a rare ossicular chain injury by using an
indigenous claw-like ear hook with review of literature, Indian J. Otol. 20 (2014)
146.
[12] T. Saito, Y. Kono, Y. Fukuoka, H. Yamamoto, H. Saito, Dislocation of the incus into
the external auditory canal after mountain-biking accident, ORL J.
Otorhinolaryngol. Relat. Spec. 63 (2001) 102–105.
[13] K. Jakse, R. Jakse, Diagnosis and therapy of stapes fractures and luxations,
Laryngorhinootologie 81 (2002) 87–92.
[14] Stefan Delrue, Nicolas Verhaert, Joost van Dinther, Andrzej Zarowski,
Thomas Somers, Christian Desloovere, Erwin Offeciers, Surgical management and
hearing outcome of traumatic ossicular injuries, J. Int. Adv. Otol. 12 (2016)
231–236.
M. Urík et al.
Case Report
Pott’s puffy tumor: A rare complication of acute otitis media in child:
A case report§
Milan Urı´k *, Josef Machacˇ, Ivo Sˇlapa´k, Dagmar Hosˇnova´
Department of Paediatric Otorhinolaryngology, Faculty of Medicine, Masaryk University and Faculty Hospital, Brno, Cˇernopolnı´ 9, 61300, Czech Republic
1. Introduction
Acute otitis media is one of the most common diseases of
childhood and may be accompanied by severe complications.
Complications of acute otitis media are classified as intracranial
and intratemporal. Complications of acute otitis are rare in today’s
age of antibiotics, but may result in increased morbidity and
significantly increase the economic cost of treatment [1–5]. Fortunately,
life-threatening intracranial complications are rare today.
Intratemporal complications include acute mastoiditis, subperiosteal
abscess, facial nerve palsy, serous labyrinthitis and
suppurative labyrinthitis. Intracranial complications include sigmoid
sinus thrombosis, epidural or intracerebral abscess, meningitis,
petrositis, and otitic hydrocephalus [6–8]. Pott’s puffy tumor
(PPT) is a subperiosteal abscess located above the lesion of cranial
osteomyelitis [9]. It is a rare disease often leading to intracranial
spread of infection. The majority of published Pott’s puffy tumor
cases has been described in adolescents and adults, overwhelmingly
in the frontal area where PPT develops as a complication of
frontal sinusitis or trauma, or rarely as a complication of maxillary
sinusitis, postoperative craniotomy, dental abscess or respiratory
tract infection. As of 2014, only 36 pediatric cases have been
reported in the literature, mostly as a complication of sinusitis
(according to MEDLINE). To date, only one case of PPT has been
described in the literature as a complication of latent mastoiditis
in an adult [10], and one case of PPT as a complication of acute
mastoiditis in a 10-year-old child [11].
2. Case report
A 6-year-old Caucasian boy, previously healthy, was referred to
our department by his practitioner for 2 days of fever (40 8C), pain
and discharge from the left ear. The patient was taking a macrolide
antibiotic for 2 days. During the examination, normal otoscopic
findings were observed on the right side, while on the left side,
the patient had purulent discharge from the external ear, but the
retroauricular and preauricular areas were without acute pathology,
with no redness or swelling, with signs of upper respiratory
tract infection. Meningeal signs were negative, without cervical
lymphadenopathy. The patient had no history of ear diseases. Pus
samples were taken for cultivation and the antibiotics were
changed to cephalosporins; the ear started clearing locally. The
next day we made a follow-up evaluation, which revealed
persistent fever (38–39 8C), the boy was tired, tearful, reported
headache and persistent secretion from the left ear. We completed
X-rays of the temporal bone (projection according to Stenvers
and Schuler) to rule out mastoiditis, which was not confirmed.
Further examination was carried out after 3 days; the patient’s
condition did not improve, and moreover, the boy developed soft
painful swelling in front of the left auricle, in the zygomatic area.
The patient was admitted to the hospital for intravenous antibiotic
(ATB) therapy. He had the following baseline results on admission:
International Journal of Pediatric Otorhinolaryngology 79 (2015) 1589–1591
A R T I C L E I N F O
Article history:
Received 17 June 2015
Accepted 19 June 2015
Available online 29 June 2015
Keywords:
Pott’s puffy tumor
Acute otitis media
Childhood
A B S T R A C T
To describe a rare case of Potts’ puffy tumor (PPT) in the zygomatic area, which developed as a
complication of acute otitis media in a 6-year-old child. To date, only one case of PPT has been described
in the literature as a complication of latent mastoiditis in an adult, and one case of PPT as a complication
of acute mastoiditis in a 10-year-old child. Urgent surgical intervention, including evacuation of the
purulent lesion, removal of inflamed soft tissue and osteolysis of the involved bone, and
antromastoidectomy, intravenous treatment with broad-spectrum antibiotics, including G+, GÀ,
anaerobes and fungi, and local therapy.
ß 2015 Elsevier Ireland Ltd. All rights reserved.
§
Supported by project of Masaryk University, Brno: MUNI/A/1157/2014 – Acute
and chronic otitis media in child.
* Corresponding author. Tel.: +420 532234225; fax: +420 532234440.
E-mail address: docttor.urik@gmail.com (M. Urı´k).
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology
journal homepage: www.elsevier.com/locate/ijporl
http://dx.doi.org/10.1016/j.ijporl.2015.06.029
0165-5876/ß 2015 Elsevier Ireland Ltd. All rights reserved.
Příloha 3: Urík M, Machač J, Šlapák I, Hošnová D, Jančíková J, Pavlovská D, Hošnová D, Žarošská E,
Ťoukálková M. Pott's puffy tumor: a rare complication of acute otitis media in child: a case report. Int J Pediatr
Otorhinolaryngol. 2015;79(9):1589-91. doi:10.1016/j.ijporl.2015.06.029.
CRP 22 mg/l, WBC 7.8, and ESR 115/h; the patient was empirically
started on intravenous (I.V.) clindamycin, I.V. ceftriaxone and oral
ketoconazole. After 2 days, the swelling above the zygomatic area
resolved, the patient was afebrile, secretion gradually decreased.
On day 8, the patient’s condition deteriorated, and he re-developed
headaches, fever, and bulging above the zygomatic area. CT scan
was performed and revealed acute mastoiditis and epidural
empyema on the left side, with signs of temporal bone osteolysis
in the zygomatic area (Figs. 1–3). Under generalized anesthesia, we
made a wide incision at the site of bulging to evacuate large
quantities of purulent content that flew out under strong pressure,
and we found a bone defect that communicated with the epidural
space containing the abscess cavity; after the evacuation of pus,
the dura returned to its original position (Fig. 4). Further, we
performed antromastoidectomy to open the antrum, which was
obstructed by granulation. The patient was in the intensive care
unit for 24 h, and thereafter he was referred to the standard ward.
No causative agent was cultivated from the pus sample or blood
cultures. During the hospitalization, no abnormalities were found
from the blood counts, urine analysis and/or internal laboratory
tests. The patient was discharged home on postoperative day 10,
and the antibiotic treatment was continued for an additional
1 week. Since then, the patient has had no difficulties, otitis or any
cosmetic or neurological defects.
3. Discussion
In 1760, Sir Percival Pott was the first to describe pericranial
abscess as a sign of a more serious epidural abscess, which
originated as a result of head injury [12].
Since then, several cases of Potts’ tumor have been published.
Only a few cases were described in the post-antibiotic era,
predominantly in adolescents. Pott’s puffy tumor is commonly
described as a complication of sinusitis or injury in the frontal area.
About 36 pediatric cases of Pott’s puffy tumor have been reported
in the age of antibiotics. The majority of cases were reported in
adolescents. The youngest case published was a 3-year-old child
with Pott’s puffy tumor resulting as a complication of acute
sinusitis [13]. Akiyama reported that adolescents are the most
commonly affected group, and only 27 cases of Potts’ puffy
tumor in adults (age 21–81 years) had been published in the
English literature from 1990 until 2011, according to the Medline
database [14]. Ketenci et al. reported that only 11 cases out of
Fig. 1. Pott’s puffy tumor in the left zygomatic area, CT scan, coronal projection.
Fig. 2. Pott’s puffy tumor in the left zygomatic area, epidural empyema on the left
side, with signs of temporal bone osteolysis, CT scan, coronal projection.
Fig. 3. Pott’s puffy tumor in the left zygomatic area, epidural empyema on the left
side, with signs of temporal bone osteolysis, CT scan, axial projection.
Fig. 4. Wide incision at the site of bulging to evacuate large quantities of purulent
content that flew out under strong pressure.
M. Urı´k et al. / International Journal of Pediatric Otorhinolaryngology 79 (2015) 1589–15911590
62 published in the English literature were in adult patients [15]. It
has been generally known that male patients are affected more
frequently [16].
Acute otitis media can cause intracranial or intratemporal
complications. Intracranial complications most commonly result
from extension of the inflammatory process from the mucoperiosteal
lining of the mastoid air cells to the brain cavity. The
inflammation further develops in the brain area, lateral sinus,
epidural, subdural and subarachnoid spaces. In many cases, the
inflammation spreads through the dehiscent bone of the tegmen
tympani or antrum, via vascular connections directly to the lateral
sinus, through the superior petrosal sinus, vascular anastomosis,
caroticotympanic canaliculi, pericarotid venous plexus, cavernous
sinus, through the dehiscent bone of the tympanic cavity, through
the endolymphatic sac, a fistula in the otic capsule, or may
develop due to ostitis of the sinodural angle or petrous apex. If the
infection enters the meninges, it may result in the development of
pachymeningitis. The dura is very resistant to any noxious agents
and adheres to the bone in response to an inflammatory irritation,
which can prevent further propagation of the infection. Large
extradural abscesses can cause compression of the temporal lobe
or even compromise the squama of the temporal bone or occipital
bone and form a subperiosteal abscess known as Pott’s puffy tumor
[9]. Some authors believe that abscess formation occurs in children
due to insufficient ossification of the bones, but this was not
confirmed in our case. We did not observe any structural
abnormalities. In our case, no infectious agents were cultured
from the pus sample or blood cultures; apparently optimal
antibiotics were chosen given the rapidly improving condition
of the patient. A patient with a Pott’s puffy tumor can exhibit
considerable variability of symptoms and the disease itself may be
masked by other symptoms. The common symptoms are ear pain,
headaches, fever, signs of respiratory tract infection, vomiting,
itching, tension in the skin of the scalp and others [17]. Ear
discharge may or may not be present; completely intact or only
mildly hyperemic eardrum have been reported in the literature, or
intracranial complications may be the only signs of mastoiditis
[11]. Our patient had fever, signs of respiratory infections, and
symptoms of acute unilateral otitis, but no signs of acute
mastoiditis were initially present. Standard laboratory tests
revealed elevated sedimentation rate, only a slight elevation of
CRP levels and normal blood leukocyte count. X-rays of the
temporal bone at the onset of the disease showed no signs of acute
mastoiditis, and subsequent CT scans due to deterioration of the
patient’s condition revealed acute left-sided mastoiditis, left-sided
epidural empyema and signs of thinning or osteolysis of the
temporal bone in the zygomatic area. CT scan confirmed the
diagnosis and proved to be very successful in this case. After
diagnosing the Pott’s puffy tumor, surgical intervention is required
with immediate evacuation of the pus from both intracranial and
extracranial areas. Soft tissues affected by inflammation and bone
affected by osteomyelitis must be removed. In our facility, we also
perform paracentesis that provides drainage from the middle ear
and antromastoidectomy. After the surgery, intensive antibiotic
therapy should continue, optimally based on the results of pus
sample cultures. Typical pathogens responsible for the development
of Pott’s puffy tumor are beta-haemolytic streptococci,
Streptococcus pneumoniae, Haemophilus influenza, and less frequently,
bacteria of the genus Bacteroides and Proteus [13]. In our
case, we failed to detect any causative agent from the pus or
blood cultures, and we have tried to cover the spectrum of G+ and
GÀ bacteria including anaerobes and fungi. During the hospitalization,
the patient was treated with triple combination therapy:
clindamycin (lincosamide antibiotics to cover G+ bacteria and
anaerobes), ceftriaxone (a third generation cephalosporin to cover
GÀ bacteria) and ketoconazole (an imidazole antifungal to cover
the molds and yeasts). Clindamycin is generally very effective in
osteomyelitis.
4. Conclusion
Acute otitis media in children is one of the most common
diseases that can be currently very well diagnosed and effectively
treated. In some cases, however, it can lead to serious or even lifethreatening
complications such as meningitis, brain abscess,
cerebral sinus thrombosis, and others. Pott’s puffy tumor is a rare
complication and its diagnosis may not always be easy. We believe
the most appropriate imaging technique is CT scanning. Once the
diagnosis is made, urgent surgical intervention is required with
sufficient evacuation of the purulent lesions, inflamed soft tissue
and the affected bone. Targeted antibiotic therapy is equally
important, and we should cover the full spectrum of potential
pathogens, unless we know the causative agent. Early diagnosis
and adequate treatment significantly improve the success of
treatment. Interdisciplinary cooperation of an otolaryngologist,
neurologist, infection specialist, and neurosurgeon is of crucial
importance.
Conflict of interest
The authors declare that they have no conflicts of interest.
References
[1] N.A. Goldstein, M.L. Casselbrant, C.D. Bluestone, M. Kurs-Lasky, Intratemporal
complications of acute otitis media in infants and children, Otol. Head. Neck Surg.
119 (1998) 444–454.
[2] D. Hyden, B. Akerlind, M. Peebo, Inner ear and facial nerve complications of acute
otitis media with focus on bacteriology and virology, Acta Otolaryngol. 127 (2006)
460–466.
[3] D.J. Kitsko, J.E. Dohar, Inner ear and facial nerve complications of acute otitis
media, including vertigo, Curr. Allergy Asthma Rep. 7 (2007) 444–450.
[4] E. Ozer, E. Sivasli, Y. Bayazit, A. Sirikci, A. Gok, S. Mumbuc, et al., Otogenic cerebral
venous infarction: a rare complication of acute otitis media, Int. J. Pediatr.
Otorhinolaryngol. 67 (2003) 1019–1021.
[5] C. Go, J.M. Bernstein, A.L. DeJong, M. Sulek, E.M. Friedman, Intracranial complications
of acute mastoiditis, Int. J. Pediatr. Otorhinolaryngol. 52 (2000) 143–148.
[6] M.A. Hafidh, I. Keogh, R.M. Walsh, M. Walsh, D. Rawluk, Otogenic intracranial
complications: a 7-year retrospective review, Am. J. Otolaryngol. 27 (2006)390–395.
[7] P.S. Mallur, S. Harirchian, A.K. Lalwani, Preoperative and postoperative intracranial
complication of acute mastoiditis, Ann. Otol. Rhinol. Laryngol. 118 (2009)
118–123.
[8] L. Migirov, Computed tomographic versus surgical findings in complicated acute
otomastoiditis, Ann. Otol. Rhinol. Laryngol. 112 (2003) 675–677.
[9] J. Skrˇivan, Otogennı´ nitrolebnı´ komplikace,(Otogenic Intracranial Complications,
Alberta Prague), 1993 (in Czech).
[10] D.P. Martin-Hirsch, S. Habashi, R. Page, A.E. Hinton, Latent mastoiditis: no room
for complacency, J. Laryngol. Otol. 105 (1991) 767–768.
[11] M. Akram Khan, Pott’s puffy tumor: a rare complication of mastoiditis, Pediatr.
Neurosurg. 42 (2006) 125–128.
[12] R.P. Babu, R. Todor, S.S. Kasoff, Pott’s puffy tumor: the forgotten entity, J.
Neurosurg. 84 (1996) 110–112.
[13] M. Gupta, E.H. Hamdi, B. Ravi, M. Vivek, Pott’s puffy tumour in a pre-adolescent
child, Int. J. Pediatr. Otorhinolaryngol. 68 (2004) 373–378.
[14] K. Akiyama, M. Karaki, N. Mori, Evaluation of adult Pott’s puffy tumor: our five
cases and 27 literature cases, Laryngoscope 122 (2012) 2382–2388.
[15] I. Ketenci, Y. Unlu, B. Tucer, A. Vural, The Pott’s puffy tumor: a dangerous sign for
intracranial complications, Eur. Arch. Otorhinolaryngol. 268 (2011) 1755–1763.
[16] S.E. Forgie, T.J. Marrie, Pott’s puffy tumor, Am. J. Med. 121 (2008) 1041–1042.
[17] E.S. Pender, Pott’s puffy tumor: a complication of frontal sinusitis, Pediatr. Emerg.
Care 6 (1990) 280–284.
M. Urı´k et al. / International Journal of Pediatric Otorhinolaryngology 79 (2015) 1589–1591 1591
2372016, 65, č. 4 OTORINOLARYNGOLOGIE A FONIATRIE
PŮVODNÍ PRÁCE
Antromastoidektomiev dětskémvěku
Urík M., Šlapák I., Machač J.
SOUHRN
Antromastoidektomie (AMT) představuje jeden ze základních
operačních výkonů v chirurgické terapii ušních
nemocí. Pokles AMT na pracovišti Kliniky dětské otorinolaryngologie
v Brně (KDORL) v posledních letech vedl
k vytvoření této studie. Cílem je analyzovat a vyhodnotit
soubor dětských pacientů s provedenou AMT v letech
1997-2013 (17 let). Ve sledovaném období byl zjištěn
pokles provedených AMT až o 82 %, obrácení poměru
akutních a plánovaných AMT ve prospěch plánovaných
operací, častější operace u chlapců, změna v zastoupení
mikrobiálních původců u akutní mastoiditidy (AM) pravděpodobně
v souvislosti se zavedením plošného očkování
polyvalentní pneumokokovou vakcínou v České
republice a výskyt tradičně popisovaných komplikací
AM i přes zavedení moderní antibiotické terapie.
KLÍČOVÁ SLOVA
antromastoidektomie, dětský věk, očkování
Klinika dětské otorinolaryngologie, Fakultní nemocnice Brno
Lékařská fakulta, Masarykova univerzita, Brno
SUMMARY
Urík M., Šlapák I., Machač J.: Antromastoidectomy
in Childhood
Antromastoidectomy (AMT) is one of the basic surgical
interventions employed in the therapy of ear
illnesses. The decreasing number of AMTs performed
at the Department of Paediatric Otorhinolaryngology
(DPORL), Brno, Czech Republic, in recent years has lead
to the publication of the present study. Its objective is to
analyse and evaluate a group of children patients after
AMT in the years 1997-2013 (a time-span of 17 years).
During that period, a decrease of as much as 82 per
cent in the number of performed AMTs occurred, as
well as a reversal in the ratio of acute and planned AMTs
in favour of the planned interventions, increasingly
frequent operations on boys, a change in the composition
of microbial causes of acute mastoiditis (AM) due
to the introduction of mass polyvalent pneumococcal
vaccination in the Czech Republic, and occurrence of
common AM complications despite the introduction of
modern antibiotic treatment.
KEYWORDS
antromastoidectomy, childhood, vaccination
PŮVODNÍ PRÁCE
ÚVOD
Antromastoidektomie (AMT) představuje jeden ze základních
operačních výkonů v chirurgické terapii ušních nemocí.
Cílem je dostatečná sanace zánětem postiženého systému
mastoidních sklípků a znovuobnovení dostatečné ventilace
středoušní dutiny přes aditus, tak, aby středoušní dutina
a dutina mastoidního výběžku tvořily jeden funkční celek.
Zprůchodnění aditu a obnovení ventilace středouší je zásadní
pro další vývoj zánětlivého onemocnění akutního i chronického.
Pokud dojde k obturaci v oblasti aditus ad antrum,
rozdělí se tento prostor na kompartmenty dva – tympanální
a mastoideální. Je zřejmé, že náchylnost oddělené tympanální
dutiny k podtlakovým změnám při uzavřené Eustachově
tubě bude v tomto případě větší (8). AMT se u dětí provádí
pouze v celkové anestezii. Řez se vede za boltcem, těsně za
úponovou rýhou, obnaží se planum mastoideum a zevní
plocha výčnělku bradavčitého. V případě rozpadu kosti se toto
ložisko odstraňuje frézou, dlátky a chirurgickými lžičkami.
Podle rozsahu nálezu a pneumatizace se trepanace rozšiřuje
z mastoidálního antra na výčnělek, za impresi esovitého
splavu, do šupiny spánkové kosti, jařmového výběžku a kosti
skalní. Postupuje se až na lamina interna. Kostěný zvukovod
a nadbubínková dutina zůstávají obvykle výkonem nedotčeny.
Vlastní bubínková dutina je drénována paracentézou.
Hojení po výkonu trvá asi týden. Sluch správně provedeným
výkonem není dotčen (7).
Indikace k provedení AMT v dětském věku představují stavy
akutní a chronické. Mezi akutní stavy patří akutní mastoiditida
(AM) s případnými komplikacemi. Indikaci k plánovanému
zákroku jsou chronické zánětlivé změny v oblasti
mastoidních sklípků, označované jako latentní mastoiditida,
které vedou k recidivám akutního středoušního zánětu nebo
se podílejí na vzniku sekretorické otitidy a dalších patologických
stavů. Indikace k výkonu se v jednotlivých zemích
liší. Některá pracoviště v současnosti preferují i u akutních
stavů konzervativní postup před chirurgickým řešením (1,
12, 15). Počet provedených akutních AMT podle některých
studií klesá v posledních létech v souvislosti se zavedením
Otorinolaryng. a Foniat. /Prague/, 65, 2016, č. 4, s. 237–240
Příloha 4: Urík M, Šlapák I, Machač J. Antromastoidektomie v dětském věku. Otorinolaryngologie a foniatrie.
2016;4(65):237-240.
238 OTORINOLARYNGOLOGIE A FONIATRIE 2016, 65, č. 4
PŮVODNÍ PRÁCE
očkování polyvalentní vakcínou proti pneumokokům (6, 17,
13). Cílem předkládané studie bylo analyzovat a vyhodnotit
soubor dětských pacientů s provedenou AMT na pracovišti
Kliniky dětské otorinolaryngologie v Brně (KDORL) v letech
1997 – 2013.
METODIKA
V rámci retrospektivní studie jsme prostřednictvím nemocničního
infomačního systému vyhledali a následně vyhodnotili
data o všech pacientech, kteří na KDORL podstoupili
AMT v letech 1997-2013. Do studie byli zahrnuti pouze pacienti
s prostou AMT, nebyly hodnoceny jiné typy sanačních výkonů,
jejichž součástí byla AMT.
VÝSLEDKY
Ze získaných dat jsme zjistili, že počet AMT u dětí rapidně
klesl. Zatímco v roce 1997 bylo provedeno 35 operací, v roce
2002 to bylo již 19 a v roce 2013 pouze 3 AMT (graf 1). Jedná se
o pokles o 82 %. Celkový počet operací v uvedeném období byl
187, z toho 90 akutních a 97 pro chronické onemocnění. Do
roku 2008 převládaly operace akutních stavů, od roku 2008
je patrné obrácení poměru ve prospěch operací plánovaných
pro chronické onemocnění ucha (graf 2). Průměrný věk pacientů
byl 6 let, operovali jsme děti ve věku 4 měsíců až 16 let.
Ve sledovaném období byl zachován poměr postižení mezi
chlapci a dívkami 3:2, chlapci byli tedy operováni častěji.
Průměrná celková doba hospitalizace činila 12 dní, u těžkých
postižení přesahovala 20 dní. Zjištění mikrobiálního původce
nemoci, která vedla k nutnosti AMT, se prokázalo v 58 %. Do
roku 2009 byl nejčastějším patogenem S. pneumoniae (31 %),
od roku 2009 byl hlavním patogenem shodně S. pyogenes
(13 %) a S. peumoniae (13 %), dále H. influenzae (11 %). Méně
často se uplatnily další bakterie (S. aureus, Pseudomonas
aeruginosa, Enterococcus a další). Procentuální zastoupení
mikrobiálních původců ukazuje tabulka 1. Výskyt a četnost
typických patologických nálezů a komplikací uvádí tabulka 2.
Pouze 12 % operovaných pacientů mělo v minulosti zavedené
ventilační trubičky v bubínku.
Tab. 1 Mikrobiální původce dle výsledků kultivace
Původce Do 2008 Od 2008
S. pneumoniae 31 % 13 %
S. pyogenes 10 % 13 %
H. influenzae 7 % 11 %
Negativní kultivace 52 % 32 %
Tab. 2 Patologické nálezy a komplikace
Obturace aditus ad antrum granulacemi 85 %
Subperiostální absces 13 %
Meningitida 8 %
Paréza n. VII 7,4 %
Perisinuózní absces 3 %
Trombóza sinus sigmoideus 2,5 %
Epidurální absces 1x
Labyrintitida 1x
Rhabdomyosarkom 1x
DISKUSE
Výrazný pokles provedených AMT na pracovišti KDORL se
neshoduje s některými zahraničními studiemi. Tyto studie
ze zahraničních pracovišť udávají vzestup AMT v posledních
letech, především z důvodu akutní mastoiditidy, uvádí
i vyšší počet intrakraniálních komplikací (2, 6, 14). Je nutno
poznamenat, že ve většině případů jde o sdělení ze zemí,
kde není zdaleka tak dobře dostupná péče otorinolaryngologa
jako v České republice, kde v současnosti připadá 1
otorinolaryngolog na 10 000 obyvatel. Česká republika se
tak řadí na 2. místo v počtu orl specialistů na počet obyvatel
v zemích Evropské unie (5, 16). Diagnostiku i terapii akutního
středoušního zánětu v těchto zemích provádějí převážně
pediatři. Jedním z faktorů poklesu operovaných dětí by mohla
být i porodnost v ČR. Dle Českého statistického úřadu došlo
k poklesu počtu narozených dětí od roku 1996 do roku 2004,
a to v průměru o 10 tisíc dětí ročně. Od roku 2005 je opět
pozorován vzestup porodnosti na hodnoty před rokem 1996
(tab. 3). Vzhledem k uvedeným počtům nepovažujeme tento
faktor za významný. Domníváme se, že pokles indikací k provedení
AMT u akutních stavů je podmíněn efektivní, včasnou
a racionální antibiotickou terapií, která je zároveň i prevencí
komplikací akutního středoušního zánětu. Důležitou roli zde
Graf 1 Počet provedených AMT na KDORL Brno v letech
1997–2013.
Graf 2 Poměr provedených AMT akutních (červeně) a plánovaných
(modře) v letech 2002–2013.
2392016, 65, č. 4 OTORINOLARYNGOLOGIE A FONIATRIE
PŮVODNÍ PRÁCE
hraje i dobrá dostupnost péče otorinolaryngologa v České
republice. V souvislosti s antibiotickou terapií, rutinně prováděnou
paracentézou u rozvinuté akutní otitidy a včasným
záchytem klesá i počet zánětlivých komplikací akutního
středoušního zánětu. Svou roli v tomto poklesu může hrát
i zavedení plošného očkování proti Haemophilus influenzae
typu B a pneumokokům, nedisponujeme však relevantními
daty, která by to prokázala.
Tab. 3 Přehled číselných ukazatelů porodnosti v ČR.
Rok Počet narozených dětí
2013 106 800
2012 108 576
2011 108 673
2010 117 153
2009 118 348
2008 119 570
2007 114 632
2006 105 831
2005 102 211
2004 97 664
2003 93 685
2002 92 786
2001 90 715
2000 90 910
1999 89 471
1998 90 535
1997 90 657
1996 90 446
1995 96 097
1994 106 579
1993 121 025
Různí autoři udávají průměrný věk pacientů v rozmezí 2,1 roku
až 2,65 roku (1, 11). Průměrný věk našich pacientů je 6 let.
Rozdíl je dán tím, že ve většině zahraničních studií převažuje
soubor pacientů s akutní AMT pro akutní mastoiditidu, kdy
je častěji postižená nižší věková skupina dětí. Snažili jsme
se pátrat po příčině většího počtu operovaných pacientů
mužského pohlaví, kdy poměr operovaných dětí KDORL činil
3:2 ve prospěch chlapců. Autoři většiny dostupných publikací
ale uvádějí, že rozdíl mezi pohlavími nepozorovali (6, 10).
Zjištění mikrobiálního původce se podařilo v 58 %. V obdobné
studii, provedené v roce 2007 na Klinice ušní, nosní a krční 2.
LF UK a FN Motol, autoři uvádějí objasnění patogenu v 57 %,
nejčastějším patogenem zde byl Streptococcus pneumoniae
(33 %), na druhém místě Haemophilus influenzae ve 25 %
a překvapivým výsledkem zde byla přítomnost Pseudomonas
aeruginosa v 12,5 % pozitivních výtěrů (8), což se v námi předkládané
studii nepotvrdilo. Pokles nálezu S. pneumoniae od
roku 2008, jako původce AM vedoucí k AMT, koresponduje
s celkovým poklesem invazivních pneumokokových onemocnění
u dětí v ČR (8). Dá se tak předpokládat efektivita
zavedení plošného očkování polyvalentní pneumokokovou
vakcínou, která je prokázaná i v jiných zemích (6, 13, 17).
V České republice jsou registrovány 4 vakcíny: PNEUMO 23,
PREVENAR, PREVENAR 13 a SYNFLORIX.
Vzhledem k neustále se vyskytujícím rizikům extra i intrakraniálních
komplikací akutního středoušního zánětu
s mastoiditidou (AM) u dětí, považujeme provedení AMT za
běžnou součást léčebného protokolu spolu s intravenózní
aplikací širokospektrých antibiotik a provedením paracentézy
ušního bubínku. Některá pracoviště v současnosti preferují
konzervativní postup. V případě AM se subperiostálním
abscesem doporučují tato pracoviště intravenózní podání
širokospektrých antibiotik, myringotomii a drenáž abscesu
punkcí. AMT je na těchto pracovištích indikována pouze
v případě, že jsou přítomny známky intrakraniálních
komplikací. Často neprovádějí ambulantní paracentézu
(1, 12, 15). Naše výsledky jednoznačně dokazují, že provedení
AMT u akutní mastoiditidy přináší pro většinu pacientů
významný benefit, urychluje proces hojení akutního
zánětu, minimalizuje riziko intrakraniálních komplikací
a recidiv akutního středoušního zánětu a představuje dobrou
prevenci vůči rozvinutí chronických zánětlivých změn
středouší a mastoidního výběžku. Mnohem více nejasností
je v definici latentní mastoiditidy i v následných indikačních
kritériích k provedení AMT u této diagnózy. Na našem
pracovišti považujeme latentní mastoiditidu za subklinický
infekční zánětlivý proces slizniční výstelky i kostních struktur
mastoidních sklípků při celistvém bubínku. Vyznačuje
se nejčastěji recidivujícím akutním středoušním zánětem.
V případě, že jsou změny pouze slizniční, přistupujeme
k provedení adenotomie (pokud je indikována) a zavedení
ventilačních trubiček do bubínku. Dítě sledujeme. Pokud
jsou na RTG nebo HRCT snímků patrné i změny kostních
struktur mastoidních sklípků, přistupujeme k provedení
antromastoidektomie. Naše zkušenosti ukazují, že v případě
pouze slizničních změn je provedení adenotomie a zavedení
TVT dostatečným léčebným postupem s velmi dobrým efektem
a benefitem pro pacienta. Postupně dochází k vyhojení
středního ucha i sliznice mastoidních sklípků, obnovuje se
ventilace středouší i mastoidu, a tím se brání dalšímu rozvoji
patologických procesů zde probíhajících. Pro pacienta se
jedná o nenáročný a málo rizikový léčebný proces.
ZÁVĚR
Ze získaných údajů lze jednoznačně pozorovat pokles provedených
AMT v posledních letech. Patrné jsou i změny
v mikrobiálním zastoupení nejčastějších původců především
akutních středoušních zánětů s následnou mastoiditidou.
Tento fakt dáváme do souvislosti se zavedením očkování
pneumokokovou vakcínou a narůstajícím počtem proočkované
populace dětí, co potvrzují i zahraniční studie. Pokles
u akutních stavů je dle nás podmíněn i efektivní, včasnou
a racionální antibiotickou terapií, která je zároveň i prevencí
komplikací akutního středoušního zánětu. Důležitou
roli zde hraje i dobrá dostupnost péče otorinolaryngologa
v České republice. V souvislosti s antibiotickou terapií, rutinně
prováděnou paracentézou u rozvinuté akutní otitidy
240 OTORINOLARYNGOLOGIE A FONIATRIE 2016, 65, č. 4
PŮVODNÍ PRÁCE
a včasným záchytem klesá i počet zánětlivých komplikací
akutního středoušního zánětu. Pokles plánovaných operací
u chronických středoušních zánětů lze dát do souvislosti
se zlepšením diagnostických procesů (otomikroskopie, CT,
MRI), včasném zachycení nemoci v příznivějších stadiích
i zavedení použití ventilačních trubiček v terapii recidivující
akutní otitidy i chronického středoušního zánětu. Nelze
zdůvodnit častější postižení mužského pohlaví v dětském
věku. Z uvedených skutečností se lze domnívat, že počet
dětských pacientů, kteří budou muset podstoupit provedení
AMT (akutní i plánované), bude v budoucnu v ČR i nadále
klesat. Z tohoto důvodu lze vyvodit, že je nutné soustředit
tyto pacienty na vyšší specializovaná pracoviště, která mají
dostatek zkušeností a trénovaného operatéra. Pro mladé
lékaře bude beze sporu těžší praktický nácvik této operace
v klinickém provozu. Je namístě, aby vznikaly laboratoře
pro výuku této operativy při klinických pracovištích, kde
bude možné trénovat na kadaverech. Rovněž lze jednoznačně
doporučit očkování dětí pneumokokovou vakcínou, alespoň
u rizikových pacientů. Pokles výskytu pneumokokových
otitid od zavedení očkování v ČR je zřejmý.
Podpora projektu: Práce vznikla za podpory projektu
MUNI/A/1266/2015.
LITERATURA
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mastoiditis in children: A 10 - year Retrospective and Validated
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I.: Pneumokokové infekce u dětí a jejich prevence. Http://zdravi.e15.cz
[online]. 2012, č. 5 [cit. 2014-01-26]. Dostupné z: http://
zdravi.e15.cz/clanek/postgradualni-medicina/pneumokokove-in-
fekce-u-deti-a-jejich-prevence-464715
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Bernáldes, P. C.: Intratemporal complications from acute otitis
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Adresa ke korespondenci:
MUDr. Milan Urík
Klinika dětské otorinolaryngologie,
Fakultní nemocnice Brno
Jihlavská 25
625 00 Brno
e-mail: docttor.urik@gmail.com
Histological analysis of retraction pocket pars tensa of tympanic
membrane in children
M. Urík a,
*, P. Hurník b,c,d
, D. Žiak b,d
, J. Machacˇ a
, I. Šlapák a
, O. Motyka e
, J. Vaculová b,c
,
J. Dvorˇácˇková b,c,d
a Department of Paediatric Otorhinolaryngology, Faculty of Medicine, Masaryk University and University Hospital Brno, Czech Republic
b Department of Pathology, University Hospital Ostrava, Czech Republic
c
Department of Pathology, Faculty of Medicine, University of Ostrava, Czech Republic
d CGB laboratory, Ostrava, Vítkovice, Czech Republic
e Nanotechnology centre, VŠB-Technical University of Ostrava, Czech Republic
A R T I C L E I N F O
Article history:
Received 1 April 2016
Received in revised form 1 May 2016
Accepted 5 May 2016
Available online 12 May 2016
Keywords:
Retraction pockets
Histological analysis
Cholesteatoma
Children
A B S T R A C T
Aims: Histological and histochemical analysis of retraction pocket of pars tensa of tympanic membrane
in children. Identification of morphological abnormalities in comparison with a healthy tympanic membrane
as it is described in standard textbook. Identification of signs typical for cholesteatoma and support
for a retraction theory of cholesteatoma formation.
Study design: A prospective study analysing 31 samples of retraction pockets taken during surgery.
Departments: University Hospital, Children’s Medical Centre
Methods: Samples of retraction pockets were processed by a standard process for light microscopy, stained
by haematoxylin-eosin. Van Gieson’s stain was used for differential staining of collagen, Verhoeff’s stain
for elastic fibre tissues, Alcian blue for acidic polysaccharides and PAS (Periodic Acid Schiff) method for
basement membrane polysaccharides.
Results: The following findings were observed in the samples of retraction pockets: hyperkeratosis (100%),
hypervascularisations (100%), subepithelial fragmented elastic fibres (96%), myxoid changes (87%), subepithelial
inflammatory infiltration (84%), rete pegs (71%), papilomatosis (71%), intraepithelial inflammatory
cellularizations, (48%), intraepithelial spongiosis (16%) and parakeratosis (3%). No basement membrane
continuity interruptions were observed. Thickness of retraction pocket, thickness of epidermis, occurrence
of rete pegs and frequency of fragmented elastic fibres was higher in a Grade III stage RP than Grade
II stage RP (according to Charachon).
Conclusion: Morphological abnormalities in the structure of retraction pockets in comparison with a healthy
tympanic membrane were described. The changes are typical for a structure of cholesteatoma (these changes
are common in matrix and perimatrix), supporting retraction theory of its origin. Our observations show
that it is inflammation that probably plays a key role in the pathogenesis of retraction pocket. The frequency
of some of the changes increases with the stage of retraction pocket (II–III according to Charachon).
Basement membrane continuity interruptions are not typical for retraction pockets.
© 2016 Elsevier Ireland Ltd. All rights reserved.
1. Introduction
Tympanic membrane retraction pocket (RP) is a localised area
of tympanic membrane retracted into tympanic cavity. Unlike a
healthy tympanic membrane, which is described as a relatively tough,
elastic membrane without a tendency to collapse, RP is flexible and
tends to collapse into the tympanic cavity. RP is characterised by
atelectasis of a part of tympanic membrane which is retracted into
the area of bony anatomical structures, such as incisura Rivini,
scutum or malleus [1]. RP can be located in all quadrants of pars
tensa of tympanic membrane; pars flaccida can also be affected. Both
parts can be affected simultaneously. RP is a result of a long-term
or relapsing negative middle ear pressure which is caused mainly
by a dysfunction of Eustachian tube [1–8]. On the other hand, there
are theories and hypotheses claiming that pathogenesis of RP includes
the presence of mesenchyme (as a residue of intrauterine
development) or an inflammatory reaction that followed otitis media,
mainly in a posterosuperior quadrant of pars tensa and in the area
Declaration: This article did not receive sponsorships and there are no competing
interests.
Supported by a project: MUNI/A/1266/2015.
* Corresponding author at: Department of Paediatric Otorhinolaryngology,
Faculty of Medicine, Masaryk University and Faculty Hospital, Cˇernopolní 9, 61300
Brno, Czech Republic. Tel.: +420532234225; fax: +420532234440.
E-mail address: docttor.urik@gmail.com (M. Urík).
http://dx.doi.org/10.1016/j.ijporl.2016.05.007
0165-5876/© 2016 Elsevier Ireland Ltd. All rights reserved.
International Journal of Pediatric Otorhinolaryngology 86 (2016) 213–217
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology
journal homepage: www.elsevier.com/locate/ijporl
Příloha 5: Urík M, Hurník P, Žiak D, Machač J, Šlapák I, Motyka O,Vaculová J, Dvořáčková J. Histological
analysis of retraction pocket pars tensa of tympanic membrane in children. Int J Pediatr Otorhinolaryngol.
2016;86:213-7. doi:10.1016/j.ijporl.2016.05.007.
of pars flaccida, the two places typical for RP and atelectasis. Some
authors claim that without inflammation, retraction does not occur
[9,10]. These two areas are generally very fragile parts of the eardrum
and therefore tend to retract in the presence of negative middle ear
pressure.
Some histological studies describe interruptions of basement
membrane continuity in cholesteatoma, supporting retraction theory
of cholesteatoma, and only one paper (in the last 5 years by PubMed)
described the histological structure of RP or signs leading to formation
of cholesteatoma [11]. Sadé studied ear drums with
atelectasis and observed degeneration of middle collagenous fibrous
layer of ear drum caused, in his opinion, by an inflammatory process
in this area [7,9]. In their works, Sudhoff and Toss describe disruptions
of basement membrane of cholesteatoma. These disruptions
of basement membrane are localised in areas of growing rete pegs
and subepithelial inflammatory infiltration focuses [12,13]. Based
on these and other observations the authors support retraction
theory of cholesteatoma based on the mechanism of tympanic membrane
invagination followed by epithelial proliferation resulting in
hyperplasia, impairment of cell migration and production and accumulation
of keratin, all these being the basis for cholesteatoma
[12,13]. Their observations dealt only with cholesteatoma and up
till now only a few works have studied retraction pockets as a possible
pre-stage of cholesteatoma.
Our study presents a histological and histochemical analysis of
retraction pocket of pars tensa of tympanic membrane (RP) in children.
It describes morphological abnormalities of RP in comparison
with a structure of a healthy ear drum and signs typical for cholesteatoma.
Also, it aims to prove or disprove hypothesis that RP
involves disruptions of basement membrane.
2. Methods
We performed a prospective study during which 31 RPs of pars
tensa of tympanic membrane were taken during standard planned
surgeries in children. These were RPs grade II and III according to
Charachon, with no signs of progression to cholesteatoma. The
indications for surgery were: hypoacusis related to RP, recurring infections
or otomicroscopically observed for disease progression. The
criteria for exclusion included RPs with clinical signs of cholesteatoma
which could not be totally extracted. In all cases, after RP
excision, tympanic membrane was reconstructed using chondroperichondrial
graft from tragus.
All patients underwent pre-operative assessment including
history, otomicroscopy examination, tympanometry and pure tone
audiometry.
RPs were divided into two groups according to Charachon classification
(II – RP controllable, fixed, III – RP uncontrollable, fixed).
RPs stage I (controllable, not fixed) are not operated [5].
After being taken, all samples were oriented and put on a stripe
of a foam fixing material and put into a histologic chamber so that
the outer (ear canal) part was upwards and the inner (middle ear)
part downwards in the block. The samples were then placed into
4% buffered formalin. After fixation (24–48 hours) the samples were
cut into max. 5 mm wide blocks. Fragments were divided into halves
and positioned on the cutting line. All tissue blocks were then dehydrated
in a Tissue – Tek® VIP® 6 Vacuum Infiltration Processor
(Sakura). After being placed into paraffin wax, 2–4 μm thin sections
were prepared. All sections were stained with a basic
histological dye – haematoxylin and eosin. Van Giesson’s stain was
used for differential staining of collagen, Verhoeff’s stain for elastic
fibre tissues, Alcian blue for acidic polysaccharides and PAS (Periodic
Acid Schiff) method for basement membrane polysaccharides.
The samples were processed into 31 paraffin blocks and preparations
stained with H-E and 31 preparations stained with Alcian blue,
PAS, Van Giesson and Verhoeff methods. With an optical microscope
Olympus BX 45 with magnification 100× and 200×, HPF 400× was
used for the assessment. Two pathologists cooperated to assess the
histological preparations. Epithelium width was measured at ten
different places, then an arithmetic mean was calculated.
3. Results
The mean age of patients with RP was 10 years and we operated
patients from 6 to 18 years old.
The average sample length was 4955.4 μm. A bigger length of
grade III RPs was observed compared to grade II (p = 0.01293, two
sample T-test), which correlates with otomicroscopic findings. The
average sample thickness was 333.5 μm. With two extreme values
excluded, the grade III thickness is bigger than in grade II group
(p = 0.001005, two sample T-test).
3.1. Epidermis: outer epithelial layer
Outer epithelial layer of tympanic membrane composing of keratinized
stratified squamous epithelium was present in 31 samples.
The average thickness of epidermis was 46.3 μm. A difference in
thickness of epidermis was observed between grade II and III, the
average thickness of epidermis in grade II was 33.4 μm, and in grade
III it was 54.4 μm, which is a statistically significant (p = 0.001469,
two sample T-test). All 31 RP samples showed increased keratinization
(hyperkeratinization) of the outer layer of tympanic
membrane (Figs. 1 and 2). Two types of keratinization were observed
– lamellar (Fig. 1) and basket-weave (Fig. 2). Lamellar
hyperkeratosis was observed in all samples; 26 samples showed signs
of basket-weave hyperkeratosis. Parakeratosis (nuclei in keratinized
layer, indicates increased cell turnover) was identified in only
one case. Papilomatosis was observed in 71% of all samples.
3.2. Epidermis: Inner basement layer
In 22 cases we observed extensions of stratified squamous
epithelium (epidermis) in a form of rete ridges – rete pegs
(Fig. 3) – rete pegs projected into the middle layer of tympanic membrane.
These epithelial extensions were more frequent in RPs grade
III (18 out of 18 patients) compared to grade II (4 out of 13 patients)
(p = 3.547e-05, Fischer exact test).
Table 1 provides detailed descriptions of the observed abnormalities
of epidermis depending on the grade of RP.
Fig. 1. Basket-weave hyperkeratosis (HE, 200×).
214 U. M. et al. / International Journal of Pediatric Otorhinolaryngology 86 (2016) 213–217
3.3. Basement membrane
A PAS method was used to identify basement membrane. No
basement membrane disruptions were identified in RP samples
(Fig. 4).
3.4. Middle fibrous layer
In 27 cases we identified disruption of a double layer of
collagen fibres and its infiltration by neutral mucosubstances
(myxoid changes). The double layer of collagen fibres was well differentiated
in only 4 cases. There was no case with a complete
absence of lamina propria. The difference in the occurrence of myxoid
changes between grade II and III is not statistically significant.
Hypervascularisation of middle layer was present in all 31 cases,
with immature capillaries in 21% cases. Subepithelial elastic fibres,
both continuous and fragmented, were found in all cases (Fig. 5).
Fragmented elastic fibres were according to frequency evaluated as
+ and ++, and they were more frequent in group III (p = 0.01699,
Fischer exact test). Intraepithelial spongiosis (intracellular oedema)
was present in 15 cases and difference between group II (30%) and
III (55%) was observed. Subepithelial chronic inflammatory infiltrate
was present in 26 out of 31 samples and contained mainly
lymphocytes, histiocytes and plasma cells, rarely oesinophils (Figs. 3
and 4). No difference in frequency of occurrence between the RP
stages was observed. To compare our findings we attach a picture
of normal eardrum (Fig. 6).
Table 2 provides detailed descriptions of the observed abnormalities
of the middle and internal layer depending on the grade
of RP.
Length and thickness of retraction pocket, thickness of epidermis,
occurrence of rete pegs and frequency of fragmented elastic
fibres was higher in a group III compared to group II.
3.5. Inner epithelial layer
Inner epithelial layer was not present in 13 out of 31 samples.
This is similar to the appearance of cholesteatoma where the
Fig. 2. Lamellar hyperkeratosis (HE, 200×).
Fig. 3. Rete pegs, subepithelial inflammation, hypervascularisation (HE, 100×).
Table 1
Listing of the epithelial layer histological features as per the grade of retraction.
External epithelial layer Charachon II,
n = 13
Charachon III,
n = 18
Summary,
n = 31
Epithelial thickness, μm
Range 22,2 – 51,2 34,9 – 106,5
Mean 35,07 54,38
Rete pegs, n (%)
Present 4 (30) 18 (100) 22 (71)
Absent 9 (70) 0 (0) 9 (29)
Papilomatosis, n (%)
Present 9 (69) 13 (72) 22 (71)
Absent 4 (31) 5 (28) 9 (29)
Parakeratosis, n (%)
Present 1 (8) 0 (0) 1 (3)
Absent 12 (92) 18 (100) 30 (97)
Hyperkeratosis, n (%)
Lamellar 13 (100) 18 (100) 31 (100)
Basket – present 10 (77) 16 (89) 26 (84)
Basket – absent 3 (33) 2 (11) 5 (16)
Intraepithelial
spongiosis, n (%)
Present 0 (0) 5 (28) 5 (16)
Absent 13 (100) 13 (72) 26 (84)
Intraepithelial
inflammation, n (%)
Present 4 (30) 10 (55) 15 (48)
Absent 9 (70) 8 (45) 16 (52)
Fig. 4. Basement membrane (PAS, 200×).
215U. M. et al. / International Journal of Pediatric Otorhinolaryngology 86 (2016) 213–217
accumulated epithelial layers are gradually surrounded by perimatrix
consisting of fibrous tissue with inflammatory infiltrate with no other
layer.
4. Discussion
This paper describes histological structure of retraction pocket
of pars tensa of tympanic membrane in children. It points out the
typical signs for the structure of cholesteatoma and their occurrence.
Some of these signs occur with a higher frequency and extent
in higher grades of RP which supports a statement that RP is a prestage
of cholesteatoma. The main histological findings include
hyperkeratosis, hypervascularisation, subepithelial fragmented elastic
fibres, myxoid changes, subepithelial inflammatory infiltrate, presence
of rete pegs and papilomatosis. Interruption of a double layer
of collagen fibres (myxoid changes) was observed in 27 cases. Inner
epithelial layer was not present in 13 out of 31 samples. PAS method
saw no disruptions in basement membrane. These findings were
not surprising as basement membrane disruptions are rather typical
for malignancies, while RP and cholesteatoma are benign. Both length
and thickness of RP, thickness of epidermis and occurrence of rete
pegs were bigger in group III than in group II. This can be explained
by increased proliferation of epidermis and disruption of
middle layer of tympanic membrane where double layer of collagen
fibres is weakened and the structure of elastic fibres is disrupted
which does not necessarily lead to visible reduction of this layer.
The bigger thickness of the epidermis in stage III can be explained
probably by higher proliferative activity in this stage of RP as described
by Shunyu et al [11].
In most papers it is the long-term negative pressure in middle
ear caused by dysfunction of Eustachian tube that is considered the
main aetiological factor of RP [1–8]. Few papers point out a fact that
negative pressure in middle ear alone is not sufficient to cause retraction
[14,15]. Some authors consider the impairment of middle
collagen layer caused by inflammation an important factor leading
to retraction of tympanic membrane [7,9,14].
In our study subepithelial chronic inflammatory infiltrate was
present in 26 out of 31 samples, disruption of a double layer of collagen
fibres and its infiltration by neutral mucosubstances (myxoid
changes) in 27 samples. Lamina propria is composed of a complex
of collagen and elastic fibres and it is collagen fibres that influence
elasticity and plasticity of tympanic membrane [16]. The
frequency of fragmented elastic fibres was highest in grade III. The
double layer of collagen fibres was well differentiated in only 4 cases,
according to some authors this may be a case of reversible RPs which
may return to normal position owing to adequate therapy, for
example after implantation of a pressure equalising tube [11]. We
assume that fragmentation of elastic fibres and disruption of a double
layer of collagen fibres significantly impair the structure of tympanic
membrane and make it more vulnerable to the effect of
negative pressure in middle ear.
In some studies the description of cholesteatoma includes epithelial
pegs invading into perimatrix [12,13]. These areas contain
more proliferating cells. Similar pegs (rete pegs) were in higher frequencies
observed in our samples (22 out of 31 patients) and were
more frequent in grade III. In our opinion it is one of the signs of
cholesteatoma.
Fig. 5. Fragmented elastic fibres (Verhoeff, 200×).
Fig. 6. Normal eardrum.
Table 2
Listing of the middle collagen layer and the inner mucosal layer histological features
as per the grade of retraction.
Charachon II,
n = 13
Charachon III,
n = 18
Summary,
n = 31
Middle layer, n (%)
Present 13 (100) 18 (100) 31 (100)
Absent 0 (0) 0 (0) 0 (0)
Myxoid changes, n (%)
Present 12 (92) 15 (83) 27 (87)
Absent 1 (8) 3 (17) 4 (13)
Elastic fibres, n (%)
Present 13 (100) 18 (100) 31 (100)
Fragmented fibres, n (%)
Present 12 (92) 18 (100) 30 (96)
Absent 1 (8) 0 (0) 1 (4)
Subepithelial inflammation,
n (%)
Absent 3 (23) 2 (11) 5 (16)
1+ 7 (58) 12 (67) 19 (61)
2+ 3 (19) 4 (22) 7 (23)
Hypervascularisation
(capillaries), n (%)
Mature 13 (100) 18 (100) 31 (100)
Immature – present 9 (69) 17 (94) 26 (84)
Immature – absent 4 (31) 1 (6) 5 (16)
Internal layer, n (%)
Present 8 (62) 10 (56) 18 (58)
Absent 5 (38) 8 (44) 13 (42)
216 U. M. et al. / International Journal of Pediatric Otorhinolaryngology 86 (2016) 213–217
Frequency and extent of some of the observed signs increase with
the grade of clinical importance of retraction pocket. Hyperkeratosis,
papilomatosis, intraepithelial spongiosis and subepithelial
inflammatory infiltrate were present in both grades at similar rates.
Parakeratosis was observed in only one case. Both length and thickness
of retraction pocket, thickness of epidermis and occurrence of
rete pegs were higher in a group III than II; higher also was the share
of fragmented elastic fibres. Higher stage of RP leads to degeneration
of the middle layer of tympanic membrane, increasing thickness
of epidermis and formation of epithelial pegs, and basement membrane
remains intact; all these are signs typical for development
of cholesteatoma. Increasing occurrence of these histological signs
in relation to clinical progression of RP (II-III) agrees with a fact that
RP is a precursor of cholesteatoma.
This paper supports retraction theory of formation of cholesteatoma
in children. It provides evidence that it is not only negative
pressure that participates in the formation of RP, but also the key
role is inflammation and destruction of middle layer of tympanic
membrane making it more vulnerable to the effect of negative
pressure.
5. Conclusion
There are only a limited number of papers that deal with the
histological analysis of retraction pockets of the pars tensa of
tympanic membranes and its influence on the formation of cholesteatoma
in children. This work corroborates and supports some
previous papers. We described histological signs such as hyperkeratosis,
growing rete pegs, subepithelial inflammatory infiltrate,
hypervascularisation and degenerative changes of middle layer of
tympanic membrane with frequent occurrence of fragmented elastic
fibres. Our observations show that it is inflammation that probably
plays a key role in the pathogenesis of retraction pocket. Whether
inflammation is a cause or an effect of the disrupted homeostasis
of the tympanic membrane and middle ear mucosa secondary to
ventilation defects is still open to debate. Our findings give evidence
to a fact that RP is a progressive disease leading to formation
of cholesteatoma. On the contrary, in contrast to some other authors,
we did not observe disruptions of basement membrane continuity.
Based on our observations we consider retraction pocket of pars
tensa of tympanic membrane in children a pre-cholesteatoma stage.
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Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology
journal homepage: www.elsevier.com/locate/ijporl
Immunohistochemical analysis of retraction pocket pars tensa of tympanic
membrane in children☆,☆☆
Milan Uríka,∗
, Pavel Hurníkb
, Dušan Žiakb
, Josef Machača
, Ivo Šlapáka
, Oldřich Motykac,d
,
Petr Jabandžieve
a
Department of Paediatric Otorhinolaryngology, University Hospital Brno and Faculty of Medicine, Masaryk University Brno, Brno, Czech Republic
b
Department of Pathology, University Hospital Ostrava and Faculty of Medicine, University of Ostrava, Ostrava, Czech Republic
c
Nanotechnology Centre, VŠB-Technical University of Ostrava, Ostrava, Poruba, Czech Republic
d
ENET – Energy Units for Utilization of Non-Traditional Energy Sources, VŠB-Technical University of Ostrava, Ostrava, Poruba, Czech Republic
e
Department of Pediatrics, University Hospital Brno and Faculty of Medicine, Masaryk University Brno, Brno, Czech Republic
A R T I C L E I N F O
Keywords:
Retraction pocket
Cholesteatoma
Immunohistochemistry
Chidren
Pars tensa
A B S T R A C T
Aims: Immunohistochemical analysis of retraction pocket pars tensa of tympanic membrane in children.
Identification of signs typical for cholesteatoma and support of retraction theory of cholesteatoma. Study design:
a prospective study analysing 31 surgically removed retraction pockets.
Department: University Hospital, Children's Medical Centre Methods: Retraction pockets processed by a standard
process for immunohistochemical analysis. The observed findings were specified using antibodies CD45 LCA
(leukocyte common antigen), CD31 (platelet endothelial cell adhesion molecule), D2-40 (marker of lymphatic
endothelium), MMP9 (marker of degradation of connective tissue extracellular matrix) and Ki67 (cellular marker
of proliferation).
Results: All observed parameters except for MMP9 had a significantly higher incidence in retraction pocket stage
III compared to stage II according to Charachon.
Conclusion: We described immunohistochemical signs of retraction pocket pars tensa of tympanic membrane in
children resulting in cholesteatoma. All the observed signs occur in the structure of matrix and perimatrix of
cholesteatoma. A significantly higher incidence of all observed parameters except from MMP9 was proved in
retraction pocket stage III, unlike in stage II. This observation proves the fact that retraction pocket is a progressive
disease and is a procholesteatoma stage.
1. Introduction
Retraction pockets of tympanic membrane (RPs) are localized parts
of the tympanic membrane retracted toward the tympanic cavity. In
contrast to a healthy tympanic membrane, which can be identified as a
relatively tough, elastic membrane with no tendency to collapse, an RP
is flexible and tending toward collapse into the tympanic cavity. An RP
is characterized by atelectasis of a part of the tympanic membrane,
which is retracted into the area of such bony anatomical structures as
the incisura Rivini, scutum, or malleus [1]. RPs can occur in all quadrants
of pars tensa tympanic membrane as well as in pars flaccida. An
RP may also affect both parts of the tympanic membrane simultaneously.
An RP forms as a result of long-term or recidive underpressure
in the middle ear, which is itself caused mainly by Eustachian tube
dysfunction [2–4]. An alternative theory claims that an RP forms due to
mucosal changes [5,6]. There also are theories and hypotheses claiming
that the pathogenesis of RPs includes a presence of mesenchyme or an
inflammatory reaction related to otitis media, mainly in a posterior
superior quadrant of pars tensa and in the area of pars flaccida, two
sites typical for formation of RP and atelectasis. Some authors state that
retraction does not occur without inflammation [7,8]. These two sites
are generally prone to invagination in the presence of underpressure in
the middle ear. We do not know why these parts of the tympanic
membrane are most frequently affected.
There have been some histological studies describing breakage of
basal membrane continuity in cholesteatoma, thus supporting the
https://doi.org/10.1016/j.ijporl.2019.04.008
Received 8 January 2019; Accepted 9 April 2019
☆
Supported by the projects MUNI/A/1002/2018 and SUp 9/16. This paper was conducted within the framework of the project LO1404: Sustainable development
of ENET Centre.
☆☆
This study was presented at ESPO 2018 in Stockholm, 2–5 June 2018.
∗
Corresponding author. Department of Pediatric Otorhinolaryngology, Černopolní 9, 61300, Brno, Czech Republic.
E-mail address: docttor.urik@gmail.com (M. Urík).
Příloha 6: Urík M, Hurník P, Žiak D, Machač J, Šlapák I, Motyka O, Jabandžiev P. Immunohistochemical
analysis of retraction pocket pars tensa of tympanic membrane in children. Int J Pediatr Otorhinolaryngol.
2019;122:111-116. doi:10.1016/j.ijporl.2019.04.008
proliferation theory of cholesteatoma formation. An even smaller group
of works describes the histological and immunohistochemical structure
of RPs and potential signs leading to cholesteatoma formation [7]. Sadé
studied atelectatic tympanic membranes and observed degeneration of
the middle stromal layer of tympanic membrane that is, in his opinion,
caused by an inflammatory process [9]. In their works, Sudhoff and Tos
describe discontinuity of basal membrane in cholesteatoma. These basal
membrane disruptions are localized in the areas of growing rete pegs
and foci of subepithelial inflammatory infiltrate [10,11]. Based on these
and other observations, the cited authors support the proliferation
theory of cholesteatoma formation based on a mechanism of tympanic
membrane invagination followed by epithelial proliferation and resulting
in hyperplasia, cell migration malfunction, and accumulation of
keratin, all of which are responsible for cholesteatoma formation. Their
observations have dealt only with cholesteatomas, and there exist to
date only a few works examining the possibility that RP could be a
precondition of cholesteatoma.
Our study presents an immunohistochemical analysis of RPs in
children. It describes the abnormities of RPs and signs typical for cho-
lesteatoma.
2. Materials and methods
In this prospective study, 31 RPs taken during standard planned
surgeries in child patients were histologically examined. These included
RPs classified in stages II and III according to Charachon classification
and without signs of cholesteatoma progression. Indications for surgical
treatment of RPs were that the RPs were related to substantial conductive
hearing loss and relapsing infections or disease progression
proven by otomicroscopy. RP cases with clinical signs of cholesteatoma
and RPs that could not be removed completely were excluded from the
study. In all cases, reconstruction of tympanic membrane using a
chondroperichondrial graft from tragus followed upon RP removal. No
patients had active infections or were being treated with ototopical
antibiotics.
All patients underwent presurgical examinations including history,
otomicroscopy, tympanometry, and audiometry.
The RPs were divided into two groups according to Charachon
classification (II – RP controllable, fixed; III – RP uncontrollable, fixed)
[12]. It is standard procedure not to operate on RPs in stages controllable
and non-fixed.
Immunohistochemistry was performed on paraffin slices 2–3 μm
thick in Benchmark XT (Ventana Medical Systems, Oro Valley, AZ,
USA.). After blockage of endogenous peroxidase H2O2 (Ventana, iVIEW
kit 760-091) and buffer revitalization of CC1 antigen (Ventana, 950-
124), the following antigens were applied: CD31 (Dako Agilent M0823,
clone JC70A, 1:20, incubation of 30 min), D2-40 (Dako Agilent M3619,
clone D2-40, 1:50, incubation of 60 min), Ki67 (Dako Agilent M7240,
clone Mib-1, 1:100, incubation of 30 min), CD45 LCA (Dako Agilent
M0701, clone 2B11 + PD7/26, 1:100, incubation of 30 min), and
MMP9 (DAKO Agilent A0150, polyclone, 1:100, incubation of 15 min).
Subsequently, the iVIEW DAB Detection Kit (Ventana, 760-091), hematoxylin
(Ventana, 760–2021), and bluing reagent (760–2037) were
used.
Positivity of cytoplasm and cell membrane for antibodies against
D2-40 and CD31, cytoplasmic activity of MMP9 antibody, and membrane
positivity for CD45 LCA antibody were evaluated quantitatively
using a BX45 optic microscope produced by Olympus. Nuclear expression
was evaluated quantitatively using Ki67 antibody. We identified
all positive immunohistochemical signs in a sample and then the
number of elements was converted to a standard measure as number
per mm of RP length (i.e., length of the sample).
The following antigens and methodology were used for immunohistochemical
processing and evaluating:
CD45 LCA (leukocyte common antigen) – This is a protein in the
tyrosine phosphatase category, typical for white blood cells of all
categories. Absolute presence of elements with membrane positivity
was evaluated in the CD45 LCA preparations. Presence of the elements
was evaluated in both epithelium and stroma of tympanic membrane.
MMP9 (matrix metalloproteinase 9) – This is a metalloenzyme from
a category of peptidases and also is known as collagen type IV gelatinase
B. It is an enzyme participating in degradation of extracellular
matrix within fibrous tissue, and its positivity is observed in some invasive
carcinomas (e.g., invasive urothelial carcinoma of urinary
bladder). It participates in the degradation of epithelial basal membrane.
All MMP9-positive elements were evaluated within the observed
preparations.
CD31 (also known as platelet endothelial cell adhesion molecule
[PECAM1]) – Owing to its strong sensitivity and specificity, CD31 is
used as a marker of blood vessel endothelium. Within the evaluated
preparations, all vessel structures with endothelium unequivocally
showing strong positivity of both cell membrane and cytoplasm were
evaluated as positive.
D2-40 (podoplanin) – This is a membrane mucoprotein used as a
marker of lymphatic vessel endothelium. All lymphatic vessel structures
with endothelium unequivocally showing strong positivity of both cell
membrane and cytoplasm were evaluated as positive in the evaluated
preparations.
Ki67 – This is a nuclear marker of cell proliferation that is expressed
in the cell nucleus, but only in the late G1, S, and G2 phases of the cell
cycle. Among the evaluated preparations, all epithelia with strong nuclear
positivity were evaluated as positive. Positive epithelia were
further classified into two categories. The first category was represented
by a basal layer of squamous epithelium, where only the cells above the
basal membrane were evaluated. The second category contained all
elements of a squamous epithelium at the higher levels (suprabasally).
All nuclear-positive epithelia were counted suprabasally due to the nonhomogenous
and asymmetrical width of squamous epithelia in individual
samples.
The multivariate principal component analysis (PCA) approach was
adopted in order to assess the general trends and relationships between
the observed variables. PCA enables determining and investigating the
main sources of variance in the data, as well as correlations between the
data and the original variables and among the original variables in
multidimensional space. PCA biplot of the sample scores (points) and
variable loadings (vectors) according to the first two components of the
PCA. The samples were distinguished by colour according to their
Charachone group. No overlap in the confidence ellipses suggests an
overall difference between the samples of the two grades. The difference
is associated mainly with the first principal component (Dim 1),
grade III samples, thus tend to have higher values in most of the variables
than grade II samples. The analysis was performed in the R environment
using the FactoMineR package. The factor Grade was taken
as a supplementary qualitative variable, which means it was only a
descriptive variable and did not contribute to the multivariate model.
3. Results
In the outer layer of tympanic membrane (epidermis), we identified
a presence of Ki67 in the cells of both basal and suprabasal layers
(Fig. 1). These observations suggest an increase of proliferation activity
in squamous epithelia. We saw significantly increased presence of Ki67
in the suprabasal layer of epidermis in comparison with the basal layer
of epidermis (p = 0.035, Mann-Whitney U test). We also observed a
significant increase of Ki67 in stage III RPs in comparison with stage II
RPs (p = 0.022, Mann-Whitney U test).
Typical for blood vessel endothelium, we identified CD31 positivity
in the central layer (connective tissue) of tympanic membrane (Fig. 2).
These observations suggest a significant increase of CD31 in stage III
compared with stage II RPs.
We observed CD45 LCA positivity, which is typical for all categories
of white blood cells, in both the tympanic membrane's external
M. Urík, et al.
epithelial layer and its central connective tissue layer (Fig. 3). These
observations suggest a significant increase of CD45 LCA in stage III
compared to stage II RPs (p = 0.005, Mann-Whitney U test).
We observed D2-40 positivity, which is typical for lymphatic
vessels, in the central layer of tympanic membrane (Fig. 4). These observations
suggest a significant increase of D2-40 in stage III compared
to stage II RPs (p = 0.047, Mann-Whitney U test).
We observed MMP9 positivity in the central tympanic membrane
layer, thereby indicating a presence of degenerative processes in the
extracellular matrix of connective tissue (Fig. 5). We did not show a
significant increase of MMP9's prevalence in stage III RPs in comparison
with stage II RPs. Box and whisker plot of the data for each variable
examined are on Fig. 6.
One outlier was detected. That was patient (sample) number 13,
which contributed most (20.8%) to the construction of the first factor
plane. The outlier was thus removed from the further analyses.
Wilk's test indicates which variable factors are the best separated on
the plane (i.e., which ones best explain the distance between individuals).
The only qualitative variable assessed – Grade – was found
to illustrate the distances between the individuals well. It significantly
separates groups alongside the first dimension (first principal component),
accounting for 41.4% of variance in the data (p = 0.008).
The results for individuals and variables were plotted together in the
form of a biplot (Fig. 7), wherein the individuals were distinguished by
the supplementary variable Grade. In the biplot, individuals are displayed
as points and variables as vectors. The distances for both, then,
provide a visual representation of their similarity (in the case of individuals)
or correlation (in the case of variables). As is apparent, the
Fig. 1. Immunohistochemistry: Ki67 in area of rete pegs (200×).
Fig. 2. Immunohistochemistry: CD31, capillaries in middle layer of tympanic
membrane. (200×).
Fig. 3. Immunohistochemistry: CD45 LCA, inflammation (200×).
Fig. 4. Immunohistochemistry: D2-40, lymphatic vessels (200×).
Fig. 5. Immunohistochemistry: MMP9 in middle layer of tympanic membrane
(200×).
M. Urík, et al.
categories of the supplementary variable Grade are highly correlated
with this dimension (correlation coefficient = 0.97). This category itself
could therefore summarize the dimension 1, although it was not
used in constructing the model. It also is apparent from this graphical
output that the variables associated with the variable length were Ki67
and CD45_str, while those associated with thickness were mostly
CD45_epi and, to a lesser extent, MMP9. Correlations were positive in
all cases, meaning that the thicker the sample, the higher was the
CD45_epi positivity. Overall, patient (sample) Grade was positively associated
with all the observed variables. This association was further
confirmed by the analysis of covariance (ANCOVA) for the variables
CD45_str, CD31, D2-40, and Ki67 (p = 0.003, p = 0.02, p = 0.03, and
p = 0.004, respectively).
4. Discussion
This work investigates the immunohistochemical structure of retraction
pockets pars tensa of tympanic membrane. It has shown abnormalities
in RP occurring in the structure of cholesteatoma. Some of
the signs occur with higher frequency and with wider range in RPs of
higher stage, thus supporting the statement that RP is a pre-cholesteatoma
stage. The immunohistochemical findings in the group of RPs we
evaluated show an increased incidence of CD45 LCA, CD31, D2-40,
MMP9, and Ki67 positivity.
In most papers on this subject, the main etiological factor responsible
for RP formation is attributed to long-term underpressure in
the middle ear cavity caused by Eustachian tube dysfunction [2–4] and
mucosal changes of the middle ear [5,6]. A few studies point out that
underpressure alone is not enough to cause retraction [11]. Some authors
are of the opinion that damage of the middle collagen layer
caused by inflammation is an important factor leading to tympanic
membrane retraction [5,7].
A middle ear cholesteatoma is characterized by hyperproliferation
of keratinocytes and signs of chronic inflammation. It is able to grow
invasively and destroy the surrounding bones and middle ear structures.
Some studies have shown that autocrine and paracrine factors are
responsible for a cholesteatoma's invasive and proliferative behavior. It
has been shown that Ki67 expression is significantly increased in cholesteatoma
in comparison to normal, healthy skin. It is therefore responsible
for hyperproliferation [13,14]. A significant overexpression
of Ki67 also has been shown in chronic otitis with cholesteatoma in
comparison to other forms of chronic otitis without cholesteatoma [15].
This expression was found to be increased in superficial (suprabasal)
layers of cholesteatoma in comparison to basal layers of matrix [13].
We observed the same phenomenon. In the study od Dornelles C de C
(2009) is no significant difference in Ki67 expression in children's
cholesteatomas versus that seen in those of adults. Therefore, Ki67 is
probably not responsible for cholesteatoma aggressivity in children
[18]. We have shown that Ki67 activity is significantly greater in stage
III RPs than in stage II RPs. This observation suggests a slow proliferation
of RPs that leads to the formation of cholesteatoma.
CD31 is used for blood vessel detection. Studies comparing microvascular
density of cholesteatoma and the skin of the auditory tube
have shown significantly greater occurrence of CD31 in the cholesteatoma
structure [17]. Microvascular density of cholesteatomas is significantly
greater in children compared to adults [18]. We have shown
that CD31 positivity is significantly greater in stage III RPs than in stage
II RPs. This observation suggests that RPs have a progressive nature and
constitute another sign pointing to cholesteatoma formation.
LCA is used for the diagnosis of hematopoietic cell-related tumors.
Fig. 6. Box and whisker plot of the data for each variable examined: a - CD45 external layer of tympanic membrane, b - CD45 middle layer of tympanic membrane, c CD31,
d - D2.40, e − MMP9, f - Ki67 basal, g - Ki67 suprabasal, h - Ki67 total.
M. Urík, et al.
Significantly greater expression of CD45 has been shown in cholesteatomas
in comparison with other forms of chronic otitis without cholesteatoma
[19]. It has been shown that the numbers of activated T
lymphocytes and macrophage cells are high in the structure of a cholesteatoma,
while the number of B lymphocytes is low [10,20]. We have
shown that CD45 occurrence is significantly greater in stage III RPs than
in stage II RPs. This observation suggests that inflammation plays a key
role in the formation and progression of RPs.
Podoplanin (D2-40) is a useful marker for tumorous invasions of
lymphatic vessels. It has been shown that a reduction and degeneration
of lymphatic vessels occurs in cholesteatoma matrix. The immunohistochemical
study analyzing the occurrence and distribution of
lymphatic vessels in the structure of normal tympanic membrane has
shown that under normal conditions lymphatic vessels occur only
around the manubrium of the malleus and in tympanic membrane pars
flaccida. If tympanic membrane is perforated, rapid regeneration occurs
within the following several days along with proliferation of lymphatic
vessels into pars tensa, as well, mainly originating from the manubrium
area. Lymphatic vessels thus probably play an important role in spontaneous
healing of the perforation [21]. Only a small number of lymphatic
vessels have been identified in the edematous part of the cholesteatoma
perimatrix. Lymphatic vessels contained mucus and
consisted of degenerated endothelial cells. Edema of perimatrix is
caused by lymphatic vessel degeneration, and normal lymphatic vessels
were found in fibrous perimatrix. Connective tissue degeneration
caused by lymphatic edema was evident in perimatrix [16,19,22]. We
have shown that D2-40 occurrence is significantly greater in stage III
RPs than in stage II RPs. This observation suggests that RPs have a
progressive nature and constitute a sign of potential future cholesteatoma
formation.
MMP9 causes collagen type IV degeneration and plays a key role in
inflammation-related cell migration. It has been shown that MMP9
occurs more frequently in cholesteatoma matrix structure than in
healthy epidermis structure, and it plays an important role in the pathogenesis
of cholesteatoma [23]. Significantly greater MMP9 activity
has been found in children's cholesteatomas than in those of adults, and
it probably participates in the greater aggressivity of cholesteatomas in
children [18]. We have shown that MMP9's occurrence is not significantly
greater in stage III RPs than in stage II RPs. MMP9's occurrence
was observed in both RP stages with high frequency, and its
presence is among the signs pointing to cholesteatoma formation.
Our work supports the retraction theory of cholesteatoma formation.
It offers proof that not only middle ear underpressure but also
inflammation activity as well as weakening and destruction of the
middle layer of tympanic membrane play key roles in RP formation.
Tympanic membrane thereby becomes more susceptible to the underpressure,
as was shown also by our previous study [8].
We are aware that our work does not include a negative control
group, but it is not possible to obtain samples of a healthy child's ear-
drum.
5. Conclusions
There exist a limited number of works dealing with histological
analysis of RPs and their relationship to cholesteatomas in children. The
present work corroborates and supports findings reported in several of
our previous papers. We describe here changes on the immunochemistry
level, showing increased occurrence of Ki67 (hyperproliferation),
CD31 (angiogenesis), CD45 (inflammation), and D2-40
(lymphatic vessels) in stage III RPs compared to stage II RPs. This
suggests RP to be a progressive disorder leading to cholesteatoma formation.
In contrast to other authors, we observed no disruption in basal
Fig. 7. Results for individuals and variables together in a biplot (where individuals were distinguished by the supplementary variable Grade).
M. Urík, et al.
membrane continuity. We observed MMP9 frequently in both RP stages,
thereby indicating connective tissues degeneration. Based on our observations,
RPs in children can be considered a precholesteatoma stage.
Conflicts of interest
The authors declare they have no conflicts of interest.
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medicina
Review
Pathogenesis of Retraction Pocket of the Tympanic
Membrane—A Narrative Review
Milan Urík 1 , Miroslav Tedla 2 and Pavel Hurník 3,4,*
Citation: Urík, M.; Tedla, M.; Hurník,
P. Pathogenesis of Retraction Pocket
of the Tympanic Membrane—A
Narrative Review. Medicina 2021, 57,
425. https://doi.org/10.3390/
medicina57050425
Received: 6 April 2021
Accepted: 27 April 2021
Published: 28 April 2021
Publisher’s Note: MDPI stays neutral
with regard to jurisdictional claims in
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iations.
Copyright: © 2021 by the authors.
Licensee MDPI, Basel, Switzerland.
This article is an open access article
distributed under the terms and
conditions of the Creative Commons
Attribution (CC BY) license (https://
creativecommons.org/licenses/by/
4.0/).
1 Department of Paediatric Otorhinolaryngology, Faculty of Medicine of Masaryk University in Brno,
University Hospital Brno, 61300 Brno, Czech Republic; urik.milan@fnbrno.cz
2 Department of Otorhinolaryngology, Head and Neck Surgery, Comenius University, University Hospital,
85107 Bratislava, Slovakia; miro.tedla@gmail.com
3 Institute of Pathology, University Hospital Ostrava, 70852 Ostrava, Czech Republic
4 Institute of Animal Physiology and Genetics, Czech Academy of Sciences, 60200 Brno, Czech Republic
* Correspondence: pavel.hurnik@gmail.com; Tel.: +420-739067065
Abstract: Several theories describe the development of the retraction pocket of the tympanic membrane
(RP). Many authors suggest that the negative middle ear pressure is the main reason responsible
for developing this condition. A narrative review has been undertaken, and conclusions are drawn
reflecting a current knowledge with our new observations in the histological and immunohistochemical
study. Recent studies show the important role of inflammation in the development and
progression of RP. A review of the available literature shows that the inflammation plays a key role
in pathogenesis of the RP and its progression to the cholesteatoma. We support this statement with
our new results from histological and immunohistochemical analysis of the RPs.
Keywords: retraction pocket; tympanic membrane; pathogenesis; pathology; chronic otitis media;
cholesteatoma
1. Introduction
Retraction pocket (RP) of the tympanic membrane is a common entity, especially
in children. The danger of RP lies in ossicular chain erosion (Figure 1), cholesteatoma
formation and potentially life-threatening complications of cholesteatoma. No consensus
exists about the optimal treatment. We have a lot of new knowledge regarding pathogenesis,
but there are still many unknown factors that influence development of this condition.
Figure 1. Retraction pocket of pars tensa caused erosion of the incudostapedial joint. Malleus,
Erosion of the incudostapedial joint.
Medicina 2021, 57, 425. https://doi.org/10.3390/medicina57050425 https://www.mdpi.com/journal/medicina
Příloha 7: Urík M, Tedla M, Hurník P. Pathogenesis of Retraction Pocket of the Tympanic Membrane A
Narrative Review. Medicina. 2021;57(5):425. doi:10.3390/medicina57050425.
Medicina 2021, 57, 425 2 of 7
Epidemiological studies have shown that the retraction pocket is a relatively rare
disease and it largely affects the child population. In 1994, Stangerup published a study
where it examined 294 healthy children aged 5 to 16 years and found the occurrence of
retraction pockets in 26% in the area pars flaccida and at 3.7% in the pars tensa area [1].
The largest long-term study in a healthy population includes 7000 children under the
age of 10, when the prevalence of retraction pockets in the pars flaccida was 9.6 % and
pars tensa 7.9%. Most retraction pockets were classified as “mild”, only a small part
represented serious cases. This study also showed frequent bilateral disability retraction
pocket [2]. Relatively frequent occurrence of retraction pockets in the younger age group
of children up to 10 years was not found in the older age group. A study of children aged
8–18 years demonstrated that a significant proportion of tympanic pathologies found on
first examination healed spontaneously during a 10-year follow-up [3].
Histologically the normal tympanic membrane consists of an external epithelial layer,
middle layer, and internal mucosal layer. A healthy tympanic membrane is relatively tough
and elastic, this is due to the structure and properties of its middle layer.
The external layer is formed by the epidermis, which has a protective function and
is composed of stratified keratinizing epithelium. Keratinizing cells from the surface
gradually migrate from the center of the tympanic membrane to the periphery and further
outwards from the ear canal. This ensures self-cleaning ability [4]. The middle layer is
formed by connective tissue with a predominance of collagen fibers arranged in two layers
(outer radial and inner circular); elastic fibers also occur [5]. The thickness of this layer in
the pars tensa region ranges from 0.04 mm in the posterosuperior quadrant up to 0.09 mm
in the annulus region. It is mostly collagen type II and IV, which typically occurs in the
cartilage. Collagen fibers with admixture of fibers elastic determine the basic mechanical
properties of the drum. The middle layer of the eardrum also contains the delicate vascular
and nerve plexus [6]. The inner layer (lamina interna membranae tympani) of the eardrum
is formed by the middle ear cavity mucosa and its function is protection, metabolism and
nutrition of the middle layer of the tympanic membrane.
The three most used classification systems of retraction pockets are Sadé [7] (for pars
tensa RPs), Tos [8] (for pars flaccida RPs), and Charachon [9]. These systems grade the
severity of RP, but they do not explain the pathogenesis or possibility of progression of
retraction pocket. The association of severity of the retraction pocket in the development of
acquired cholesteatoma is the key point, however, there is no consensus existing staging
systems on retraction pockets or cholesteatoma in serving this purpose [10].
Retraction pocket is a localized area of the tympanic membrane invaginated into the
tympanic cavity. It is much more common in children than in adults. The incidence of
atrophy increases from 4% at age 4 years to 11% at age 16 years [11]. A normal tympanic
membrane is tough and elastic, but the retraction pocket is flexible and collapses into the
tympanic cavity. The place of weakness is most often located near the anatomical structures
such as incisura Rivini, scutum or malleus [12]. It can affect both parts of the TM, pars
tensa or pars flaccida. Many theories explain the pathomechanism of the development of
the retraction pocket.
2. The Eustachian Tube and Pathogenesis of the Retraction Pocket
One of the theories identifies the long-term negative middle ear pressure caused
by dysfunction of the Eustachian tube as a main reason causing development of the
retraction pocket. Normal TM has a certain capacity to buffer the pressure changes [13].
Ears that have anomalies in the volume and ventilation of the epitympanum may be
more susceptible to retraction pockets [14]. The healthy Eustachian tube equilibrates
the pressure between middle ear and nasopharynx. There are a lot of factors causing
dysfunction of the Eustachian tube (congenital malformations, mucosal oedema, adenoid
vegetations and many others). This ex vacuo theory is based on clinical studies and
animal experiments [15–17], but the results are contradictory. In many children after
adenoidectomy or ventilation tube insertion, retraction pocket or cholesteatoma [18,19]
Medicina 2021, 57, 425 3 of 7
developed. RP can occur even in the absence of negative pressure in the middle ear
while the ventilation tube is inserted in the TM. It is probably caused by the presence of
chronic inflammatory changes in the musculus tensor tympani, which retracts malleus [20].
Some studies point out a fact that negative middle ear pressure alone is not sufficient to
cause retraction [21–24]. In conclusion, there is no clear evidence that negative middle ear
pressure alone will cause invagination of normal tympanic membrane.
3. The Relationship of Otitis Media and Pathogenesis of the Retraction Pocket
It is true that the retraction pockets are commonly observed in children with recurrent
acute otitis media or otitis media with effusion. Sadé described that inflammation
as responsible for the degeneration of the lamina propria of the tympanic membrane [6].
Experimental study showed that the initial reason for the development of cholesteatoma
from the retraction pocket is local inflammation of the mucosa of the middle ear [25]. A
wide spectrum of theories describes the pathogenesis of acquired cholesteatoma [26] and
tries to clarify why keratinizing squamous epithelium starts growing from the outer layer
of the tympanic membrane into the cavity of the middle ear (metaplasia theory, migration
theory, papillary ingrowth theory). However, there is no clear evidence why the epithelial
cells prefer to migrate medially rather than laterally as is normal for cleaning of the healthy
tympanic membrane. Of course, some common middle ear pathogens increase proinflammatory
cytokines in monocytes, especially intracellular adhesion molecule–1(ICAM-1)
which mediate to adhesion of leukocytes to the vascular epithelium and the transition to
the extravascular space by the proinflammatory signaling, and soluble forms increase in
the middle ear effusion and serum due to autogenic infections. ICAM-1 was significantly
higher in patients with cholesteatoma, ossicular chain defects and tympanic membrane
retraction [27].
In 2018, Hüttenbrink published the self-healing hypothesis as the starting point of a
tympanic membrane retraction [28]. This idea interprets the horizontal migration of skin
into the middle ear cavity as a self-healing process, repairing an underlying inflammation
in the tympanic cavity, through the overgrowth and contact with immunologically active
tissue. This theory is supported by analogous phenomena that exist, e.g., the migration of
the omentum towards a local inflammation in the abdomen [29]. The findings in 209 s look
surgeries (from authors) provide the first explanation of the origin of retraction pockets
that is compatible with the various characteristics of original or recurrent cholesteatoma.
The process of changes in the TM structure may evolve to the cholesteatoma formation [30].
4. The Role of Inflammation in Pathogenesis of the Retraction Pocket
We conducted the histological and immunohistochemical analysis of 31 pars tensa
retraction pockets (stadium II and III by Charachon) in children. We identified the pathological
anomalies in the structure of external and middle layers of the tympanic membrane
compared with normal healthy tympanic membrane. We identified subepithelial inflammation
infiltrate in 86% of patients, disruption of a double layer of collagen fibers and
its infiltration by neutral mucosubstances in 87% and fragmented elastic fibers in 96%
(Figure 2) [31]. Degeneration of the middle collagenous fibrous layer caused by inflammation
was observed by Sade [1] or Yoon [32]. We also described hyperkeratosis (100%) and
ingrowing rete pegs (71%) [31]. In the outer layer of the tympanic membrane (epidermis),
we identified a presence of Ki67 in the cells of both basal and suprabasal layers. These
observations suggest an increase of proliferation activity in squamous epithelia. The process
of keratinocyte proliferation within cholesteatoma epithelium is important not only
for understanding the complex mechanisms of the pathogenesis of this disease, but also
to anticipate the possible development or recurrence of cholesteatoma [30–33]. We identified
CD45 LCA positivity, which is typical for all categories of white blood cells, in both
the tympanic membrane’s external epithelial layer and its central connective tissue layer
(Figure 3). We observed MMP9 positivity in the central tympanic membrane layer, thereby
indicating a presence of degenerative processes in the extracellular matrix of connective tis-
Medicina 2021, 57, 425 4 of 7
sue (Figure 4) [34]. MMP affect both inhibitory and stimulating effect on cell apoptosis [35].
Our observations show that it is inflammation that plays a key role in the pathogenesis of
the retraction pocket. This statement is supported by other studies [26,36–38]. Of course,
these anomalies commonly occur in the matrix and perimatrix of cholesteatoma, so our
observations support the retraction theory of development of cholesteatoma.
Figure 2. Fragmentation of the elastic fibers in the middle layer of tympanic membrane, histology of
retraction pocket, Verhoeff, (400×). External epithelial layer, Basal membrane, Fragmented
elastic fibers in the middle layer.
Figure 3. Immunohistochemistry–CD45 LCA, inflammation (200×). External epithelial layer,
Basal membrane, Rete peg (area of ingrowing squamous epithelium), White blood cells of all
categories (CD45 LCA+).
Medicina 2021, 57, 425 5 of 7
Figure 4. Immunohistochemistry–MMP9 in the middle layer of tympanic membrane (200×).
External epithelial layer, Middle layer of the tympanic membrane, MMP9+.
Degeneration of the lamina propria of the tympanic membrane is a long-term process.
Inflammation leads to destruction of the collagen and elastic fibers. Mediators of
inflammation lead to the release of collagenases and destroy the disulfate bridges. For
this reason, the tympanic membrane becomes thin and elastic properties are changed.
Weakened tympanic membrane loses its resistance to pressure changes. After the initial
insult and loss of strength, inflammation is no longer necessary for progression of the
retraction pocket. The problem of retraction pockets lies in the loss of original histological
and anatomical structure, therefore its inability to resist the negative middle ear pressure.
Based on this knowledge, it is very important to prevent and treat the inflammation
early. All children with recurrent acute otitis media or secretory otitis media should have a
comprehensive examination by a pediatrician and otorhinolaryngologist. We can perform
an adenoidectomy, insertion of the ventilation tube to the tympanic membrane or a special
treatment in case of immunodeficiency. Watch and wait strategy can be dangerous and
it can lead to damage of the middle ear ossicles or to development of cholesteatoma in
many cases.
5. Conclusions
All of these claims also support the basic mechanism of why an intact tympanic
membrane invaginates into the tympanic cavity and also support the retraction theory
of cholesteatoma. The first step in pathogenesis of the retraction pocket is affection of
the inflammation from the middle ear mucosa to the tympanic membrane. Inflammation
caused the pathological changes in the middle layer of the tympanic membrane: hypervascularisation,
degradation of the double layer of collagen fibers, fragmentation of the
elastic fibers and others. These changes lead to significant weakening of the tympanic
membrane, especially in pars tensa. This area of the tympanic membrane is more flexible
and less resistant to negative pressure in the middle ear in children with dysfunction of the
Eustachian tube. Inflammation is important for the development of the retraction pocket,
as we can see the presence of retraction in children with normal function of the Eustachian
Medicina 2021, 57, 425 6 of 7
tube. Therefore, prevention of inflammation is important in the treatment strategy of the
retraction pocket.
Author Contributions: Conceptualization, M.U. and P.H.; methodology, M.U. and M.T.; writing—
original draft preparation, M.U. and P.H.; writing—review and editing, M.T. All authors have read
and agreed to the published version of the manuscript.
Funding: This work was supported by the Ministry of Health, Czech Republic, Conceptual Development
of Research Organization (FNBr, 65269705), (NU20-08-00205 to PH lab) and by Masaryk
University(MUNI/A/1118/2020).
Institutional Review Board Statement: This article does not contain any studies with human participants
or animals performed by any of the authors.
Informed Consent Statement: Not applicable.
Data Availability Statement: Not applicable.
Conflicts of Interest: The authors declare no conflict of interest.
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Contents lists available at ScienceDirect
Am J Otolaryngol
journal homepage: www.elsevier.com/locate/amjoto
Experience with cholesteatoma behind an intact tympanic membrane in
children
Milan Uríka,⁎
, Andrea Kaliarikováa
, Josef Machača
, Michal Jurajdab
a
Department of Pediatric Otorhinolaryngology, University Hospital Brno and Faculty of Medicine, Masaryk University Brno, Brno, Czech Republic
b
Department of Pathological Physiology, Faculty of Medicine, Masaryk University Brno, Brno, Czech Republic
A R T I C L E I N F O
Keywords:
Cholesteatoma
Intact tympanic membrane
Children
A B S T R A C T
Introduction: To systematically investigate all surgeries for cholesteatoma behind an intact tympanic membrane
at our department. To identify predictive factors that can help the surgeon to plan surgery, surgical techniques,
and follow-up treatment.
Material and methods: This retrospective study evaluates 21 child patients, who were operated in the period
2007–2017 on for cholesteatoma behind an intact tympanic membrane.
Results: A total of 202 primary operations were performed for cholesteatoma. In 21 cases (10,4%) there was a
cholesteatoma behind an intact tympanic membrane and in 11 (5,45%) cases of it there was the congenital
cholesteatoma. The most frequently affected area was the anterior-superior quadrant. The preoperative hearing
loss increased significantly with disease severity (I–IV by Potsic).
Conclusions: The classification system according to Potsic is sufficient and fully corresponds to the surgeon's
needs. It has been clearly shown that a higher CC stage is associated with worse postoperative hearing results.
1. Introduction
Cholesteatoma behind an intact tympanic membrane (CITM) is a
rather rare pathology defined by clear diagnostic criteria. CITM is most
commonly congenital (CC), but it may also be acquired. The best known
theory of acquired CITM is the inclusion theory (Tos) or metaplasia
theory of the middle ear mucosa (Sadé). The prevalence of CC is about
2–5%. Diagnostic criteria for congenital cholesteatoma were originally
defined by Derlack and Clemis [1], later amended by Levenson as white
matter present behind an intact tympanic membrane, with no history of
tympanic membrane perforation, ear discharge, or middle ear surgery.
Patients with external ear canal atresia, intra-membranous cholesteatoma,
and extensive cholesteatoma (large-scale cholesteatoma) are excluded.
The history of acute otitis without spontaneous perforation and
paracentesis does not exclude the patients [2–4]. Cholesteatoma behind
an intact tympanic membrane requires surgical treatment. Several
classification systems for CC were developed in order to maximise optimal
treatment results; the most commonly used one is the system by
Potsic et al. [5]:
Stage 1 single quadrant: No ossicular involvement or mastoid ex-
tension
Stage 2 multiple quadrant: No ossicular involvement or mastoid
extension
Stage 3 Ossicular involvement, no mastoid involvement
Stage 4 mastoid extension (regardless of findings elsewhere)
The aim of our work was to systematically investigate all CITM
surgeries at our department during the last 10 years. We wanted to
identify predictive factors that can help the surgeon to plan surgery,
surgical techniques, and follow-up treatment.
2. Methods
A retrospective study that was conducted between 1 January 2007
and 31 December 2017, at the Department of Pediatric
Otorhinolaryngology of University Hospital in Brno. At the beginning,
we identified all patients diagnosed with cholesteatoma. Anyone who
met the Levenson's diagnostic criteria was considered a CITM patient.
Medical records of these patients were then examined and a database
containing the following data was created:
- Demographic data: age, gender, and CITM location (right, left, bi-
lateral).
https://doi.org/10.1016/j.amjoto.2019.102379
Received 24 October 2019
⁎
Corresponding author at: Department of Pediatric Otorinolaryngology, Faculty Hospital Brno, Černopolní 9, 61300 Brno, Czech Republic.
E-mail addresses: urik.milan@fnbrno.cz (M. Urík), kaliarikova.andrea@fnbrno.cz (A. Kaliariková), machac.josef@fnbrno.cz (J. Machač),
mjuraj@med.muni.cz (M. Jurajda).
Příloha 8: Urík M, Kaliariková A, Machač J, Jurajda M. Experience with cholesteatoma behind an intact
tympanic membrane in children. Am J Otolaryngol. 2020;41(2):102379. doi:10.1016/j.amjoto.2019.102379
- History of disease: history of acute otitis, preoperative imaging
method (CT or MRI).
- Peroperative findings: CITM stage according to Potsic et al., quadrant
localisation, localisation in relation to middle ear bones, and
potential anatomical abnormalities.
- Procedures performed: type of primary surgery, bone removal required,
total number of surgeries (second and third look), and reconstruction
procedure type.
- Treatment: preoperative hearing performance (pure tone average) at
250, 500, 1000, 2000, and 4000 Hz in decibels, postoperative
hearing performance after reconstruction, CITM residue and its lo-
calisation.
- Complications: facial nerve injury, perceptual hearing loss, recurrent
infections, and others.
Selected important characteristics for each event are summarized in
Table 1
Residual cholesteatoma was defined as CITM finding at all sites,
except for the tympanic membrane and the external auditory canal,
which after the first surgery unintentionally kept tissue that was surgically
unobservable and detected during the next procedure.
We used descriptive statistics methods (median, standard deviation,
quartile interval), binomial test (sex, laterality), Spearman's correlation
(stadium of Potsic), Fischer exact test (residual disease) and Wilcoxon
test (auditory thresholds) to analyze the results.
3. Results
Between 2007 and 2017, a total of 202 primary cholesteatoma
surgical procedures were performed. In 21 cases (10.4%), it was cholesteatoma
behind an intact tympanic membrane, and 11 patients
(5.45% from all 202) met the stricter initial criteria for CC (without the
history of acute otitis). Out of the total number of 21 patients, there
were 12 boys (57.1%) and 9 girls (42.9%). The increased CITM incidence
in men in our cohort is not statistically significant (binomial
test, p = .331). The ratio of children and the number of operated ears
was 1:1. Cholesteatoma behind an intact tympanic membrane was
found 14 times on the left side and 7 times on the right side. The increased
CITM incidence on the left side in our cohort is not statistically
significant (binomial test, p = .095). There were no cases of bilateral
CC. During the reporting period, the number of surgeries per year was
1.9 on average. The average age of children was 8.17 years (SD +/−
4.33), the median age was 6.33 (quartiles 4.92 and 12.15). The average
age according to Potsic was 5 years in the stage I, 6.25 years in the stage
II, 7.9 years in the stage III, and 7.3 in the stage IV. We have not found
any age-related increase of cholesteatoma severity according to Potsic
(Spearman's correlation, coefficient 0.28, p = .227). History of acute
otitis was present in 10 patients (48%). Cholesteatoma stage was determined
during the surgery according to the criteria set by Potsic et al.
Stage III was predominant, with 11 (53%) cases of cholesteatoma behind
an intact tympanic membrane. Stage II followed, with 4 (19%)
cases. The number of stage I and stage IV patients was identical, 3
(14%) (Table 2). The most frequently affected area was the anteriorsuperior
quadrant (16 out of 21, ie 76%) – Figs. 1, 2. The posterior
upper quadrant was affected in 14 cases (67%). Single-quadrant involvement
was reported in 3 patients (14%), the age of the children was
2, 3, and 4 years. The primary surgical procedure was the retro-auricular
transmeatal approach according to Portman. All primary surgeries
were performed using the canal wall up technique; the canal wall down
Table 1
Characteristics of 21 ears in 21 patients.
Age, median (range), y Value
Age, category, No. (%), y 6 (3–17)
< 4 5 (24)
4 to 5 4 (19)
> 6 12 (57)
Sex, No. (%)
Male 12 (57)
Female 9 (43)
Disease laterality, No. (%)
Right 7 (33)
Left 14 (67)
Bilateral 0 (0)
Disease stage by Potsic, No. (%)
1 3 (14)
2 4 (19)
3 11 (53)
4 3 (14)
Before surgery hearing, median (range), db 31.4
Before surgery hearing category, PTA, No. (%)
0–20 db 3 (14%)
21–39 db 8 (38%)
> 40 db 10 (48%)
dB - decibel.
PTA - Pure Tone Average.
Table 2
Demographic characteristics of the patients with CC.
ID Age (Years) Side Gender Stadium Second look
1 14 L F III Y
2 3 R F IV N
3 14 R F IV Y
4 5 L F III Y
5 7 L F I Y
6 3 R F II Y
7 4 L M I N
8 8 L M III Y
9 15 L F II N
10 12 R M III Y
11 6 L M III Y
12 12 L M III Y
13 5 L M IV Y
14 3 L M II Y
15 4 L M II Y
16 17 L F III Y
17 6 R M III Y
18 7 R F III Y
19 4 R M I N
20 5 L M III Y
21 7 L M III Y
Fig. 1. Cholesteatoma behind an intact tympanic membrane in the typical localisation
– anterosuperior quadrant.
M. Urík, et al.
technique was chosen only in one case due to the extent of the disease.
All surgeries we start with microotoscopy, we look for the external ear
canal and tympanic membrane and we make a pre-tympanic incision
(localisation of this incision depends on the localisation of the cholesteatoma).
Then we continue with retroauricular incision (separately
dermis and muscular tissue and separately incision of the periosteum).
We identified posterior wall of the external meatus and make a tympanomeatal
flap. Next step is tympanotomy, we identify the localisation
and size of the cholesteatoma and we identify the important structures
as chorda tympani, ossicles and of course the facial nerve. If the cholesteatoma
growth into the mastoid, we provide the antromastoidectomy
and we remove step by step all cholesteatoma, if it is possible
and safe, we remove it in toto. At the end of the surgery we provide the
reconstruction of the tympanic membrane if it is necessary with tamponade
of the external meatus with silk strip and gealfoam. Four patients
underwent only one surgery without the need for a second look.
Second look surgery was performed in 17 patients (81%) within
7–13 months of first surgery (9 months on average). A third look was
required in 1 patient for suspected disease recurrence. Tympanic
membrane reconstruction was performed 19 times (90%): in 15 cases
(79%) using a chondro-perichondrial graft from the tragus and in 4
cases (21%) from temporal muscle fascia. Reconstruction of middle ear
bones was performed in 10 patients (48%). Autologous materials were
used in the reconstruction of the middle ear bones in 4 patients (columella
from anvil or cortical layer, cartilage interposition); biocompatible
material (TORP) was used in 6 patients. Residual disease was
found in 4 cases (19%). 3 cases of residual disease were children under
the age of 7, one case was older than 7 years. 3 patients were male, 1
was female. The presence of residual disease is not gender-related
(Fisher exact test p = 1.00). The left side was affected in 3 cases, the
right side in one case. The presence of residual disease is not related to
the affected side (Fisher exact test p = .624). In 3 cases, it was primarily
a stage III, one case was stage II. The mean age of patients with residual
disease was 7.25 years. One or more middle ear bones were collected in
10 (48%) patients. The preoperative hearing threshold was 31.4 dB on
average. Scattergram of preoperative hearing results shows Fig. 3.
Postoperative hearing performance (6 weeks after surgery) was available
in 85% of patients. The average PTA in this period was 29 dB HL.
The average PTA after the second look was 27.9 dB HL. Scattergram of
postoperative hearing results shows Fig. 4. Comparison of the auditory
threshold before and after the surgery, regardless of the stage of the
disease, shows improvement in post-operative hearing (Wilcoxon test
p = .055) – Fig. 5. When considering the individual stages according to
Potsic et al., the average PTA before and after the surgery was: stage I
(20/20 dB), stage II (22.7/20 dB), stage III (35.8/31.4 dB) and stage IV
(36.5/34.5 dB). The comparison of the auditory threshold before the
surgery shows statistically significant differences between the stages
according to Potsic (Kruskal Wallis ANOVA p = .029). The comparison
of the auditory threshold after the surgery shows statistically significant
differences between the stages according to Potsic (Kruskal Wallis
ANOVA p = .038). There were no severe complications such as facial
nerve palsy, cerebrospinal fluid leakage, perceptive hearing loss, or
poor healing of the surgical wound. Tympanosclerosis developed in one
case. The most common symptom of cholesteatoma behind an intact
tympanic membrane was hearing impairment. Conductive hearing loss
was present mainly in more advanced stages (Potsic stage III and IV).
Lower stages (I and II) were diagnosed predominantly by chance
(otoscopy during ENT examination of unrelated reasons - check-up after
otitis, examination of adenoid vegetation, etc.). In one case, we diagnosed
the disease based on transient peripheral facial nerve palsy. Due
to the extent of cholesteatoma (stage II) and the fact that we did not
find a clear damage to the facial nerve bone cover, we can assume that
facial nerve palsy was not directly related to the presence of cholesteatoma
in this case. We follow up all patients to 18 years of life.
4. Discussion
The CC classification system provides a unified view of the disease
and the comparison of patient populations, epidemiological data, surgical
findings and procedures, complications, and recurrences of CC.
Our aim was to find and compare the factors that are involved in the
onset of CC, the factors we find during the surgery and that may be
associated with residual disease risk. Some of our findings confirmed
the importance of CC classification according to Potsic et al. [5]. In our
study population, the preoperative hearing loss increased significantly
with disease severity (I–IV). While hearing is normal in stage I, stage IV
is associated with the progression to 36.5 dB. We also observed the
same correlation in hearing loss after surgery, which was again the
lowest at stage I and the most significant at stage IV. This observation is
confirmed by other authors [6]. Compared with available data from
other studies, the incidence of acute otitis before the surgery was very
similar – 48% [7]. Various studies indicate different involvement of
individual CC quadrants. In our group, the involvement was most
commonly present in the anterior-superior quadrant (76%), like in
many other studies [5–9]. This finding supports the theory of the CC
origin and progression published by Koltai in 2002 [10]. He described
the progression of the CC from the anterior-superior quadrant to the
posterior-superior quadrant, to the attic and to the mastoid. The final
stage of CC was discussed, which included a discharge from the ear of
the child, conductive hearing loss, perforation in the tympanic membrane
and non-traditional cholesteatoma localisations or an extensive
cholesteatoma filling the entire mastoid. We cannot provide further
information to this discussion as we have limited the choice of patients
by the presence of an intact tympanic membrane. An advantage to our
study is the fact that one surgeon performed the procedure in all the
patients during the monitored period. Hence, the findings and procedures
are very consistent. The risk of residual disease ranges from 34%
to 44% [5,7,8], but CC residuum was found in only 19% of patients in
our study population. Less than two surgeries per one cholesteatoma
have been performed on average, which is a very good result compared
to other studies. Especially when we consider that we prefer the canal
wall up at our site, which generally involves a higher risk of residual
disease. This fact proves that canal wall up in children with CC is optimal
to avoid problems that affect patients with open cavity. It provides
much higher quality of life for the child. One of the major factors
in our opinion is the fact that we prefer removing middle ear bones at
the time of the first surgery, if the ossicles are eroded or in contact with
cholesteatoma. This philosophy is supported by other authors [6].
Imaging methods play an important role in diagnostics, planning and
Fig. 2. Cholesteatoma behind an intact tympanic membrane in the typical localisation
– anterosuperior quadrant (white pearl).
M. Urík, et al.
post-operative monitoring. Preoperative CT scanning of the temporal
bone is considered a standard that allows to identify key anatomical
structures and also describe the extent and localisation of cholesteatoma.
A temporal bone CT scan was standard for all patients in our
population. Magnetic resonance imaging can be used for postoperative
monitoring of patients. There are many studies today that have confirmed
that MRI is a suitable method for detecting residual disease, thus
reducing the need for further surgery in patients [11–13]. We did not
perform MRI routinely in our CC patient group, but we are currently
introducing this method as a part of our protocol for monitoring patients
after cholesteatoma surgery based on very good experiences.
Fig. 3. Scattergram of preoperative hearing results.
Fig. 4. Scattergram of postoperative hearing results.
M. Urík, et al.
5. Conclusion
The classification system according to Potsic is sufficient and fully
corresponds to the surgeon's needs. It has been clearly shown that a
higher CC stage is associated with a worse postoperative hearing results
in the patient. Removing middle ear ossicles when they are eroded or in
contact with cholesteatoma appears to be the correct procedure, as
confirmed by the very small number of relapses in our group.
Ethics approval
The study was approved by Ethical committee of University
Hospital in Brno, number of protocol: 11-130219/EK.
Funding
Supported by project MUNI/A/1002/2018 (Masaryk University
Brno, Czechia).
Authors contributions
All authors declare that they participated in the preparation of this
study. Milan Urík provided the design of the study, analysis of the
clinical data and managed and controlled the study. Andrea Kaliariková
provided the analysis of the clinical and statistical data. Josef Machač
provided the analysis of the clinical data and consultation, he is the
main surgeon of these patients. Michal Jurajda provided the statistical
analysis of all data.
CRediT authorship contribution statement
Urík Milan: Conceptualization, Methodology, Writing - original
draft, Supervision. Kaliariková Andrea: Formal analysis, Writing original
draft, Resources. Machač Josef: Conceptualization,
Methodology. Jurajda Michal: Software, Validation.
Declaration of competing interest
The authors declare that they have no competing interests.
Acknowledgements
Not applicable.
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disease and hearing outcomes. Arch Otolaryngol Head Neck Surg
2012;138:280–5.
[7] Nelson M, Roger G, Koltai PJ, Garabedian EN, Triglia JM, Roman S, et al. Congenital
cholesteatoma: classification, management, and outcome. Arch Otolaryngol Head
Neck Surg 2002;128:810–4.
[8] Lazard DS, Roger G, Denoyelle F, Chauvin P, EN G ’dian. Congenital cholesteatoma:
risk factors for residual disease and retraction pockets—a report on 117 cases.
Laryngoscope 2007;117:634–7.
[9] Shirazi MA, Muzaffar K, Leonetti JP, Marzo S. Surgical treatment of pediatric
cholesteatomas. Laryngoscope 2006;116:1603–7.
[10] Koltai PJ, Nelson M, Castellon RJ, Garabedian EN, Triglia JM, Roman S, et al. The
natural history of congenital cholesteatomu. Arch Otolaryngol Head Neck Surg
2002;128:804–9.
[11] Rajan GP, Ranjeeta A, Lisa W, Rataphol CD, Jafri K, Zenia CH, et al. Preliminary
outcomes of cholesteatoma screening in children using non-echo-planar diffusionweighted
magnetic resonance imaging. Int J Pediatr Otorhinolaryngol.
2010;74:297–301.
[12] Patel B, Hall A, Lingam R, Singh A. Using non-echoplanar diffusion weighted MRI in
detecting cholesteatoma following canal wall down mastoidectomy - our experience
with 20 patient episodes. J Int Adv Otol 2018;14:263–6.
[13] Laske RD, Roth TN, Barath K, Schuknecht B, Huber AM, Roosli CH. The role of nonechoplanar
diffusion-weighted magnetic resonance imaging in diagnosis of primary
cholesteatoma and cholesteatoma recidivism as an adjunct to clinical evaluation.
Ann Otol Rhinol Laryngol 2018. https://doi.org/10.1177/0003489418800833.
[ref. 2018-11-01].
Fig. 5. Preoperative pure-tone average decibel (dB) hearing level as a function
of stage (p = .029) and postoperative hearing results (most recent postoperative
audiogram results) by initial stage from all patients.
M. Urík, et al.
12 OTORINOLARYNGOLOGIE A FONIATRIE 2017, 66, č. 1
PŮVODNÍ PRÁCEPŘEHLEDOVÝ ČLÁNEK
Urík M., Machač J., Šlapák I.
SOUHRN
Úvod: Článek prezentuje problematiku středoušních
rekonstrukcí v dětském věku. Poskytuje pohled na
možnosti operačního řešení, jak je provádíme na Klinice
dětské otorinolaryngologie Lékařské fakulty Masarykovy
univerzity v Brně.
Metodika: Retrospektivní studie, ve které bylo hodnoceno
89 pacientů ve věku 5 až 19 let, u kterých byla provedena
středoušní rekonstrukce v letech 2005 až 2012.
Závěr: Studie ukazuje, že i v dětském věku je možné
rekonstrukcí převodního aparátu dosáhnout dobrých
výsledů jak anatomických tak funkčních.
KLÍČOVÁ SLOVA
chirurgie, střední ucho, rekonstrukce, sluch, dětský
věk
SUMMARY
Urík M., Machač J., Šlapák I.: Middle Ear Reconstruction
in Child
This article presents the problems of middle ear reconstruction
in children. It provides insight into the
possibilities of surgical treatment, as performed at the
Department of Pediatric Otolaryngology, Medical Faculty
of Masaryk University in Brno. Here are the results of a
retrospective study in which we evaluated a group of
89 patients aged 5-19 years, who had undergone middle
ear reconstruction in the years 2005 to 2012. The study
shows that even in childhood, it is possible to achieve
good results data, both anatomical and functional.
KEYWORDS
surgery, middle ear, reconstruction, hearing,
childhood
Klinika dětské otorinolaryngologie LF MU a FN Brno
Otorinolaryng. a Foniat. /Prague/, 66, 2017, č. 1, s. 12–15
Středoušnírekonstrukceudětí
ÚVOD
Středoušní operace jsou operační výkony, jejichž cílem
je odstranit patologický proces ve středouší a následně
provést obnovení převodního systému středního ucha.
Rekonstrukce převodního systému s cílem co nejlepšího
sluchového zisku je obzvláště v dětském věku pro další
život pacienta zásadní. Existuje několik typů a klasifikací
středoušních rekonstrukcí. Na Klinice dětské otorinolaryngologie
v Brně (KDORL) používáme operační postupy
dle profesora Tose s velmi dobrými výsledky a sluchovými
zisky u dětských pacientů. Hlavním cílem středoušní
operace neboli tympanoplastiky je odstranění aktivního
patologického procesu, který probíhá v oblasti středního
ucha, zabránit jeho další recidivě a docílit zlepšení sluchu
(11). V případě chronického středoušního zánětu dochází
k poškození středoušních kůstek, a tedy i k poškození převodní
složky sluchu poměrně často (1, 10). Příčiny, které
vedou k poškození středoušních kůstek, se obecně dělí do 2
skupin: cholesteatom a ostatní formy chronického zánětu
středního ucha bez výskytu cholesteatomu. Mezi méně časté
příčiny poškození převodního systému středouší patří úrazy
a vývojové anomálie v oblasti středního ucha. Středoušní
operace začíná fází „sanační“. Cílem této fáze je odstranění
patologie z oblasti středního ucha a dostatečná „sanace“,
která minimalizuje riziko recidivy onemocnění, především
v případě cholesteatomu. Po dostatečné sanaci onemocnění
přistupujeme k fázi „rekonstrukční“, kdy se snažíme o obnovení
převodního systému středního ucha s cílem co nejlepšího
sluchového zisku pro pacienta. Rekonstrukční fáze
se provádí zpravidla až v tzv. druhé době, kdy provedeme
kontrolu, zda nedošlo k recidivě onemocnění (8). V případě
PŮVODNÍ PRÁCE
Graf 1 Typy středoušních rekonstrukcí a jejich počet.
Příloha 9: Urík M, Machač J, Šlapák I. Středoušní rekonstrukce u dětí. Otorinolaryngologie a foniatrie.
2017;66(1):12-15.
132017, 66, č. 1 OTORINOLARYNGOLOGIE A FONIATRIE
PŮVODNÍ PRÁCE
méně rozsáhlé patologie a dobrých anatomických i funkčních
podmínkách možno provést rekonstrukci i v první době
na závěr tympanoplastiky, po sanaci.
TERMINOLOGIE A KLASIFIKACE
V terminologii a klasifikaci středoušních operací i rekonstrukcí
vycházíme z pojmů a postupů profesora Tose (11).
Pokud operujeme pouze na bubínku s cílem uzavření perforace
a středouší je zcela intaktní, mluvíme o myringoplastice.
Pokud provádíme pouze rekonstrukci středoušních kůstek,
pak mluvíme o osikuloplastice. V případě, že se oba výkony
kombinují, jedná se o tympanoplastiku. K rekonstrukci
používáme materiály autologní i biokompatibilní.
TYPY STŘEDOUŠNÍCH REKONSTRUKCÍ NA KDORL
A) Autologní materiály
Tympanoplastika I. typu dleTose.
Jedná se o odstranění drobné patologie většinou z oblasti
epitympana (retrakční kapsa, drobný cholesteatom) a následnou
rekonstrukci bubínku chondroperichondriovým štěpem
z tragu. Středoušní kůstky jsou zcela intaktní.
Tympanoplastika II. typu dleTose
Představuje interpozici chybějícího člena do řetězu středoušních
kůstek mezi hlavičku třmínku a kovadlinku, eventuálně
bubínek. Provádí se na závěr sanační fáze v případě defektního
řetězce kůstek. Třmínek je zachován. K interpozici používáme
chrupavku (u malých defektů dlouhého výběžku kovadlinky),
méně často kost nebo zbylou část opracované kovadlinky.
Tympanoplastika III. typu dleTose
Je vložení „kolumely“ z kovadlinky mezi ploténku třmínku
a kladívko, eventuálně bubínek. Používá se v případě zachovalé
kovadlinky a poškozených ramínek třmínku, které
se odstraní. Kovadlinka se ofrézuje tak, aby spodní části
nasedla na ploténku třmínku a horní na rukověť kladívka,
eventuálně na bubínek.
Myringostapedopexe a neomyringostapedopexe
Provádíme transpozici bubínku přímo na hlavičku třmínku.
Předpokladem je malá vzdálenost mezi hlavičkou třmínku
a bubínkem a dobrá pohyblivost třmínku. V případě, že je
nutná i rekonstrukce bubínku. mluvíme o neomyringosta-
pedopexi.
Manubriostapedopexe
U tohoto výkonu nejdříve uvolňujeme manubrium kladívka
z bubínku až po umbo, pak následuje transpozice manubria
kladívka přímo na hlavičku třmínku. Používá se při destrukci
kůstek v epitympanu. Předpokladem je zachovalé manubrium
kladívka, dostatečná pohyblivost třmínku a výhodou je
opět malý prostor mezi bubínkem a třmínkem.
B) Biokompatibilní (umělé) materiály
PORP - Partial Ossicular Replacement Prosthesis
Protéza, která je vložena mezi hlavičku třmínku a bubínek.
TORP -Total Ossicular Replacement Prosthesis
Protéza, která je vložena mezi ploténku třmínku a bubínek.
Středoušní protézy mohou být zhotoveny z různých materiálů
(kov, keramika a jiné). My používáme již delší dobu protézy
titanové s dobrými výsledky.
METODIKA
Provedli jsme retrospektivní studii, ve které jsme hodnotili
soubor všech pacientů se středoušní rekonstrukcí, které
byly provedeny v letech 2005 až 2012. Jedná se o soubor 89
pacientů ve věku 5 až 19 let. Z dostupných informací získaných
z nemocničního informačního systému a operačních
záznamů jsme hodnotili věkové rozložení pacientů, pohlaví,
stranové postižení, primární patologii, která vedla k nutnosti
středoušní rekonstrukce, typy a počty provedených operací
a re - operací a audiogram před a po rekonstrukční operaci.
VÝSLEDKY
Ve studii jsme nezjistili žádný významný rozdíl v počtu
provedených rekonstrukcí mezi chlapci a dívkami ani v rámci
stranového postižení. Nejčastějším typem středoušní
rekonstrukce bylo zavedení protézy typu TORP, a to ve 46
případech. Zavedení protézy typu PORP je až na posledním
místě u 8 pacientů. Tyto protézy byly téměř vždy zavedeny
až ve druhé době (operace typu second look). Jednalo se převážně
o pacienty po operaci cholesteatomu. V pořadí druhým
nejčastějším výkonem byla tympanoplastika II. a III. typu
dle Tose, tedy interpozice chrupavkou (ev. kostí) a vložení
kolumely z kovadlinky. Tento výkon jsme provedli u 25 pacientů.
Třetím nejpočetnějším typem operace bylo provedení
manubriostapedopexe, myringostapedopexe a neomyringostapedopexe,
a to u 10 pacientů. Na rozdíl od rekonstrukce
pomocí PORPu a TORPu se v případech použití autologních
materiálů jednalo vždy o rekonstrukci již v první době, tedy
na závěr sanační fáze středoušní operace (graf 1).
Nejčastější patologií, která vedla k nutnosti rekonstrukce
středoušních kůstek, byl v našem souboru jednoznačně výskyt
cholesteatomu ve středním uchu, a to až u 69 pacientů,
to představuje 78 % všech pacientů. Další příčinou byl výskyt
těžší formy adhezivní otitidy s fixací nebo destrukcí kůstek
Graf 2 Příčiny vedoucí ke středoušní rekonstrukci.
chole – cholesteatom, adhesiva – Otitis media chronica adhesiva,
VVV – vrozené vývojové vady
14 OTORINOLARYNGOLOGIE A FONIATRIE 2017, 66, č. 1
PŮVODNÍ PRÁCE
(13 %) a minoritní podíl na postižení středoušních kůstek
tvoří vývojové anomálie středního ucha (7 %) a úrazy (2 %).
Důležitým parametrem sledovaným ve studii bylo samozřejmě
hodnocení sluchového zisku. Graf 3 ukazuje průměrné
vzdušné vedení před rekonstrukční operací (červeně) a po
operaci (modře). Z audiogramu je patrné že z průměrného
sluchu na úrovni 52,5 dB před operací došlo ke zlepšení sluchu
na průměrnou hodnotu 31,6 db po operaci. Hodnoty sluchu
jsme získali jako průměr naměřených hodnot na daných
frekvencích získaných z audiogramů před rekonstrukční
operací a cca 1 rok po operaci. Věkové rozložení pacientů
ukazuje graf 4, nejmladší pacient byl ve věku 5 let.
Ve 24 případech byla nutná revizní operace. Indikací k revizní
operaci bylo ve všech případech zhoršení sluchu. Tabulka 1
ukazuje příčiny, které vedly ke zhoršení sluchu.
Graf 4 Věkové rozložení pacientů.
DISKUSE
Rekonstrukcí převodního systému středního ucha v dětském
věku můžeme úspěšně obnovit sluch narušený patologickým
procesem v této oblasti, a to dokonce s velmi dobrým
sluchovým ziskem. Dobrý sluch je v dětském věku nezbytný
pro správný vývoj řeči a učení jedince. Uzávěr perforace bubínku,
eradikace zánětu ve středouší a obnova převodního
aparátu s cílem zlepšení sluchu jsou u dětí s chronickým
zánětem středouší velmi významné, protože kvalita života
i mentální a jazykové dovednosti a vývoj dětí jsou operací
pozitivně ovlivněny (3). Různá pracoviště používají podobné
chirurgické přístupy, často se liší používané materiály a systémy
k rekonstrukci středouší, včetně vlastních navržených
protéz (4). Na našem pracovišti preferujeme u dětí retroaurikulární
přístup. Můžeme potvrdit, že se jedná o přístup,
který umožňuje dobrou orientaci a přehlednost operačního
pole, nízké riziko komplikací a dobré hojení. Ztotožňujeme
se s názorem jiných autorů, že retroaurikulární přístup
nepředstavuje v rukou zkušeného operatéra žádný trvalý
kosmetický defekt (7). Z hlediska použitých materiálů,
pokud to je možné, upřednostňujeme autologní materiál
(chrupavka, kovadlinka, kost). Jedná se o tympanoplastiku
I., II. a III. typu, dále o manubriostapedopexi, myringostapedopexi
a neomyringostapedopexi. Tyto výkony provádíme
vždy v první době, navazují tedy přímo na sanační fázi,
a to v případě, kdy jsou k tomu vhodné funkční podmínky
(drobný cholesteatom odstraněn in toto, vzdušné středouší
i systém mastoidních sklípků, ucho bez aktivního zánětu
a sekrece, nízké riziko recidivy cholesteatomu) i podmínky
anatomické (zachování třmínku, dobrá pohyblivost třmínku,
velikost bubínkové dutiny a podobně). Tyto faktory
potřebné k úspěšné rekonstrukci jsou obecně známé (6).
Z literatury popisovaných komplikací jako trvalá percepční
ztráta sluchu, tinitus a vertigo (13) jsme v našem souboru pacientů
pozorovali pouze 2x tinitus přechodného charakteru.
K častému použití autologních tkání nás vede přesvědčení,
že vlastní tkáně se lépe přihojí, lépe fungují, nepůsobí jako
cizí těleso, vydrží dlouhou dobu beze změny. Tyto názory
podporují i histopatologické studie, které ukazují, že autologní
materiály (kladívko, kovadlinka, chrupavka, kost) si
udržují po implantaci svůj tvar, velikost a funkci i víc než
30 let, na rozdíl od syntetických protéz z porézního plastu
nebo biokompatibilního skla, kde byla pozorována reakce
na cizí těleso i částečný rozpad materiálu po určité době
(2). Určitou nevýhodou autologních materiálů je možnost
vzniku fixace po různě dlouhé době. V případě dobrých anatomických
poměrů je možné provést manubriostapedopexi,
myringostapedopexi nebo neomyringostapedopexi. Jedná se
dnes o málo používané, ale efektivní postupy s velmi dobrým
průběhem hojení, výborným sluchovým ziskem a trvalým
úspěchem operace bez nutnosti re - operace (9). Při rozhodování,
zdali použít autologní nebo biokompatibilní materiál,
je nutno brát na vědomí fakt, že v mnoha případech, kdy si
můžeme vybrat jednu nebo druhou variantu jsou funkční
výsledky velmi podobné, ale operace s využitím vlastní
tkáně je mnohem levnější (5). Stavy, kdy nejsou vhodné
funkční a anatomické podmínky k rekonstrukci pomocí
autologního materiálu, řešíme náhradu řetězce kůstek biokompatibilními
protézami PORP a TORP. Tyto rekonstrukce
probíhají ve většině případů až ve druhé době, po dostatečné
Graf 3 Sluchový zisk.
Recidiva základního onemocnění (cholesteatom) 2x
Jiné (tympanoskleróza, změna polohy TORPu) 10x
Výměna kolumely za TORP (z důvodu fixace kolumely) 7x
Adheze a srůsty ve středouší 5x
Tab. 1 Příčiny, které vedly ke zhoršení sluchu.
152017, 66, č. 1 OTORINOLARYNGOLOGIE A FONIATRIE
PŮVODNÍ PRÁCE
sanaci zánětu. Nejčastějším typem rekonstrukce s využitím
biokompatibilního materiálu bylo zavedení protézy typu
TORP. Na našem pracovišti používáme výhradně titanové
TORPy, se kterými máme dlouhodobě dobré zkušenosti. I jiná
pracoviště považují titanové TORPy za vhodné k úspěšné
rekonstrukci převodního systému středního ucha u dětí,
s nízkou četností extruze a dlouhodobě dobrými sluchovými
zisky (13). Průměrný zisk sluchu po rekonstrukci dosáhl
hladiny 20,9 dB a kostně vzdušní rozdíl (ABG – air bone gap)
nepřesáhl 10 dB. Průměrná ztráta sluchu před operací činila
52,5 dB. Naše výsledky jsou na dobré úrovni i v porovnání
se zahraničními pracovišti (4, 6, 12, 13, 14). Někteří autoři
považují tympanoplastiku za operaci vhodnou až od určitého
věku dítěte (12). Věk dítěte nepovažujeme za zcela rozhodující
kritérium úspěšnosti operace. V našem souboru jsou
děti od 5 let věku.
ZÁVĚR
Středoušní rekonstrukce v dětském věku představují složitou
problematiku, kterou je potřeba řešit komplexně,
vždy s cílem dosáhnout co nejlepšího sluchového zisku
pro pacienta, pokud je to možné. Na naší klinice používáme
postupy a klasifikace operací dle profesora Tose.
V dětském věku doporučujeme retroaurikulární přístup
a použití autologních materiálů, pokud je to možné. Vždy
je nutné pečlivě zvážit indikaci k rekonstrukci vzhledem
k anatomickým a funkčním poměrům ve středním uchu
u daného pacienta. Výsledky studie ukazují, že i v dětském
věku je možné dosáhnout dobrých výsledků jak operačních
tak sluchových. Naše výsledky jsou srovnatelné s mnoha
zahraničními pracovišti.
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Samim, E.: Type 2 ossiculoplasty: prognostic determination of
hearing results by middle ear risk index. Am. J. Otolaryngol., 31,
2010, 5, s. 325-331.
7. Hong, P., Arseneault, T., Makki, F.: A long-term analysis of
auricular position in pediatric patients who underwent post-auricular
approaches. Int. J. Pediatr. Otorhinolaryngol., 78, 2013, 3, s.
471-473.
8. Chrobok, V., Pellant, A., Profant, M.: Cholesteatom spánkové
kosti. 1. vyd., Havlíčkův Brod, Česká republika, Tobiáš; 2008.
9. Janzen, V. D.: Malleus to stapes assembly. J. Otolaryngol., 14,
1985, 1, s. 57-58.
10. Jensen, R. G., Koch, A., Homøe, O.: The risk of hearing loss
in a population with a high prevalence of chronic suppurative otitis
media. International Journal of Pediatric Otorhinolaryngology
[online], 77, 2013, 9, s. 1530-1535.
11. Tos, M.: Manual of middle ear surgery: Approaches,
Myringoplasty, Ossiculoplasty, Tympanoplasty. Thieme, Stuttgart,
1993.
12. Uyar, Y., Keles, B., Koc, S.: Tympanoplasty in pediatric patients.
Int. J. Pediatr.
13. Wolter, N. E., Holler, T., Cushing, S. L., ChadhA, N. K.,
Gordon, K. A., James, A. L., Papsin, B. C.: Pediatric ossiculoplasty
with titanium total ossicular replacement prosthesis.
Laryngoscope, 2014, DOI: 10.1002/lary.24896.
14. Zheng, C., Guyot, J. P., Montandon, P.: Ossiculoplasty by
interposition of a minor columella between the tympanic membrane
and stapes head. Am. J. Otol., 17, 1996, 2, s. 200-202.
Adresa ke korespondenci:
MUDr. Milan Urík
Klinika dětské ORL LF MU a FN
Černopolní 9
613 00 Brno
e-mail: docttor.urik@gmail.com
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology
journal homepage: www.elsevier.com/locate/ijporl
First experiences with a new adhesive bone conduction hearing device in
children
Milan Uríka,∗
, Dagmar Hošnováa
, Ivo Šlapáka
, Jana Jančíkováa
, Jan Odstrčilíka
, Jiří Jarkovskýb
,
Wolf-Dieter Baumgartnerc
a
Department of Pediatric Otorhinolaryngology, University Hospital Brno and Faculty of Medicine, Masaryk University Brno, Brno, Czech Republic
b
Institute of Biostatistics and Analyses, Faculty of Medicine and the Faculty of Science, Masaryk University, Brno, Czech Republic
c
Department of Otorhinolaryngology, Medical University of Vienna, Vienna, Austria
A R T I C L E I N F O
Keywords:
Pressure-free
Bone conduction hearing aid
Children
A B S T R A C T
Objectives: To evaluate the hearing benefit, advantages, and disadvantages in a series of children using a new,
nonimplantable, pressure-free, adhesive bone conduction hearing aid.
Methods: Seventeen children were included in the study. 5 children suffered from bilateral conductive hearing
loss (CHL), 6 children with unilateral CHL and 6 children with unilateral sensorineural hearing loss. An
audiological tests were provide. Additionally, sound quality (SSQ10) and quality of life (AQoL-6D) were assessed
using questionnaires.
Results: The average value of speech audiometry with bubble noise in children with SNHL is 21.33 ( ± 5.72) dB
HL with the device and 27.67 ( ± 4.59) dB HL without the device, which is a statistically significant gain
(p = 0.027). The analysis showed the average value of hearing threshold in sound field in the group of children
with CHL supported 20.23 ( ± 16.84) dB HL and not supported 33.52 ( ± 27.27) by the hearing aid for bone
conduction, which i a statistically significant gain (p = 0.008). The average value of speech audiometry is 23.45
( ± 14.45) dB HL with the device and 37.27 ( ± 26.65) dB HL without the device, which is a statistically significant
gain (p = 0.012). The average value of speech audiometry with bubble noise is 30.55 ( ± 10.03) dB HL
with the device and 45.45 ( ± 18.41) dB HL without the device, which is a statistically significant gain
(p = 0.008). No patient referred pain or irritation.
Conclusion: This new device for bone conduction show a hearing benefit for a paediatric patient, without any
concomitant aesthetic and other complications.
1. Introduction
Children with conductive hearing loss or unilateral sensorineural
hearing loss can be rehabilitated using bone conduction hearing devices
(BCCHD) which are located outside as a softband or are surgically
implanted into temporal bone. Dumping effect of skin is overcome by
implantation of the device into skin or by increased pressure on skin
[1,2]. Implantable devices are divided into passive and active. Passive
systems include bone implants (BAHI), for example Cochlear BAHA
Connect and Attract, series Oticon Ponto and Sofono, Soundbite (teeth),
Spectacle (glasses) or other adhesive retained bone conduction devices
that are available for use in children (conventional bone conduction
aids (with metal headband) that have been modified to be adhesive
retained (Carroll-Ann Trotman and all.). Active devices for children
include Vibrant Soundbridge (VSB, Med-El, Innsbruck, Austria) and
Bonebridge (BB, Med-El, Innsbruck, Austria). Numerous studies proved
the benefit of BAHI for hearing development, however, on the other
hand, frequent infections near the device required seeing the doctor
repeatedly and despite the conservative treatment surgical revisions
were often necessary [3]. When increased pressure on skin is used to
improve the effect, the skin may be irritated, red and itchy, leading to
significant discomfort. Skin necrosis was also reported [4]. Of course a
lot of children don't wear them because of pressure on the head,
sweating, or cosmetic stigma.
In order to overcome complications related to pressure on skin and
implantations, Med-El company developed a new device for bone
conduction which minimises pressure on skin and may not be implanted.
It is ADHEAR. The device consists of two parts, an adapter and
https://doi.org/10.1016/j.ijporl.2019.109614
Received 1 May 2019; Received in revised form 24 June 2019; Accepted 27 July 2019
*
Corresponding author. Head of Department of Pediatric Otorinolaryngology, University Hospital Brno and Faculty of Medicine, Masaryk University Brno
Černopolní 9, 61300, Brno, Czech Republic.
E-mail address: urik.milan@fnbrno.cz (M. Urík,>.
Příloha 10: Urík M, Hošnová D, Šlapák I, Jančíková J, Odstrčilík J, Jarkovský J, Baumgartner WD. First
experiences with a new adhesive bone conduction hearing device in children. Int J Pediatr Otorhinolaryngol.
2019;126:109614. doi:10.1016/j.ijporl.2019.109614
a vibrator. Audio processor contains a battery, microphone, amplifier
and vibrator. The processor transforms sound waves into vibrations.
Vibrations are then transferred from the processor to adapter, to the
skin and through skull bone into cochlea. There is no constant pressure
on the skull, the contact is maintained only via the adhesive glue of the
adapter. The device is depicted in Fig. 1. The patent details are available
[5]. The aim of the study is to present first experience with the new
device in 11 children with conductive hearing loss (CHL) and 6 children
with unilateral sensorineural hearing loss (SNHL).
2. Methods
11 paediatric patients with CHL and 6 with SNHL were included in
this study. Demographic characteristics of the patients are summarised
in Table 1 and Table 2. An audiologist checked the device before its use.
For each patient an ideal site behind the auricle in the area near planum
mastoideum was identified and cleaned with alcohol before the adapter
application. Patients were instructed on how to put on the device, how
to set volume and manipulate with the device. Hearing was examined
right after the device was set, then after 2 weeks, and also without the
device. Based on the manufacturer recommendations and a previous
study, 14 days interval seems to be adequate to assess the device effect.
The study was performed with the approval of ethics committee of
University Hospital Brno. The pressure free adhesive bone conduction
device is called ADHEAR and distributed by Med-El.
Fig. 1. A, The adhesive adapter attached behind the ear. B, The audio processor coupled to the adhesive adapter.
Table 1
Patients details and Pure tone average treshold for 0.5, 1, 2 and 4 kHz in dB HL. F – Female, M − Male, R – right, L – Left, B – Billateral, Ac – Air Conduction, BC –
Bone Conduction, SNHL – Sensorineural Hearing Loss, Atresia – congenital atresia of external ear canal, Stenosis – congenital stenosis of external ear canal, Cholest –
Cholesteatoma (after canal wall down), AUDIO before – warble in the sound field with the previously device (mean in db for 0,5,1,2, and 4 kHz).
ID AGE (Y) SEX SIDE AC right BC right AC left BC left DIAGNOSIS Previously aided Further treatment AUDIO before
P1 11 F R 111 0 10 5 SNHL 0 0 P2
16 M R 92 0 7,5 5 SNHL 0 Cross P3
9 M R 107 0 8,75 5 SNHL 0 0 P4
11 F L 3,7 2,5 92,5 0 SNHL 0 0 P5
14 F R 102 0 2,5 2,5 SNHL 0 Cross P6
5 M R 112 0 7,5 5 SNHL 0 0 P7
10 F R 45 11 2,5 0 Atresia 0 0 –
P8 10 M B 60 8,75 55 10 Atresia BAHA 5 BAHA Attract 25
P9 11 M B 58 10 60 15 Atresia BAHA Divino Bonegridge 31.25
P10 13 M B 57 5 60 5 Atresia Bone hearing aid Bonebridge 26.25
P11 13 F L 19 15 57,5 15 Atresia 0 Bonebridge –
P12 5 M R 55 0 10 5 Atresia 0 0 –
P13 10 M L 20 15 61,25 15 Atresia 0 0 –
P14 14 M B 59 20 57,5 15 Atresia 0 0 –
P15 18 M L 20 15 62,5 15 Stenosis 0 0 –
P16 10 F B 58 25 67,5 23,7 Cholest Hearign aid Hearing aid 45
P17 19 M R 92 24 12,5 15 Cholest 0 Cross –
Table 2
Summary of demographic parameters of patients.
Characteristicsa
Patients with
SNHL (N = 6)
Patients with
CHL (N = 11)
Total (N = 17)
Age (years) 11.00 (5.00;
16.00)/11.00
( ± 3.85)
11.00 (5.00;
19.00)/11.71
( ± 3.84)
11.00 (5.00;
19.00)/11.71
( ± 3.84)
Sex Male 3 (50.0%) 8 (72.7%) 11 (64.7%)
Female 3 (50.0%) 3 (27.3%) 6 (35.3%)
Side L 2 (33.3%) 3 (27.3%) 5 (29.4%)
R 4 (66.7%) 3 (27.3%) 7 (41.2%)
R + L 0 (0.0%) 5 (45.4%) 5 (29.4%)
a
Absolute and relative frequencies for categorical variables; median supplemented
with minimum and maximum, mean supplemented with standard
deviation for scale variable.
M. Urík,><, et al.
2.1. Audiological evaluations
All audiological examinations were performed with and without the
device. In order to minimise the hearing input from the contralateral
ear, a standard masking of the nontest ear based on the level of contralateral
hearing loss was performed. Sound-field audiometry was
performed using a narrow band masking noise. All examinations were
performed using Interacoustic AC 40 audiometer.
The threshold values for the hearing field were measured on the
ipsilateral ear at frequencies 500, 1000, 2000 and 4000 Hz. Frequencymodulated
tones were presented from the speaker from the distance of
3 m. Sound-field audiometry (SF), speech audiometry (SPEECH) and
speech audiometry with bubble noise (SPEECH + W) were performed.
The results are presented as average values of hearing threshold for
individual frequencies with and without the device.
All patients have a standard pure tone audiometry at the start of the
study.
2.2. Hearing-related questionnaire
The Speech, Spatial, and Qualities of Hearing Scale (SSQ) [6] is
designed to measure auditory disability across a wide variety of domains,
reflecting the reality of hearing in everyday life. A short form of
the questionnaire (SSQ 10) comprises 10 instead of 49 items and is
therefore less time consuming, while providing similar outcomes [10].
Items are scored with ratings of 1–10 on a visual analogue scale (Likert
scale), with higher numbers representing greater satisfaction. Apart
from a total score, SSQ10 provides three subscores for speech understanding,
spatial hearing and other qualities of hearing. The SSQ10 was
completed by the subjects at the beginning to assess their hearing
ability before the study and after wearing the device for 2 weeks.
2.3. Quality of life assessment
The AQoL-6D (Assessment of Quality of Life-6 Dimensions) questionnaire
is a multiattribute utility measure for health-related quality of
life evaluation [7]. It comprises 20 questions covering a multitude of
quality of life aspects. Weighted utilities were generated using the algorithms
provided on the AQoL website [8] and used for statistical
analysis.
2.4. Device-specific questionnaire
Subjects completed a device-specific questionnaire to report any
encountered adverse events during the testing period, battery-life, and
the durability (continuous wearing time) of the adhesive adaptor.
All questionnaires were completed by children or both children with
parents depending on age.
2.5. Statistical analysis
Standard descriptive statistics was applied in the analysis; absolute
and relative frequencies for categorical variables, median supplemented
with minimum and maximum and mean supplemented with standard
deviation for continuous variables. Statistical significance of differences
in paired data was tested using Wilcoxon paired test. Analysis was
computed using SPSS 25.0.0.1 (IBM Corporation, 2018) and R version
3.5.1 with ggplot2 package.
3. Results
3.1. Audiology results – patients with SNHL
The analysis showed the average value of hearing threshold in
sound field in the group of children with SNHL supported 9.00
( ± 5.16) dB HL and not supported 13.54 ( ± 5.50) by the hearing aid
for bone conduction with an average functional gain of 4.54 ( ± 3.81)
dB HL, which is not a statistically significant gain (p = 0.058). The
average value of speech audiometry is 10.17 ( ± 6.31) dB HL with the
device and 12.33 ( ± 6.35) dB HL without the device, with an average
functional gain of 2.17 ( ± 10.23) dB HL, which is not a statistically
significant gain (p = 0.916). The average value of speech audiometry
with bubble noise is 21.33 ( ± 5.72) dB HL with the device and 27.67
( ± 4.59) dB HL without the device, with the average gain of 6.33
( ± 5.28) dB HL, which is a statistically significant gain (p = 0.027).
Any patients had never used a hearing aid before.
3.2. Audiology results – patients with CHL
The analysis showed the average value of hearing threshold in
sound field in the group of children with CHL supported 20.23
( ± 16.84) dB HL and not supported 33.52 ( ± 27.27) by the hearing
aid for bone conduction with an average functional gain of 13.30
( ± 13.88) dB HL, which i a statistically significant gain (p = 0.008).
The average value of speech audiometry is 23.45 ( ± 14.45) dB HL with
the device and 37.27 ( ± 26.65) dB HL without the device, with an
average functional gain of 13.82 ( ± 14.11) dB HL, which is a statistically
significant gain (p = 0.012). The average value of speech audiometry
with bubble noise is 30.55 ( ± 10.03) dB HL with the device and
45.45 ( ± 18.41) dB HL without the device, with the average gain of
14.91 ( ± 14.10) dB HL, which is a statistically significant gain
(p = 0.008). 4 patients had used a hearing aid before.
The complete results for the audiological outcomes between children
with SNHL and CHL are illustrated in Figs. 2–4.
3.3. Questionnaire results – patients with SNHL
The SSQ6 questionnaire revealed a mean total score of 199.88
( ± 85.68) at the first visit and improved to 267.99 ( ± 50.39) after 2
weeks of device usage. There was a statistically significant difference
between the two test intervals in terms of total score (p = 0.028).
The mean utility of the AQoL-6D questionnaire increased from 0.80
( ± 0.24) out of 1 at the first visit to 0.90 ( ± 0.11) at the 2 - week visit.
Statistical analysis not revealed a statistically significant difference
(p = 0.144) between the two test intervals. In the subcategories were
any statistically significant differences between the two test intervals.
The complete results for the questionnaire outcomes in children with
SNHL are illustrated in Table 3a.
3.4. Questionnaire results – patients with CHL
The SSQ6 questionnaire revealed a mean total score of 215.67
( ± 54.45) at the first visit and improved to 261.39 ( ± 73.02) after 2
weeks of device usage. There was not a statistically significant difference
between the two test intervals in terms of total score (p = 0.050).
The mean utility of the AQoL-6D questionnaire increased from 0.78
( ± 0.25)) out of 1 at the first visit to 0.87 ( ± 0.24) at the 2 - week
visit. Statistical analysis not revealed a statistically significant difference
(p = 0.059) between the two test intervals. In the subcategories
were statistically significant differences between the two test intervals
in independent living (0.012). The complete results for the questionnaire
outcomes in children with CHL are illustrated in Table 3b.
The complete results for the questionnaires between children with
SNHL and CHL are illustrated in Figs. 5 and 6.
3.5. Clinical experience
Based on this study, 2 patients with SNHL decided for Cross hearing
aid (at their own cost, because in Czech Republic is any financial support
from insurance company for hearing aid in the case of unilateral
SNHL). In the group of patients with CHL 1 patient decided for Cross
hearing aid, 1 for classic hearing aid, 1 for BAHA Attract and 3 for
M. Urík,><, et al.
Bonebridge. In all patients who chose not to continue with ADHEAR,
the resason was that this device is not funded in our country now.
The patient details about previously hearing aid and further treatment
are in Table 1. A battery change was necessary 1–2 times in 14
days. No patient referred pain or irritation at the site of ADHEAR application
and no patient considered the device a cosmetic problem. The
doctor asked each patient/parents at the end of the study “Is the ADHEAR
cosmetically acceptable for you?” If NO, why?”
4. Discussion
Our study showed that children with CHL or SNHL may have a good
benefit in hearing as well as in speech comprehension when using the
novel hearing aid ADHEAR. 15 patients were fully satisfied and wanted
to continue using the device after the study finished (if it will be
possible). The success in using the hearing aid depends on the ratio
between the severity of the hearing loss and the level of improvement
which can be brought by the device to a compliant patient. Various
studies state that a reasonable gain when using the hearing devices is
hearing threshold is 30–35 dB HL [9]. Our paediatric patients gained
the average hearing threshold in sound field of 9.00 ( ± 5.16) dB HL in
SNHL group and 20.23 ( ± 16.84) dB HL in CHL group. Conventional
hearing aids for bone conduction are limited mainly by a dumping effect
of skin, mainly at frequency of 1 kHz. We did not examine the
thickness of skin and subcutaneous tissue volume as previous studies
proved no significant difference in hearing gain depending on skin and
subcutaneous tissue thickness [10]. Common hearing aids for bone
conduction are placed by a principle of certain pressure affecting the
skin. The device used in our study is attached to skin using glue and
adapter which might give an impression of insufficient effectiveness.
Fig. 2. Soundfield tresholds with and without Adhear (AD) in group with SNHL and CHL. Hearing in dB HL.
Fig. 3. SPEECH audiometry with and without ADHEAR in group of patients with SNHL and CHL. Hearing level in dB HL.
M. Urík,><, et al.
For common hearing aids for bone conduction the effect of pressure on
skin cannot be omitted. Even studies including children using softband
proved irritation and pain at the site of pressure [11]. In our study, no
patient had any irritation, pain or local complications. This novel
hearing aid overcomes the problem with pressure on skin using an
adhesive adapter that sticks to the skin. And there is the positive cosmetic
implication of the ADHEAR, which is an important benefit of this
technology. BAHA Softband is the most commonly used hearing aids in
Fig. 4. SPEECH audiometry in noise with and without ADHEAR in group of patients with SNHL and CHL. Hearing in db HL.
Table 3a
Summary of clinical parameters of patients with SNHL before usage of AD and after with usage of AD.
N = 6 Patients without AD1
: Patients with AD1
: Difference1
: p-value2
SF 15.00 (3.75; 18.75)/13.54 ( ± 5.50) 9.38 (2.50; 17.50)/9.00 ( ± 5.16) −5.63 (−8.75; 1.25)/−4.54 ( ± 3.81) 0.058
SPEECH 11.00 (4.00; 23.00)/12.33 ( ± 6.35) 12.50 (0.00; 16.00)/10.17 ( ± 6.31) 1.00 (−18; 11)/−2.17 ( ± 10.23) 0.916
SPEECH + w 28.50 (22.00; 34.00)/27.67 ( ± 4.59) 21.00 (12.00; 29.00)/21.33 ( ± 5.72) −5.00 (−15.00; −1.00)/−6.33 ( ± 5.28) 0.027
SSQ 209.00 (102.50; 298.50)/199.88 ( ± 85.68) 279.67 (174.00; 310.00)/267.99 ( ± 50.39) 37.15 (1.00; 181.00)/68.12 ( ± 78.47) 0.028
Independent Living 0.95 (0.81; 1.00)/0.94 ( ± 0.07) 0.95 (0.81; 1.00)/0.94 ( ± 0.07) 0.00 (0.00; 0.02)/0.00 ( ± 0.01) 0.317
Relationships 1.00 (0.47; 1.00)/0.89 ( ± 0.21) 1.00 (0.49; 1.00)/0.90 ( ± 0.20) 0.00 (0.00; 0.03)/0.00 ( ± 0.01) 0.317
Mental Health 0.68 (0.08; 0.95)/0.60 ( ± 0.30) 0.90 (0.65; 0.98)/0.84 ( ± 0.13) 0.00 (0.00; 0.25)/0.04 ( ± 0.10) 0.317
Coping 0.68 (0.35; 0.98)/0.68 ( ± 0.24) 0.90 (0.41; 0.98)/0.85 ( ± 0.15) 0.00 (0.00; 0.58)/0.16 ( ± 0.26) 0.180
Pain 1.00 (0.66; 1.00)/0.91 ( ± 0.15) 1.00 (0.77; 1.00)/0.94 ( ± 0.10) 0.00 (0.00; 0.19)/0.03 ( ± 0.08) 0.317
Senses 0.82 (0.41; 0.99)/0.79 ( ± 0.22) 0.95 (0.88; 0.99)/0.94 ( ± 0.05) 0.13 (−0.08; 0.47)/0.15 ( ± 0.21) 0.141
Utility Score 0.87 (0.35; 0.99)/0.80 ( ± 0.24) 0.93 (0.72; 0.99)/0.90 ( ± 0.11) 0.06 (−0.02; 0.37)/0.10 ( ± 0.15) 0.144
2
p-values are computed using Wilcoxon test.
1
Absolute and relative frequencies for categorical variables; median supplemented with minimum and maximum, mean supplemented with standard deviation for
scale variable.
Table 3b
Summary of clinical parameters of patients with CHL before usage of AD and after with usage of AD.
N = 11 Patients without AD1
: Patients with AD1
: Difference1
: p-value2
SF 20.00 (0; 68.75)/33.52 ( ± 27.27) 17.50 (0.00; 48.75)/20.23 ( ± 16.84) −8.75 (−35.00; 0.00)/−13.30 ( ± 13.88) 0.008
SPEECH 20.00 (8.00; 80.00)/37.27 ( ± 26.65) 22.00 (6.00; 46.00)/23.45 ( ± 14.45) −8.00 (−34.00; 2.00)/−13.82 ( ± 14.11) 0.012
SPEECH + w 40.00 (23.00; 76.00)/45.45 ( ± 18.41) 30.00 (14.00; 54.00)/30.55 ( ± 10.03) −10.00 (−38.00; 0.00)/−14.91 ( ± 14.10) 0.008
SSQ 219.67 (115.00; 295.00)/215.67 ( ± 54.45) 287.36 (82.00; 329.50)/261.39 ( ± 73.02) 36.94 (−117.56; 114.34)/45.72 ( ± 65.50) 0.050
Independent Living 0.95 (0.21; 1.00)/0.82 ( ± 0.24) 0.97 (0.37; 1.00)/0.89 ( ± 0.19) 0.02 (0.00; 0.31)/0.07 ( ± 0.10) 0.012
Relationships 0.90 (0.42; 1.00)/0.88 ( ± 0.17) 1.00 (0.42; 1.00)/0.88 ( ± 0.19) 0.00 (−0.23; 0.13)/0.00 ( ± 0.08) 1.000
Mental Health 0.63 (0.11; 0.95)/0.65 ( ± 0.24) 0.82 (0.09; 0.95)/0.74 ( ± 0.25) 0.08 (−0.11; 0.39)/0.09 ( ± 0.15) 0.066
Coping 0.90 (0.34; 0.96)/0.79 ( ± 0.20) 0.90 (0.41; 0.96)/0.85 ( ± 0.16) 0.00 (−0.02; 0.42)/0.06 ( ± 0.13) 0.080
Pain 0.85 (0.10; 1.00)/0.77 ( ± 0.31) 1.00 (0.06; 1.00)/0.85 ( ± 0.28) 0.00 (−0.23; 0.74)/0.08 ( ± 0.26) 0.345
Senses 0.89 (0.41; 0.99)/0.84 ( ± 0.17) 0.97 (0.63; 1.00)/0.90 ( ± 0.12) 0.02 (−0.34; 0.58)/0.06 ( ± 0.22) 0.314
Utility Score 0.88 (0.26; 0.99)/0.78 ( ± 0.25) 0.95 (0.15; 1.00)/0.87 ( ± 0.24) 0.04 (−0.11; 0.56)/0.09 ( ± 0.18) 0.059
2
p-values are computed using Wilcoxon test.
1
Absolute and relative frequencies for categorical variables; median supplemented with minimum and maximum, mean supplemented with standard deviation for
scale variable.
M. Urík,><, et al.
paediatric patients. The average hearing gain in children with ear canal
atresia, using BAHA Softband, is 27 dB HL [13]. In our study, the
average hearing gain in patients with ear canal atresia was 19.5 dB HL.
Active or passive transcutaneous or percutaneous hearing aids for bone
conduction are other possibilities than the ones mentioned above.
These, however, do not account for better hearing rehabilitation as they
do not achieve sufficient effect at higher frequencies. The best values of
hearing threshold of normal tone or speech audiometry were found in
patients with BAHA anchored on abutment compared to patients using
BAHA Softband. Hearing threshold improved by 5–20 dB HL in sound
field and by 4–7 dB HL in speech audiometry at frequencies 1–4 kHz. No
differences were observed at frequencies 125–500 Hz [12]. This new
bone conduction device, attached using an adhesive adapter, represents
a good option for children with CHL or unilateral SNHL. There is no risk
of surgery and general anaesthesia. It is a valuable alternative for
children who only need a bone conduction hearing aid for a limited
period of time. This adhesive BCD can also be used in the same way as
the Baha Softband: as a temporary solution for small children who do
not have sufficient thickness of the skull bones to allow implantation
[12]. The ADHEAR show a potential additional benefit for kids that are
too young for implantable bone conduction hearing devices. These first
results of 17 paediatric patients using a new pressure-free, adhesive
bone conduction hearing aid show promising results. Our observation is
in agreement with other authors and prove the fact that ADHEAR represents
a good options for bone conduction [14,15]. We are aware of
the limitations of this study because we did not compare the ADHEAR
with the already existing aids for bone conduction and we did not use it
in small children under 5 years. The other problem can be the limitations
of this device in young babies which is particularly relevant in the
setting of bilateral atresia where a device is required in infancy.
Fig. 5. SSQ6 score with and without ADHEAR in group of patients with SNHL and CHL. Range = 0–300.
Fig. 6. AQoL-6D score with and without ADHEAR in group of patients with SNHL and CHL. Range = 0–1.
M. Urík,><, et al.
Likewise there may be challenges in having enough surface area for
application in young children with atresia particularly those with
hemifacial microsomia.
5. Conclusion
In our pilot study we present our first experiences with a new nonadhesive
hearing aid for bone conduction in children which is very well
tolerated and brings a good benefit for paediatric patients. An improvement
was proved using questionnaires which were filled in at the
beginning and at the end of the study. Sound-field audiometry, speech
audiometry and speech audiometry with buble noise proved a hearing
benefit with the device. No patient suffered from skin irritation or pain
at the site of adhesion. This new device for bone conduction show a
hearing benefit for a paediatric patient, without any concomitant aesthetic
and other complications.
Funding
Supported by project of Masaryk University, Brno, Czech Republic
MUNI/A/1002/2018.
Conflicts of interest
The ADHEAR study devices were provided by the manufacturer
(MED-EL, Innsbruck, Austria). All authors received travel and congress
support by MED-EL for workshop participation and congress presenta-
tions.
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