BrazJOtorhinolaryngol.2017;83(1):117---118
www.bjorl.org
Brazilian
Journal
of
OTORHINOLARYNGOLOGY
CASE
REPORT
Bilateral
oropharyngeal
hairy
polyps:
a
rare
cause
of
dyspnea
in
newborns
夽
Pólipos
pilosos
bilaterais
de
orofaringe:
uma
causa
rara
de
dispneia
em
neonatos
Rasim
Yilmazer
∗,
Burak
Kersin,
Erkan
Soylu,
Gokhan
Altin,
Asli
Cakir,
Fahrettin
Yilmaz
IstanbulMedipolUniversity,DepartmentofOtorhinolaryngology,Istanbul,Turkey
Received16April2015;accepted4June2015 Availableonline16October2015
Introduction
Hairypolyp(HP)isoneofthecausesofcongenitaldyspnea, araredevelopmentalmalformationofbigerminaloriginthat comprisesbothectodermalandmesodermalelements for-eign tothe site in which itis found. Ittypically presents asapedunculatedmassintheoropharynxandnasopharynx. Majorsymptomsatpresentationarerelatedwithrespiratory obstructionandfeedingproblems.Intheliterature, congen-italpharyngealhairypolypsaregenerallyunilateral.1,2
Case
report
A 3440g white femalenewborn was born by spontaneous vaginal delivery at 39 weeks gestation of a 20 year-old G2P1A1female,followinganormalpregnancy.Immediately atbirth,shehadrespiratorydistressandcyanosis,andthen requiredoropharyngealintubationandmechanical ventila-tion. On oropharyngeal examination, there was only one
夽
Pleasecitethisarticleas:YilmazerR,KersinB,SoyluE,AltinG, CakirA,YilmazF.Bilateraloropharyngealhairypolyps:ararecause ofdyspneainnewborns.BrazJOtorhinolaryngol.2017;83:117---8.
∗Correspondingauthor.
E-mail:ryno78@gmail.com(R.Yilmazer).
PeerReviewundertheresponsibilityofAssociac¸ãoBrasileirade OtorrinolaringologiaeCirurgiaCérvico-Facial.
soft,skin-coveredmassoriginatingfromtheposteriorpillar oftherighttonsilandelongatingtothelevelofthe orophar-ynx;however,onnasopharyngoscopy,anothersimilarmass originatingfromtheposterior pillarof the lefttonsil was visualized,elongatingthroughthenasopharynx(Fig.1Aand B).Additionally, bilateral low-set earanomaly wasfound. Magneticresonanceimaging(MRI)oftheneckdemonstrated awell-defined,hyperintensepolypoid massesattachingto posteriorside ofthe tonsils(Fig.1C). MRIfeatures ofthe masslesions were definedas heterogeneoushyperintense appearanceonT1andT2-weightedseries.Infat-suppressed sequences, it was suppressed, and in the post-contrast series,therewasnosignificantcontrastenhancement.
Under general anesthesia, HPs were completely removed through transoral route under endoscopic visu-alization by cold dissection and bipolar electrocautery (Fig. 1D). Pathological examination showed two HPs, 27mm×18mm×14mm and 23mm×13mm×13mm in
size(Fig. 1E). She wasdischarged after twodays. Unfor-tunately,afterthefirstfollow-up, thepatientwaslost to follow-upandthusitwasnotpossibletodeterminewhether shehadasyndrome.
Discussion
Themostcommonlocalizationof HPisfromthe nasopha-rynx;arisingfromthesuperioraspectofthesoftpalateor
http://dx.doi.org/10.1016/j.bjorl.2015.06.001
118 YilmazerRetal.
Figure1 (A)Hairypolyporiginatingfromtheposteriorpillaroftherighttonsil(M,mass;U,uvula;RT,righttonsil).(B)Hairy polyporiginatingfromtheposteriorpillarofthelefttonsilelongatingthroughthenasopharynx(M,mass;u,Uvula).(C)Magnetic resonanceimageofthehairypolyporiginatingfromtheposteriorpillaroftherighttonsil.(D)Theexcisedspecimens(RS,right specimen;LS,leftspecimen).(E)Keratinizedsquamousepitheliumonthesurfaceandmesenchymaltissuecomponents,suchas hairfolliclesliningthefibroadiposecore(HE40×).
lateralpharyngealwall.Femaleinfantsaresixtimesmore likelytobeaffectedthanmales.3
ThemostcommonsymptomoftheHPisrespiratory dis-tress.Airwayobstruction mayoccur ifthepolyp becomes impacted in the larynx,and deaths have been reported.1
Differential diagnosis of the pharyngeal obstruction in a newborn includes choanal atresia, intranasal glioma, and encephalocele.3 Since HP is rare and may present with
respiratorydistressinnewborns,itshouldbeconsideredin differentialdiagnosis,andacarefulendoscopicevaluation byotolaryngologistsmustbeperformed.
There are few multiple bigerminal (HPs) and trigermi-nal teratomas that have been reported in the head and neck,andwithintheairway.2Morgan4reportedamale
new-born with two HPs that originated from the left side of the oropharynx and nasopharynx. Since the laterality of thenasopharyngeal masswas not noted,it wasnot clear whether they were bilateral. Franco et al.5 reported a
58 year-oldfemale with bilateral oropharyngeal HPs.She wasreferred to them because of feeding difficulties and milddyspnea,butshehadhadnorelevantmedicalhistory. Hence, they are probably not congenital. To the best of the authors’ knowledge, this is the first case of bilateral pharyngeal HPs in a newborn in the literature. A thor-ough examination of the oronasopharynx is mandatory in newbornswithanoropharyngealmassinordertoavoid over-lookingasecondmass.
Theendoscopicapproachaidsavoidanceofinjurytothe eustachiantubeorifice, aswellasexcisingthepathologic lesionsmorecompletely.However,velopharyngeal dysfunc-tionisobservedasacomplicationofsurgicalapproach.6
Final
remarks
Since HPscan bebilateral, diagnosis requires acomplete otorhinolaryngological examination. Transoral endoscopic resection should be performed to avoid undesirable complications.
Conflicts
of
interest
Theauthorsdeclarenoconflictsofinterest.
References
1.Agrawal N, Kanabar D, Morrison GA. Combined transoral and nasendoscopic resection of an eustachian tube hairy polypcausing neonatalrespiratorydistress.Am JOtolaryngol. 2009;30:343---6.
2.YilmazM,IbrahimovM,OzturkO,KaramanE,AslanM.Congenital hairypolypofthe softpalate. IntJPediatr Otorhinolaryngol. 2012;76:5---8.
3.KellyA,BoughIDJr,LuftJD,ConardK,ReillyJS,TuttleD.Hairy polypof theoropharynx: casereportand literaturereview.J PediatrSurg.1996;31:704---6.
4.Morgan.Acaseofdermoidpolypofpharynxandnasopharynx.J LaryngolOtol.1964;78:965---8.
5.FrancoV,FlorenaAM,LombardoF,RestivoS.Bilateralhairypolyp oftheoropharynx.JLaryngolOtol.1996;110:288---90.
