Bilateral oropharyngeal hairy polyps: A rare cause of dyspnea in newborns

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

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1808-8694/©2015Associac¸˜aoBrasileiradeOtorrinolaringologiaeCirurgiaC´ervico-Facial.PublishedbyElsevierEditoraLtda.Thisisanopen accessarticleundertheCCBYlicense(http://creativecommons.org/licenses/by/4.0/).

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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.2_Morgan4_{reportedamale}

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.

6.VarshneyR,PitaroJ,AlghonaimY,LacroixY.Hemorrhagichairy polypcausing velopharyngealdysfunction ina newborn.Cleft PalateCraniofacJ.2014[Epubaheadofprint].

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