Braz. j. . vol.81 número4

BrazJOtorhinolaryngol.2015;81(4):451---453

www.bjorl.org

Brazilian

Journal

of

OTORHINOLARYNGOLOGY

CASE

REPORT

Visual

loss:

a

rare

complication

of

maxillary

sinus

mucocele

夽

,

夽夽

Perda

visual:

complicac

¸ão

rara

de

mucocele

de

seio

maxilar

Juliana

Caminha

Simões,

Francisco

Bazílio

Nogueira-Neto,

Luciano

Lobato

Gregório,

Fábio

de

Azevedo

Caparroz,

Eduardo

Macoto

Kosugi

RhinologyDivision,EscolaPaulistadeMedicina,UniversidadeFederaldeSãoPaulo(EPM/UNIFESP),SãoPaulo,SP,Brazil

Received26January2015;accepted16March2015 Availableonline6June2015

Introduction

Mucoceles are cystic formations lined with respiratory epithelium that contain a mucoid or mucopurulent fluid. Although benign,mucoceles arelocally expansive,due to continuousproductionandaccumulationofmucus,resulting inerosionandunderlyingboneremodeling.Thismayaffect theorbit,skullbase,orsofttissueoftheface.Frontal, eth-moidal,andfronto-ethmoidalsinusesarethemostcommon locations,while the, maxillaryandsphenoidalsinuses are lessfrequentlyaffected.Maxillarysinusmucocelesarerare, accountingforlessthan10%ofallmucocelesintheUSAand Europe.1

Orbitalsymptomsarerelativelycommoninpatientswith mucocele,duetotheexpansionofthelesionintotheorbit.1 These symptoms are usually caused by ethmoidal, sphe-noidal, and frontal mucoceles. Ophthalmologic impact is unusualfrommaxillarymucoceles.Astoorbitalsymptoms,

夽 _{Pleasecitethisarticleas:SimõesJC,Nogueira-NetoFB,Gregório}

LL,CaparrozFA,KosugiEM.Visualloss:ararecomplicationof max-illarysinusmucocele.BrazJOtorhinolaryngol.2015;81:451---3.

夽夽_{Institution: Rhinology Sector, Department of}

Otorhinolaryn-gology and Headand Neck Surgery,Escola Paulistade Medicina, UniversidadeFederaldeSãoPaulo(EPM/UNIFESP),SãoPaulo,SP, Brazil.

∗_{Correspondingauthor.}

E-mail:[email protected](E.M.Kosugi).

edemaandperiorbitalpainarethemostcommonfindings. Amaurosisisthemostfearedcomplication,butfortunately itoccurswithlowfrequency,2_{anditsoccurrenceinmaxillary} mucocelesisextremelyrare.1---5

The objective of this study is toreport a rare case of maxillarymucoceleleadingtoamaurosis.

Case

presentation

Thisreportconcernsamalepatient,67yearsofage,with proptosis and a progressive bulging in left malar region, in association with a lasting continuous ipsilateral nasal obstruction/clear rhinorrhea. In about five months, his visualacuitybegantodecrease,accompanied by progres-sive eye pain on the left side. Clinical history revealed facialtrauma 18 yearsin the past, hypertensionand dia-betes mellitus. Physical examination revealed left malar areadeformity,withpainonpalpationinassociation with ipsilateralproptosis.Nasalendoscopyshowedmedialization oftheleftlateralnasalwall,causingacompleteocclusion ofthe nasal cavity. An ophthalmologicevaluation showed decreasedleftpupillaryreflexes,visualacuity,and extraoc-ularmotility.Computedtomographyoftheparanasalsinuses showedan expansible lesioninto theleft maxillarysinus, withdensity compatiblewithsofttissue,withnocontrast enhancement,andremodelingoftheadjacentbone struc-ture,suggestiveofmaxillarymucocele(Fig.1).

Thepatientunderwentmaxillarymucocele marsupializa-tionthrough nasal endoscopyby a widemaxillary middle

http://dx.doi.org/10.1016/j.bjorl.2015.03.008

452 SimõesJCetal.

Figure1 (A)Computedtomography (CT)(axialsection),showinganexpansivelesionoccupyingtheentireleftmaxillarysinus andpromoting remodelingofbonelimits. (B)CT(coronalsection),showinganextensionofthelesiontotheorbitalcone.(C) Preoperativepictureshowingbulgingofthemalarareaandproptosis(leftside).(D)Postoperativephotographyshowingaclear improvementofbulgingandproptosis.

meatal antrostomy, resulting in a profuse draining of a yellow-citrinefluid.Intheimmediatepostoperativeperiod (IPP),thecase evolvedwithsignificant reductionof facial deformityandresolutionofvisualcomplaints.An ophthal-mologic examination in the IPP revealed full recovery of visualacuityandextrinsicocularmotility;afewdayslater the patient showed improvement of pupillary reflex. A one-yearfollow-upshowedthatthepatientremains asymp-tomatic,withamildresidualfacialasymmetryandnoother complaints.

Discussion

Amaurosisis the most serious orbital complicationof any mucocele.Thelargestpublishedseriesofcasesof mucoce-leswithorbitalinvolvementshowedanincidenceof18.8% ofamaurosis,whenonly patientswithorbitalinvolvement wereconsidered.2 _{Smaller serieshave demonstrated} vari-ationfrom 6.7% to40% for amaurosis, always considering onlypatientswithorbitalinvolvement.2---5_{Whenallcasesof} mucocelewereconsidered,loweroccurrenceofamaurosis hasbeenreported,approximately5%.2

Almost 30% of all mucoceles may erode toward the orbit, and the fronto-ethmoidal mucoceles are primarily responsible for this extension, followed by frontal and ethmoidal mucoceles.1 _{Maxillary mucoceles, in addition} to being rare, seldom invade the orbit.1---5 _{Even in the} faceof aninvasion,usuallytheonlyorbitalsymptomsare eye pain,3,5 _proptosis,3 _{or diplopia.}3 _{A reportof} amauro-sis caused by maxillary mucocele occurred in a patient who had been subjected toskeletal surgery (Le Fort III), where this previous procedure, by fracturing the orbital floor and sinus, could have facilitated both the devel-opment of the mucocele and its extension toward the orbit.4 _{The patient of the present maxillary mucocele} reportwasnotinvolved withprevious surgery,thus revea-ling a case of atypical development of a relatively rare disease.

Final

considerations

Visualloss:ararecomplicationofmaxillarysinusmucocele 453

loss.Earlydiagnosisfollowedbyanappropriateandprompt treatmentcanleadtoimmediaterecoveryofvision.

Conflict

of

interests

Theauthorsdeclarenoconflictofinterests.

References

1.Scangas GA, GudisDA, Kennedy DW. The natural history and clinicalcharacteristicsofparanasalsinusmucoceles:aclinical review.IntForumAllergyRhinol.2013;3:712---7.

2.KimYS,KimK,LeeJG,YoonJH,KimCH.Paranasalsinus mucoce-leswithophthalmologicmanifestations:a17-yearreviewof96 cases.AmJRhinolAllergy.2011;25:272---5.

3.Loo JL, Looi AL, Seah LL. Visual outcomes in patients with paranasal mucoceles. Ophthal Plast Reconstr Surg. 2009;25:126---9.

4.PatelPA,WarrenSM,McCarthyJG.Maxillarymucocelewith prop-tosisand visual impairment:a latecomplicationofLeFortIII distraction.JCraniofacSurg.2013;24:2000---2.