BrazJOtorhinolaryngol.2015;81(1):113---114
Brazilian
Journal
of
OTORHINOLARYNGOLOGY
www.bjorl.org
CASE
REPORT
Headache
induced
by
the
sphenoid
sinus
mucocele
夽
Cefaleia
atribuída
a
uma
mucocele
no
seio
esfenoidal
Kyu
Eun
Lee,
Kyung
Soo
Kim
∗DepartmentofOtorhinolaryngology-HeadandNeckSurgery,Chung-AngUniversityCollegeofMedicine,Seoul,SouthKorea
Received5February2013;accepted23February2013 Availableonline3July2014
Introduction
Althoughheadacheisawell-knownpresentingsymptomof isolatedsphenoidsinus disease,1itusuallydelaysits
diag-nosis, since sinonasal symptoms are lacking; thus, these patientsinitiallypresenttoneurologistsandneurosurgeons toassesstheheadache.
The mucocele,which isabenign,cysticlesion linedby respiratoryepithelium,isoneofthediseaseentities origi-natingfromthesphenoidsinus.However,thesphenoidsinus istheleastcommonsiteoftheparanasalsinuses, represent-ing1---2%.2
The authors report a case of isolated sphenoid sinus mucocelethat only presented with headachefor over six months,andalsoreviewtheliteratureregardingthe patho-physiologyofsuchheadaches.
Case
report
A 23-year-old, previously healthy male presented with right-sided headache over six months. The headache was intermittent and dull, but recently it had become more
夽 Please cite this article as: Lee KE, Kim KS. Headache
inducedbythesphenoidsinusmucocele.BrazJOtorhinolaryngol. 2015;81:113---4.
∗Correspondingauthor.
E-mail:99-21045@hanmail.net(K.S.Kim).
severeinintensity.The neurologicalexaminationwas nor-mal. In order to exclude other symptomatic headaches, brainmagneticresonanceimaging(MRI)wasperformedby hisneurologist, which showed no intracerebral structural lesions.However,itrevealedacysticlesionintheright sphe-noidsinus,withouterosionofthebonywall(Fig.1).Since hisneurologistthoughtthislesion wasnotassociatedwith theheadache,itwasdecidedtoobservechangesinsizefor sixmonths.Althoughnon-steroidalanti-inflammatorydrugs (NSAIDs)hadsomeeffect,hisheadachewasnotcompletely relieved,andrecentlybecamemoreaggravated.Sixmonths later, brain MRI was re-evaluated because of his ongoing andaggravatedheadache;itshowednochangeinsize,but minimalchangesinsignalintensity(Fig.1).Theprovisional diagnosisattributedtheheadachetotherightsphenoidsinus mucocele.Aftertheright-sidedsphenoidotomy,abrownish cysticmasswasfoundandexcisedcompletely(Fig.1).The patienthasbeen completelyrelieved ofhissymptoms for thelast12months.
Discussion
The most common symptom of sphenoidsinus mucoceles isheadachecausedby lesiondimensions.1,3 The headache
induced by sphenoid sinus mucoceles usually occurs as a bilateral,slowlyprogressiveworseningpain,andcan tem-porarilyberelievedbyNSAIDs.4
Therearethreeimportantpointstoexplainthe mecha-nismoftheheadache.
http://dx.doi.org/10.1016/j.bjorl.2014.05.029
114 LeeKE,KimKS
Figure1 (A---D)Themuoceleiscomposedoftwoseparateparts.T1-weightedmagneticresonanceimaging(MRI)scan(A,axial) showsahyperintensepart(blackstar: highprotein content)andamildlyhyperintensepart(whitearrow:highwatercontent). T2-weightedMRIscans(B,axial;C,coronal)showsahypointensepartcentrally,withasurroundingrimofhyperintensemucosa (blackstar)andapartofahighsignal.MRAscan(D)showsnormalarterialvasculatureandamucocele.(E---G)Sixmonthslater,brain MRIwasre-evaluated.Itshowednochangesinsize,butminimalchangesinsignalintensityofthelesion.TheT2-weightedsignalis lowerthanexpectedinthepartofhighwatercontent(whitearrow),indicatingtheincreaseofproteinwithinthemucocele.(H) Afterwidesphenoidotomy,theblackishcysticmass(blackstar)wasfoundandcompletelyremovedbyendoscopicsurgery.
First,the headache induced bythe presence of muco-celeisgenerallyofthesomatictype,deep,visceral,andits natureisclearlyinteroceptive,characteristicofthemucosa oftheparanasalsinuses.Theheadacheinducedbysphenoid sinusmucocelesisdirectly relatedtomechanicalpressure onthesinusanatomy,suchasexpansionandinfection.Itcan bereferredtoanypartofthehead,sincethenervesupply ofthesphenoidsinuscomesfromtheophthalmicdivisionof thetrigeminalnerve.5
Second, the headache is presumably due to stretching oftheduracoveringtheplanum sphenoidaleandthefloor oftheanteriorcranialfossa,becausemucocelesare expan-sileandlocallydestructive,withtheabilitytoresorbbone, causingerosionsofthesinuswall.5
Last, the headache is associated with inflammatory cytokines such as IL-1, IL-6, TNF alpha, and PGE2 found in mucoceles. Studies have found that cytokinesare one oftheprincipalcausesofosteolysisofthebone surround-ingmucoceles.6Cytokinesareinvolvedininflammation,in
modulation of the pain threshold, and also in trigeminal sensitization.
Mucoceles may have variable signal intensities on MRI dependingontheirproteincontent,inspissation,and pos-sible superinfection.2 In the present patient, there was
a hyperintense lesion on T1-weighted MRI, which showed lower than expected enhancement on T2-weighted MRI, consistentwithhighproteincontents.2,6Onfollow-upMRI,
thereweresomechangesinsignalintensity,suggestingan increaseofproteincontentwithinthemucoceleassociated withincreaseofinflammatorycytokines.
Final
remarks
Althoughthe mucocelemaybeofsmallsize,theclinician shouldconsideritinthecaseofintractableheadachewith regardtodifferentialdiagnosis.
Conflicts
of
interest
Theauthorsdeclarenoconflictsofinterest.
References
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3.GiovannettiF,FiliaciF,RamieriV,UngariC.Isolatedsphenoid sinus mucocele: etiologyand management. JCraniofac Surg. 2008;19:1381---4.
4.Bauer B, Evers S, Lindörfer HW, Schuierer G, Henningsen H, HusstedtIW.Headachecausedbyasphenoidmucocelebut pre-sentingasanergotamine-inducedheadache.Headache.1997;37: 460---2.
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