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CASEREPORTOpenAccessManifestationofasellarhemangioblastomaduetopituitaryapoplexy:acasereportRalphTSchr1*,IstvanVajtai2,RahelSahli3andRolfWSeiler1AbstractIntroduction:Hemangioblastomasarerare,benigntumorsoccurringinanypartofthenervoussystem.
Mostarefoundassporadictumorsinthecerebellumorspinalcord.
However,theseneoplasmsarealsoassociatedwithvonHippel-Lindaudisease.
Wereportararecaseofasporadicsellarhemangioblastomathatbecamesymptomaticduetopituitaryapoplexy.
Casepresentation:An80-year-old,otherwisehealthyCaucasianwomanpresentedtoourfacilitywithsevereheadacheattacks,hypocortisolismandblurredvision.
Amagneticresonanceimagingscanshowedanacutehemorrhageofaknown,stableandasymptomaticsellarmasslesionwithchiasmaticcompressionaccountingforourpatient'sacutevisualimpairment.
Thetumorwasresectedbyatransnasal,transsphenoidalapproachandhistologicalexaminationrevealedacapillaryhemangioblastoma(WorldHealthOrganizationgradeI).
Ourpatientrecoveredwellandsubstitutionaltherapywasstartedforpanhypopituitarism.
Afollow-upmagneticresonanceimagingscanperformed16monthspostoperativelyshowedgoodchiasmaticdecompressionwithnotumorrecurrence.
Conclusions:Areviewoftheliteratureconfirmedsupratentoriallocationsofhemangioblastomastobeveryunusual,especiallywithinthesellarregion.
However,intrasellarhemangioblastomamustbeconsideredinthedifferentialdiagnosisofpituitaryapoplexy.
IntroductionHemangioblastomas(HBLs)arebenign,slowlygrowingandhighlyvasculartumorsofthecentralnervoussys-tem(CNS),accountingforjust1%to2.
5%ofallintra-cranialneoplasms,and7%to12%ofprimarytumorslocatedintheposteriorfossa[1].
InuptooneinfourcasesofHBLthereisanassociationwithvonHippel-Lindau(VHL)disease[2],arareautosomaldominantconditionthatpredisposespatientstomultisystemicneoplasticdisorderssuchasHBLsoftheCNS,retinalangiomas,renalcellcarcinoma,pheochromocytomas,serouscystadenomasandneuroendocrinetumorsofthepancreas.
VHL-associatedHBLstendtooccurinyoungerpatientsandareoftenmultipleinoccurrence[2-4].
SporadicHBLs,however,aremostlysolitarylesionsandpredominantlyfoundwithinthecerebellumorspinalcord.
SupratentorialHBLs,whicharemoreoftenassociatedwithVHLdisease[3,4],arearareentitywithjustover100reportedcasestodate[5].
HBLsori-ginatingfromthesellarorsuprasellarregionareexcep-tional,especiallyincaseswithnoassociationwithVHLdisease.
Wereportherewhatis,tothebestofourknowledge,theseventhsporadiccaseintheliteratureofsellarHBL,whichpresentedwithpituitaryapoplexy.
WealsoreviewtheliteratureoncasesofHBLwithinthesellarandsuprasellarregion.
CasepresentationAn80-year-oldCaucasianwomanwasadmittedtoourhospitalwitha12-yearhistoryofanendocrineinactivesteadysellarmasslesion(13mmindiameter;Figure1A,B).
Ourpatienthadbeenpreviouslyasymptomaticwithnopituitaryhormonedeficiencyorvisualimpair-ments.
Moreover,ourpatienthadamedicalhistoryofgoodhealthwithonlyminorhealthissuesthatincludedhypertensionandosteoporosis.
However,priortohospi-taladmission,shehadrecentlyexperiencedtwosevere*Correspondence:ralph.
schaer@insel.
ch1DepartmentofNeurosurgery,Inselspital,UniversityHospitalBern,3010Bern,SwitzerlandFulllistofauthorinformationisavailableattheendofthearticleSchretal.
JournalofMedicalCaseReports2011,5:496http://www.
jmedicalcasereports.
com/content/5/1/496JOURNALOFMEDICALCASEREPORTS2011Schretal;licenseeBioMedCentralLtd.
ThisisanOpenAccessarticledistributedunderthetermsoftheCreativeCommonsAttributionLicense(http://creativecommons.
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0),whichpermitsunrestricteduse,distribution,andreproductioninanymedium,providedtheoriginalworkisproperlycited.
Figure1MRIimagesofpatient'sbrain.
(A,B)T1-andT2-weightedMRIscanstakentwoyearspriortocurrentpresentation.
(C)T1-weightedMRIscanofpatient'sbrain,revealingapartlyvesicularhyperintense,andslightlyincreased(comparedtoAandB)intrasellarandsuprasellarmassof16mmindiameter,withprogressivecompressionoftheprechiasmaticportionsofheropticnervesbilaterally.
(D)T2-weightedMRIscanshowingthevesicularportionashypointense;normalpituitarytissuecouldnotbeclearlydelineated.
(E,F)TherewasnoevidentenhancementonT1-weightedimagingafterintravenousadministrationofgadolinium.
(G,H)AnMRIscantaken16monthspostoperativelyshowedregulardisplayoftheremainingpituitaryglandwithgoodchiasmaticdecompressionandnosignsoftumorrecurrence.
Schretal.
JournalofMedicalCaseReports2011,5:496http://www.
jmedicalcasereports.
com/content/5/1/496Page2of6headacheattacks;thelastepisodewasaccompaniedbynausea,vomitingandblurredvision.
Hyponatremia(120mEq/L)withlowserumosmolality(247mOsm/L)andhighlyelevatedurineosmolality(695mOsm/L)weredetected.
Anendocrinologicalinvestigationrevealedhypocortisolismwithnootherhormonedisturbances.
Fundoscopyshowednopathologicalfindings.
However,furtherophthalmologicexaminationwithGoldmanperi-metryconfirmedabitemporalhemianopsiaaccentuatedonherrightside.
Herneurologicalexaminationresultswereotherwisenormal.
Aftersubstitutiontherapywithhydrocortisone,ourpatientrapidlyimprovedandherheadachessubsided.
Findingsfromamagneticresonanceimaging(MRI)scanweresuggestiveofanacutehemorrhageofthesellarprocess,consistentwithpituitaryapoplexy(Figure1C-F).
Exceptforanage-consistentvascularleukoence-phalopathy,thediagnosticimagingshowednofurtherpathologicalfindings.
Ourtentativediagnosisatthispointwasapituitaryadenomawithpituitaryapoplexy.
Duetotheseclinicalandradiologicalfindings,thedecisionwasmadetosurgicallyremovethetumor.
Agrosstotalextirpationusingatransnasal,transsphenoi-dalapproachtothepituitarymasswassuccessfullyper-formed.
Intraoperatively,thetumorappearedyellowish-brown,wasrelativelyfirmandwaslocatedwithinasellarhematomacavity,whichwasevacuated.
Postoperatively,ourpatient'svisualfielddeficitsimprovedmarkedlyonclinicalexaminationandGold-manperimetryconfirmedapartialrecoveryofherbitemporalvisualfielddeficits.
Endocrinologicalstudiesshowedpanhypopituitarismwithpartialandtransientdiabetesinsipidus.
Ourpatientreceivedsubstitutiontherapywithhydrocortisone,levothyroxineandtransienttherapywithdesmopressin.
Overall,ourpatientremainedingoodhealthwithasatisfactorylevelofper-formance.
ArepeatMRIscantaken16monthsaftersurgeryshowedgoodchiasmaticdecompressionwithnoresidualtumormass(Figure1G,H).
Theresectedtumorwasexaminedwithlightmicro-scopy,whichrevealedasmall,wellcircumscribed,non-adenomatoustumorsurroundedbyslightlycompressedremnantsofadenohypophysealparenchyma(Figure2A-C).
Thetumorwasrichlyvascularizedwithanobserva-blereticularmeshofthin-walledcapillariesinterspersedwithlargeepithelioid-lookingcells(Figure2D,E).
Paleeosinophiliccytoplasmshowedxanthomatousorvacuo-larchange(Figure2F).
Immunohistochemistrycon-firmedtheexpressionoftheendothelial-associatedmarkersCD31andCD34intheintratumoralcapillaries,althoughnotinthestromalcellsthemselves.
Conversely,thestromalcellswerediffuselyimmunoreactiveforvimentin,withaminorityofcellsalsocoexpressingS100proteinandepithelialmembraneantigen(Figure2G).
Noinflammatoryinfiltratewasdetectedexceptfortheoccasionalmastcell(Figure2H).
Stainingforcyto-keratinstestednegative,asdidtheLangerhans-cell-asso-ciatedmarkerCD1a.
Lessthan1%oflesionalcellnucleiwerelabeledwiththecellproliferation-associatedanti-genKi-67.
Giventheabovefindings,weidentifiedthetumorasanintrapituitaryexampleofcapillaryhemangioblastoma(WorldHealthOrganizationgradeI).
SinceourpatientdisplayednoclinicalstigmataofVHLdisease,genetictestingwasnotperformed.
DiscussionBasedonpreviousstudies,theoccurrenceofsupraten-torialHBLsisthoughttobeintherangeof2%to8%ofallHBLs[3,4,6],accountingfor116reportedcasesfrom1902to2004[5].
Supratentorialtumorsweremostlyfoundinthefrontal,parietalortemporallobes[7].
Nomorethan27reportedcasestodate(includingourpatient'scase)describeHBLsoriginatinginthesellarandsuprasellarregion(see[1]andreferencestherein,and[2,8-11])ofwhich18wereconfirmedwithhisto-pathology(Table1).
Ofthe27cases,onlyseven(26%)weresporadic.
Inaccordancewithpreviousstudies,theaverageageatpresentationofpatientswithsporadicHBLs(52.
4years)wasgreaterthanpatientsaffectedwiththeVHLsyndrome(35.
8years),excludingtwocaseswithpostmortemdiagnosis(Table1,cases1and2)andonecasenotstatingVHLassociation[10].
WhileinformationonclinicalfeaturesisderivedfromreportsofsellarandsuprasellarHBLscausingsymptomsgenerallyrelatedtomasseffect,alongpresymptomaticstagecanbeassumed.
Ofatotalof250patientswithVHLdiseaseenrolledinaprospectivestudy,eightincidentallydiscoveredHBLslocatedinthepituitarystalkremainedstableduringameanfollow-upof41.
4±14months[8].
Also,inourpatient'scase,thesellarlesion,initiallydiag-nosedasanincidentalfindingonMRIperformedforanunrelatedreason,remainedstablefor12years.
Overall,theunexpectednatureandtheunspecificpre-sentationrenderanaccuratepreoperativediagnosisofsporadicHBLschallenging.
Inourpatient,theapoplexyofawellknownsellarmasssuggestedapituitarymacro-adenoma;clinicalapoplexywasobservedin0.
6%to9.
0%ofthesecases[12].
Thetypical,albeitnotpathognomo-nic,radiologicalfeatureofHBLsisthattheycanbeidentifiedasanenhancinglesiononT1-weightedMRIscans.
Thisfindingwaslackinginourcaseduetoacutehemorrhageofthelesion.
ThemainhistologicaldifferentialdiagnosisofHBL,irrespectiveoflocation,ismetastaticclearcellcarci-noma.
Inourpatient,lackofimmunoreactivityforcyto-keratinsalongwithanegligiblylowproliferationindexallowedforthisalternativetobeconfidentlyruledout.
Schretal.
JournalofMedicalCaseReports2011,5:496http://www.
jmedicalcasereports.
com/content/5/1/496Page3of6Inthepeculiarcontextofintrapituitaryoccurrence,wealsoaddressedthepossibilityofxanthomatoushypophy-sitisandLangerhanscellhistiocytosis[13,14].
Thenon-inflammatorycharacterofthelesioninourcasestronglyarguedagainstxanthomatoushypophysitis(orsellarxanthogranuloma).
However,thecircumscribedratherthaninfiltrativepatternofthissolitaryintrapituitarynodule,onedevoidofCD1aimmunoreactivity,wasanintuitiveobstacleagainstseriouslyconsideringLanger-hanscellhistiocytosis.
ConclusionsSupratentorialHBLsarerare,especiallywithinthesellarregionandwithoutanassociationwithVHLdisease.
However,ourpatient'scaseshowsthatintrasellarHBLmustbeconsideredinthedifferentialdiagnosisofpitui-taryapoplexy.
ConsentWritteninformedconsentwasobtainedfromthepatientforpublicationofthiscasereportandanyaccompanyingFigure2OverviewshowingwellcircumscribedHBLnodulepartlysurroundedbyacrescent-shapedmantleofperitumoralpituitaryparenchyma.
(A)OpticalcontrastbetweenthefainteosinophilichueoftheHBLnidusandbrightredgranularqualityofadjacentsomatotrophs.
(B,C)Adjacentsectionplanestreatedwithimmunohistochemistry,showingsegregationofadenohypophysealneuroendocrinecells(B)andmesenchymal-likeimmunophenotype(C)oftheHBLnodule.
(D)Detailviewofboxedareain(A)showstheHBLtobecomprisedofanirregularreticularmeshworkoftortuous,thin-walledcapillariesthattendtobeinterspersedwithpalestromalcells.
(E)Gomori'sreticulinstainhighlightingthebrisktransitionfromtheacinaroutlineofnativeadenohypophysealfollicles(upperthird)tothevascular-dominatedbasementmembranepatternofHBL.
(F)High-powerviewofHBLshowingpolygonalcontoursandcytoplasmicvacuolationofstromalcellsencasedbycapillaries.
Somenuclearpleomorphism,asalsoevidentinthismicroscopicfield,isofnoprognosticsignificance.
(G)Aminorityofstromalcellswerestainedforepithelialmembraneantigen.
(H)ScatteredmastcellsareacharacteristiccomplementofHBL.
Ifnotlabeledotherwise,microphotographshavebeenmadeusinghematoxylinandeosinstain.
Originalmagnifications:(A-C)*30;(D,E,H)*100;(F,G)*400.
Schretal.
JournalofMedicalCaseReports2011,5:496http://www.
jmedicalcasereports.
com/content/5/1/496Page4of6images.
AcopyofthewrittenconsentisavailableforreviewbytheEditor-in-Chiefofthisjournal.
AcknowledgementsWewouldliketothankourpatientforkindlyallowingpublicationofthiscase.
Therewasnofundingforthisstudy.
TheauthorsthankSusanWieting,BernUniversityHospital,DepartmentofNeurosurgery,PublicationsOffice,BernSwitzerlandforproofreadingthefinalmanuscript.
Authordetails1DepartmentofNeurosurgery,Inselspital,UniversityHospitalBern,3010Bern,Switzerland.
2SectionofNeuropathology,InstituteofPathology,UniversityofBern,3010Bern,Switzerland.
3DivisionofEndocrinology,DiabetesandClinicalNutrition,Inselspital,UniversityHospitalBern,3010Bern,Switzerland.
Authors'contributionsRTSwasresponsiblefortheconceptionanddraftingofthemanuscript,andanalyzedandreviewedtheliteraturerelevanttothiscasereport.
IVperformedthehistologicalexaminationandwasamajorcontributortoTable1LiteraturereviewofreportedcasesofHBLconfirmedbyhistopathologyinthesellarregionCaseReferenceAge(years),sexSymptomsLocationVHLSurgeryforsellarHBLFollow-up1[15]84,MNoneIntrasellar(anteriorlobe)YesNone,autopticfindingNA2[16]26,MBlurredvision,headache,ataxiaIntrasellar(anteriorlobe)YesNone,autopticfindingNA3[17]19,MNausea,vertigo,ataxiaSuprasellarYesTotalresectionNA4[18]19,FHeadache,amenorrhea-galactorrheaPituitarystalkNoTotalresectionPanhypopituitarism5[2]35,FHeadache,amenorrhea,diabetesinsipidusPituitarystalkNoYes,detailsNANA6[9]60,FPartialhemianopsiaSuprasellarYesNone,gammakniferadiosurgerySyndromeofinappropriatesecretionofantidiuretichormoneat22-monthfollow-up7[19]11,FHeadache,bitemporalhemianopsia,adrenocorticotropichormoneandgrowthhormonedeficiencyIntrasellarYesSubtotalresectionandadjuvantradiosurgeryHeadacheimproved,noresidualtumor,panhypopituitarism8[20]57,FDiplopia,sixthnervepalsyIntrasellarandsphenoidsinusNoSubtotalresectionPartialimprovementofsixthnervepalsy9[21]20,FPanhypopituitarism,diabetesinsipidusSuprasellarandpituitarystalkYesTotalresectionStablepanhypopituitarism,noresidualtumorat53-monthfollow-up10[22]33,FIrregularmensesPituitarystalkYesSubtotalresectionNoneurologicaldeficitsorpituitarydysfunction,stableresidualtumoratsix-monthfollow-up11[23]62,MVisualdisturbanceSuprasellarNoTotalresectionNA12[24]60,MBitemporalhemianopsia,panhypopituitarismIntrasellarandsuprasellarNoTranssphenoidalbiopsyNA13[25]40,FOligomenorrhea,cognitiveimpairmentIntrasellarandsuprasellarYesSubtotalresectionandgammakniferadiosurgeryNA14[26]54,MHeadache,visuallossSuprasellarNoTotalresectionPartialimprovementofvisualloss,notumorrecurrenceatfive-yearfollow-up15[26]38,MHeadache,visuallossSuprasellarYesSubtotalresectionNA16[1]51,FBlurredvisionPituitarystalkYesTotalresectionPanhypopituitarism,visualacuityimproved17[27]59,FFatigue,visuallossSuprasellarNSTotalresectionPanhypopituitarism,notumorrecurrenceatthree-yearfollow-up18Presentcase80,FHeadache,bitemporalhemianopsia,hypocortisolismIntrasellarNoTotalresectionHeadachesubsided,visualfielddeficitsimproved,panhypopituitarism,notumorrecurrenceat16-monthfollow-upF:femalepatient;M:malepatient;NA:notavailable.
Schretal.
JournalofMedicalCaseReports2011,5:496http://www.
jmedicalcasereports.
com/content/5/1/496Page5of6writingthemanuscript.
RSwaslargelyinvolvedinpatientmanagementandalsocontributedtowritingthearticle.
RWSperformedtheoperativeresectionofthetumorandcriticallyrevisedthearticle.
Allauthorsreadandapprovedthefinalmanuscript.
CompetinginterestsTheauthorsdeclarethattheyhavenocompetinginterests.
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