CorrespondingauthorDepartmentofNeurosciencesDivisionofPsychiatryBrainandEmotionLaboratoryLeuvenBELLONIIHerestraat49bus10223000LeuvenBelgiumEmailaddressjanvandenstockmedkuleuvenbeJVa ID: 379757
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LettertotheEditorAstrangefaceinthemirror.Face-selectiveself-misidentiÞcationinapatientwithrightlateralizedoccipito-temporalhypo-metabolismJanVandenStock,BeatricedeGelder,Franc¸ois-LaurentDeWinterKoenVanLaereandMathieuVandenbulckeBrainandEmotionLaboratoryLeuven(BELL),DivisionofPsychiatry,DepartmentofNeurosciences,KULeuven,Leuven,BelgiumTilburgUniversity,CognitiveandAffectiveNeuroscienceLaboratory,Tilburg,TheNetherlandsAthinoulaA.MartinosCentreforBiomedicalImaging,MassachusettsGeneralHospital,HarvardMedicalSchool,Charlestown,MA,USAKULeuven,DivisionofNuclearMedicine,UZGasthuisberg,Leuven,Belgium1.IntroductionDelusionsareintriguingneuropsychiatricphenomena.SometypesareassociatedwithspeciÞcneurologicalconditions,particularlymonothematicdelusions,characterizedbyasingleabnormalbelief(Coltheartetal.,2011).Nexttothewell-knownCapgrasdelusion(CapgrasandReboul-Lachaux,1923)(thebeliefthatpeoplearereplacedbydoubles),anotherremarkabledelusionisthemirrored-selfmisidentiÞcationormirrorsign(inabilitytorecognizeoneÕsself-imageinthemirror).Althoughdelusionslikethemirrorsigncanoccurinmultiplestagesofsyndromeslikeschizophreniaanddementia(ConnorsandColtheart,2011),abetterunderstandingofmonothematicdelusionsrequiresfocusingonthesymptomratherthanthesyndrome(Coltheart,2011).Thetwo-factortheory(TFT)proposedbyColtheartetal.(2011)statesthatidentiÞcationofboththespeciÞcorigin(whatcausedtheparticulardelusion?)andpersistence(whyisnÕttheabnormalbeliefcorrected?)isrequiredtounderstandthedelusionalnatureandgenesis.TwomirrorsigncaseshavebeenstudiedandinterpretedfromtheTFT(Breenetal.,20002001).Foronepatient,impairedface-processingwaswithheldastheoriginatingunderlyingneuro-psychologicalabnormality,whereasfortheotherpatientitwasmirroragnosia.Thesecondfactor(persistence)iscommonforalldelusionsandrelatestoimpairedbeliefevaluation,hypo-theticallyassociatedwithrightlateralprefrontalcortexpathology(Coltheart,20072010Coltheartetal.,2007Langdonetal.,2008).Here,wedescribeneuropsychologicalandneuro-imaginginvestigationsinpatientMVdisplayingmirrorsign.2.CasereportMV(85,female)wasreferredtoourgeriatricpsychiatryunitaftershowingsignsofhallucinations:caregiversheardhertalkingtoanotherpersonwhennobodywasaround.Addi-tionally,MVdisplayedaphantomboarderdelusion:shewasconvincedawomanwaslivinguninvitedinherhouseandlaterinthehospitalatMVÕsexpenses.MVhadnoneurologicalorpsychiatrichistoryanddisplayednoclinicalfeaturesofparkinsonism.OphthalmologicexaminationrevealednodeÞ-cits.NeuropsychologicalevaluationshowedmilddeÞcitsofmemory,executive,languageandvisuo-spatialfunctions.TheMiniMentalStateExamination(MMSE-score)equaled21/30.MagneticResonanceImaging(MRI)indicatedcortico-subcorticalatrophyand[18F]FluorodeoxyglucosePositronEmissionTomography(FDG-PET)showedclearrightlateralizedlateralprefrontal,parietalandparticularlyoccipito-temporalhypo-metabolism(Fig.1).AdiagnosisofprobablemildAlz-heimerÕsdiseasewaswithheld.Duringadmission,itbecameclearthatMVdisplayedamirrorsign.SheidentiÞedhermirroredfaceastheÒphantomboarderÓ(SupplementaryVideo).Confrontedwithhermirroredfaceimage,MVexplainedshehadnoideawherethewomancamefrom,butshealways Correspondingauthor.DepartmentofNeurosciences,DivisionofPsychiatry,BrainandEmotionLaboratoryLeuven(BELL),O&NII,Herestraat49bus1022,3000Leuven,Belgium.E-mailaddress:jan.vandenstock@med.kuleuven.be(J.VandenStock). Availableonlineat Journalhomepage:www.elsevier.com/locate/cortex cortexxxx(2012)1 Pleasecitethisarticleinpressas:VandenStockJ,etal.,Astrangefaceinthemirror.Face-selectiveself-misidentiÞcationinapatientwithrightlateralizedoccipito-temporalhypo-metabolism,Cortex(2012),doi:10.1016/j.cortex.2012.03.003seefrontmatter2012ElsevierSrl.Allrightsreserved.10.1016/j.cortex.2012.03.003 dressedjustlikeher.Weadministeredthemirror-marktestGallop,1970)inwhichamarkwasunnoticeablyplacedonMVÕsface.Subsequently,MVwaspositionedbeforeamirror.Whensubjectswithintactself-faceawarenessdiscoverthemarkontheirfaceafterlookinginthemirror,theytypicallyspontane-ouslytrytotouchorremoveit.Remarkably,MVdidnotreactinanywaytothemark.MVnoticedthemarkontheotherwomanÕsfacewhenshewasexplicitlyquestionedaboutit.ShehadnodifÞcultyaccuratelylocatingthemarkwhenitwasunnoticeablyplacedonthepersonnexttoher(SupplementaryVideo).MVwasalsoabletorecognizeotherpersonsinthemirror.Supplementaryvideorelatedtothisarticlecanbefoundatdoi:10.1016/j.cortex.2012.03.003Toinvestigatewhethertheself-misidentiÞcationcomprisedalsoherbody,wepositionedMVinfrontofatil-tedmirror,reßectingherwholebodyexceptthehead.MVthenreportedshesawherselfinthemirror,basedonherposture.However,assoonassheapproachedthemirrorandalsosawherfacereßected,MVreportedtheotherwomanappeared(SupplementaryVideo).Wepresentedonebyonefacephotographsofcelebrities(20),closefamily(8),MV(10),andunfamiliarpersons(20).WeaskedMVtoindicatewhetherthefacewasfamiliarorunfamiliar.Comparedtoanelderlycontrolgroup(23;meanage63.7years;standarddeviation3.80),thefamiliarityjudgmentsofMVwereimpairedforcelebrityfaces(.001),butnotforunfamiliarfaces(.15).Sheperformedatchanceforfamilyandselffaces.ShecategorizedrecentphotographsofherownfaceasÔfamiliarÕ;however,shespontaneouslyiden-tiÞedherastheÔphantomboarderÕ.Subsequently,wepre-sentedthecelebrityfaceswiththreecelebritynamesprintedunderneathandinstructedMVtomatchthefacewithaname.Sheperformedwithinthenormalrange(.09).UsingfunctionalMRI(fMRI),welocalizedthebrainareasthatselectivelyrespondtobodiesandfaces(Supplementary).Althoughfaceandbody-selectivebrainareasaretypicallyobservedintherighthemisphere,weobservedactivationintheleftbutnotrightfusiformfaceareawhencomparingtheperceptionoffaceswithobjects.Contrarily,weobservedactivationoftherightbutnotleftfusiformbodyareawhencomparingbodieswithobjects.3.DiscussionMVÕsface-processingabilitiesindicatethattheoriginatingfactorinherdelusionpertainstoselectivelyimpaired Fig.1StatisticalmapofbrainactivationofpatientMV(A)inresponsetobodiescomparedtoobjectsandfacescomparedtoobjects(stimulusexamplesshownontop).TheregionsdeÞnedbythewhiteandredcontoursdemarktheareascontainingthepooledindividualactivationmapsintheleftandrightfusiformgyrusforbodiescomparedtoobjects(whitecontours)andfacescomparedtoobjects(redcontours)in31controlsubjects(.001).TheactivationmapsareplottedonaventralviewofacorticalreconstructionofMVÕsbrain,showinggyriinlightgrayandsulciindarkgray.B:StereotacticsurfaceprojectionsofanFDG-PETbrainscaninMV,withareasofsigniÞcanthypo-metabolismsuperimposed(-scoreswithrespecttonormaldatasetof30controlsubjects). cortexxxx(2012)1 Pleasecitethisarticleinpressas:VandenStockJ,etal.,Astrangefaceinthemirror.Face-selectiveself-misidentiÞcationinapatientwithrightlateralizedoccipito-temporalhypo-metabolism,Cortex(2012),doi:10.1016/j.cortex.2012.03.003 face-familiarityrecognition.Thisextendsapreviousreportofabnormalface-processinginapatientwithmirrorsign,whocontrarilyshowedover-familiarization(Breenetal.,2001).Ourreportadditionallyshowsthatfamousface-namematchingmayremainrelativelyintact.Althoughthedelusiononlyper-tainedtoherownface,theface-familiaritydeÞcitalsoappliedtootherfaces,includingcloserelatives.Possibly,MVreliedonnon-facecueslikebodyorvoicetorecognizefamiliarpersons,whereasself-identiÞcationinthemirrorprimarilyreliesonface-cues.InlinewithMVÕsfamiliarity-selectiveface-deÞcit,patientswithCapgrasdelusionfailtoexhibittheenhancedskinconductanceresponsetofamiliarfaces(Brighettietal.,2007Ellisetal.,1997HirsteinandRamachandran,1997).ThedisconnectionbetweenMVÕsface-identityandface-familiarityprocessingislikelyrelatedtotherightoccipito-temporalhypo-metabolism.Theself-faceandbodyrecognitionshowsaninterestinganalogywiththefMRI-results.Intactself-bodyrecognitionisassociatedwithrightlateralizedbody-selectiveactivation,whereasdeÞcientself-facerecognitionisassoci-atedwithabsenceofface-selectiveactivityinrightoccipito-temporalcortex.ThisextendspreviousÞndingsthatself-recognitioninvolvestherighthemisphere(Keenanetal.,2001).Concluding,ourdatasuggestanimpairedmappingofaperceivedfaceonthestoredmemoryrepresentation(resultinginimpairedface-familiarity),withrelativelypreservedpersonidentiÞcationbynameorbodycues.MVadditionallyshowsrightdorsalprefrontalhypo-metabolism,presumablyunderlyingherimpairedbeliefevaluation(Coltheartetal.,2011J.V.d.S.isapost-doctoralresearchersupportedbyFWO-Vlaanderen.B.d.GispartlysupportedbyEuropeanCommission(COBOL)Grant,FP6FET-Open,COBOLgrantenFP7-FET-Opengrants.KVLisSeniorClinicalInvestigatorofFWO-Vlaanderen.SupplementarymaterialSupplementarydatarelatedtothisarticlecanbefoundonlinedoi:10.1016/j.cortex.2012.03.003BreenN,CaineD,andColtheartM.Mirrored-selfmisidentiÞcation:Twocasesoffocalonsetdementia.,7(3):239254,2001.BreenN,CaineD,ColtheartM,RobertsC,andHendyJ.TowardsanunderstandingofdelusionsofmisidentiÞcation:FourcaseMindandLanguage,15(1):74110,2000.BrighettiG,BonifacciP,BorlimiR,andOttavianiC.ÒFarfromtheheartfarfromtheeyeÓ:EvidencefromtheCapgrasdelusion.CognitiveNeuropsychiatry,12(3):189197,2007.CapgrasJandReboul-LachauxJ.Illusiondessosiesdansundeliresystematizechronique.BulletindelaSocieCliniquedeMe,11:616,1923.ColtheartM.Cognitiveneuropsychiatryanddelusionalbelief.QuarterlyJournalofExperimentalPsychology,60(8):1041ColtheartM.Theneuropsychologyofdelusions.AnnalsoftheNewYorkAcademyofSciences,1191:1626,2010.ColtheartM.Themirrored-selfmisidentiÞcationdelusion.,1(6):521523,2011.ColtheartM,LangdonR,andMcKayR.Schizophreniaandmonothematicdelusions.SchizophreniaBulletin,33(3):642ColtheartM,LangdonR,andMcKayR.Delusionalbelief.AnnualReviewofPsychology,62:271298,2011.ConnorsMHandColtheartM.Onthebehaviourofseniledementiapatientsvis-a-visthemirror:Ajuriaguerra,StrejilevitchandTissot(1963).Neuropsychologia,49(7):1692,2011.EllisHD,YoungAW,QuayleAH,andDePauwKW.ReducedautonomicresponsestofacesinCapgrasdelusion.oftheRoyalSocietyBBiologicalSciences,264(1384):1085GallopGG.Chimpanzees:Self-recognition.,167(914):87,1970.HirsteinWandRamachandranVS.Capgrassyndrome:Anovelprobeforunderstandingtheneuralrepresentationoftheidentityandfamiliarityofpersons.oftheRoyalSocietyBBiologicalSciences,264(1380):437KeenanJP,NelsonA,OÕConnorM,andPascual-LeoneA.Self-recognitionandtherighthemisphere.,409(6818):305,LangdonR,McKayR,andColtheartM.Thecognitiveneuropsychologicalunderstandingofpersecutorydelusions.InFreemanD,GaretyP,andBentallR(Eds),PersecutoryDelusions:Assessment,TheoryandTreatment.London:OxfordUniversityPress,2008:221 cortexxxx(2012)1 Pleasecitethisarticleinpressas:VandenStockJ,etal.,Astrangefaceinthemirror.Face-selectiveself-misidentiÞcationinapatientwithrightlateralizedoccipito-temporalhypo-metabolism,Cortex(2012),doi:10.1016/j.cortex.2012.03.003