Selective loss of Purkinje cells in a patient with anti-gliadin-antibody-positive autoimmune cerebellar ataxia 英文参考文献.docVIP

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Selective loss of Purkinje cells in a patient with anti-gliadin-antibody-positive autoimmune cerebellar ataxia 英文参考文献.doc

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Selective loss of Purkinje cells in a patient with anti-gliadin-antibody-positive autoimmune cerebellar ataxia 英文参考文献

Nanrietal.DiagnosticPathology2011,6:14 /content/6/1/14 CASE REPORT OpenAccess SelectivelossofPurkinjecellsinapatientwith anti-gliadin-antibody-positiveautoimmune cerebellarataxia KazunoriNanri1*,MakotoShibuya2,TakeshiTaguchi1,AkiraHasegawa1,NobuyukiTanaka1 Abstract Thepatientwasan84-year-oldwomanwhohadtheonsetoftruncalataxiaatage77andahistoryofBasedow’s disease.Herataxicgaitgraduallydeteriorated.Shecouldnotwalkwithoutsupportatage81andshewas admittedtoourhospitalatage83.Gaze-evokednystagmusanddysarthriawereobserved.Mildataxiawas observedinalllimbs.Herdeeptendonreflexandsenseofpositionwerenormal.IgAanti-gliadinantibody,IgG anti-gliadinantibody,anti-SS-A/Roantibody,anti-SS-B/Laantibodyandanti-TPOantibodywerepositive.A conventionalbrainMRIdidnotshowobviouscerebellaratrophy.However,MRIvoxelbasedmorphometry(VBM) andSPECT-eZISrevealedcorticalcerebellaratrophyandreducedcerebellarbloodflow.IVIgtreatmentwas performedandwasmoderatelyeffective.Afterherdeathatage85,thepatientwasautopsied.Neuropathological findingswereasfollows:selectivelossofPurkinjecells;noapparentdegenerativechangeintheefferentpathways, suchasthedentatenucleiorvestibularnuclei;noprominentinflammatoryreaction.Fromthesefindings,we diagnosedthiscaseasautoimmunecerebellaratrophyassociatedwithglutenataxia.All3autopsiespreviously reportedonglutenataxiahavenotedinfiltrationofinflammatorycellsinthecerebellum. Inthiscase,wepostulatedthattheinfiltrationofinflammatorycellswasnotfoundbecausethepatient’ scondition wasbasedonhumoralimmunity.Theclinicalconditionsofglutenataxiahavenotyetbeenproperlyelucidated, butareexpectedtoberevealedasthenumberofautopsiedcasesincreases. Background The autopsy after her death at 85 showed selective Ithasrecentlybeenreportedthatautoimmunecerebel- loss of Purkinje cells and a diagnosis of autoimmune lar ataxias, such as gluten ataxia [1] and anti-glutamic cerebellaratrophywasconfirmed.However,thepatholo- acid decarboxylase (GAD)-antibody-positive cerebellar gical findings differed to