Anti-Jo-1 antibody-positive patients show a characteristic necrotizing perifascicular myositis

被引：119

作者：

Mescam-Mancini, Lenaig ^{[1
]}

Allenbach, Yves ^{[2
,3
]}

Hervier, Baptiste ^{[2
,4
]}

Devilliers, Herve ^{[5
]}

Mariampillay, Kuberaka ^{[2
]}

Dubourg, Odile ^{[6
]}

Maisonobe, Thierry ^{[6
]}

Gherardi, Romain ^{[7
]}

Mezin, Paulette ^{[1
]}

Preusse, Corinna ^{[3
]}

Stenzel, Werner ^{[3
]}

Benveniste, Olivier ^{[2
]}

机构：

[1] CHU Grenoble, Dept Pathol, Inst Pathol, F-38043 Grenoble, France

[2] Univ Paris 06, Dept Med Interne & Immunol Clin, Ctr Reference Malad Neuromusculaires Paris Est, Hop La Pitie Salpetriere,AP HP,DHU I2B,INSERM UMR, Paris, France

[3] Charite, Dept Neuropathol, D-13353 Berlin, Germany

[4] UPMC, INSERM CIMI Paris, INSERM UMRS 1135, Paris, France

[5] Hop Dijon, Dept Med Interne, Dijon, France

[6] Hop La Pitie Salpetriere, AP HP, Ctr Reference Malad Neuromusculaires Paris Est, Lab Neuropathol, Paris, France

[7] Univ Paris Est Creteil Val de Marne UPEC, INSERM, U955, Inst Mondor Rech Biomed,IFR10, F-94010 Creteil, France

来源：

BRAIN | 2015年 / 138卷

关键词：

anti-synthetase; idiopathic inflammatory myopathies; dermatomyositis; polymyositis; necrotizing myopathies; IDIOPATHIC INFLAMMATORY MYOPATHY; ANTISYNTHETASE SYNDROME; AUTOANTIBODIES; IMMUNE; DERMATOMYOSITIS; MUSCLE; CLASSIFICATION; POLYMYOSITIS; FEATURES;

D O I：

10.1093/brain/awv192

中图分类号：

R74 [神经病学与精神病学];

学科分类号：

摘要：

Idiopathic inflammatory myopathies can be classified as polymyositis, dermatomyositis, immune-mediated necrotizing myopathy, sporadic inclusion body myositis or non-specific myositis. Anti-Jo-1 antibody-positive patients are assigned to either polymyositis or dermatomyositis suggesting overlapping pathological features. We aimed to determine if anti-Jo-1 antibody-positive myopathy has a specific morphological phenotype. In a series of 53 muscle biopsies of anti-Jo-1 antibody-positive patients, relevant descriptive criteria defining a characteristic morphological pattern were identified. They were tested in a second series of anti-Jo-1 antibody-positive patients and compared to 63 biopsies from patients suffering from other idiopathic inflammatory myopathies. In anti-Jo-1 antibody-positive patients, necrotic fibres, which strongly clustered in perifascicular regions, were frequently observed. Sarcolemmal complement deposition was detected specifically in perifascicular areas. Inflammation was mainly located in the perimysium and around vessels in 90.6%. Perimysial fragmentation was observed in 90% of cases. Major histocompatibility complex class I staining was diffusely positive, with a perifascicular reinforcement. Multivariate analysis showed that criteria defining perifascicular pathology: perifascicular necrosis, atrophy, and perimysial fragmentation allow the distinction of anti-Jo-1 antibody-positive patients, among patients suffering from other idiopathic inflammatory myopathies. Anti-Jo-1 antibody-positive patients displayed perifascicular necrosis, whereas dermatomyositis patients exhibited perifascicular atrophy.

引用

页码：2485 / 2492

页数：8

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