Anti-HMGCR Autoantibodies in Juvenile Idiopathic Inflammatory Myopathies Identify a Rare but Clinically Important Subset of Patients

被引：51

作者：

Tansley, Sarah L. ^{[1
,2
]}

Betteridge, Zoe E. ^{[2
]}

Simou, Stefania ^{[3
]}

Jacques, Thomas S. ^{[4
,5
]}

Pilkington, Clarissa ^{[6
]}

Wood, Mark ^{[7
]}

Warrier, Kishore ^{[8
]}

Wedderburn, Lucy R. ^{[5
,9
,10
]}

McHugh, Neil J. ^{[2
,11
]}

机构：

[1] Royal United Hosp Fdn Trust, Royal Natl Hosp Rheumat Dis, MRCP, London, England

[2] Univ Bath, Bath, Avon, England

[3] UCL, Inst Child Hlth, London, England

[4] UCL, Inst Child Hlth, FRCPath, MRCP, London, England

[5] GOSH, London, England

[6] GOSH, FRCP, FRCPCH, London, England

[7] Leeds Childrens Hosp, MRCPCH, Leeds, W Yorkshire, England

[8] Nottingham Univ Hosp NHS Trust, Nottingham, England

[9] UCL, Inst Child Hlth, FRCP, London, England

[10] Adolescent Rheumatol UCL, UCLH, Arthrit Res UK Ctr, London, England

[11] Royal United Hosp Fdn Trust, Royal Natl Hosp Rheumat Dis, FRCPath, FRCP, London, England

来源：

JOURNAL OF RHEUMATOLOGY | 2017年 / 44卷 / 04期

基金：

英国惠康基金;

关键词：

PEDIATRIC DERMATOMYOSITIS/POLYMYOSITIS; PEDIATRIC RHEUMATIC DISEASES; AUTOANTIBODIES; PROGNOSIS; NECROTIZING MYOPATHIES; NATIONAL REGISTRY; DERMATOMYOSITIS; VALIDATION; EXPERIENCE; REPOSITORY; CHILDREN; IRELAND; DISEASE; COHORT;

D O I：

10.3899/jrheum.160871

中图分类号：

R5 [内科学];

学科分类号：

1002 ; 100201 ;

摘要：

Objective. We aimed to establish the prevalence and clinical associations of anti-HMG-CoA-reductase (anti-HMGCR) in a large UK cohort with juvenile myositis. Methods. There were 381 patients investigated for anti-HMGCR using ELISA. Results. Anti-HMGCR autoantibodies were detected in 4 patients (1%). These children had no or minimal rash and significant muscle disease. Muscle biopsies were considered distinctive, with widespread variation in fiber size, necrotic fibers, and chronic inflammatory cell infiltrates; all had prolonged elevation of creatine kinase and all ultimately received biologic therapies. Conclusion. Anti-HMGCR in UK children with myositis are associated with severe disease that is poorly responsive to standard treatment.

引用

页码：488 / 492

页数：5

共 14 条

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