Rie Karasawa1, Kazuo Yudoh1, Toshiko Sato1, Megumi Tanaka1, Mayumi Tamaki1, Sara E Sabbagh2,3, Terrance P O'Hanlon4, Payam Noroozi-Farhadi4, Ira N Targoff5, Willy A Flegel6, Andrew L Mammen2, Frederick W Miller4, Mark D Hicar7, Lisa G Rider4, James N Jarvis7,8. 1. Department of Frontier Medicine, St. Marianna University School of Medicine, Kawasaki, Japan. 2. Muscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health (NIH), Bethesda, MD. 3. Division of Rheumatology, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, WI. 4. Environmental Autoimmunity Group, Clinical Research Branch, National Institute of Environmental Health Sciences, National Institutes of Health (NIH), Bethesda, MD. 5. Oklahoma City VA Health Care System, University of Oklahoma Health Sciences Center, and Oklahoma Medical Research Foundation, Oklahoma City, OK. 6. Department of Transfusion Medicine, NIH Clinical Center, National Institutes of Health (NIH), Bethesda, MD. 7. Department of Pediatrics, Jacobs School of Medicine and Biomedical Sciences. 8. Genetics, Genomics, & Bioinformatics Program, Jacobs School of Medicine and Biomedical Sciences, University at Buffalo, Buffalo, NY, USA.
Abstract
OBJECTIVES: JDM is an inflammatory myopathy characterized by prominent vasculopathy. AECAs are frequently detected in inflammatory and autoimmune diseases. We sought to determine whether AECAs correlate with clinical features of JDM, and thus serve as biomarkers to guide therapy or predict outcome. METHODS: Plasma samples from 63 patients with JDM, 49 patients with polyarticular JIA and 40 juvenile healthy controls were used to detect anti-heat shock cognate 71 kDa protein (HSC70) autoantibodies, a newly identified AECA, in ELISA assays. Clinical features were compared between JDM patients with and without anti-HSC70 autoantibodies. RESULTS: Anti-HSC70 autoantibodies were detected in 35% of patients with JDM, in 0% of patients with JIA (P < 0.0001) and in 0% of healthy donors (P < 0.0001). Both the presence of cutaneous ulcers (59% vs 17%, P < 0.002) and the use of wheelchairs and/or assistive devices (64% vs 27%, P < 0.007) were strongly associated with anti-HSC70 autoantibodies in JDM. High scores on the severity of myositis damage measures at the time of measurement of anti-HSC70 autoantibodies and an increased number of hospitalizations were also associated with anti-HSC70 autoantibodies. Intravenous immunoglobulin therapy was used more often in anti-HSC70 autoantibody-positive patients. CONCLUSION: Anti-HCS70 autoantibodies are detected frequently in children with JDM and are novel myositis-associated autoantibodies correlating with disease severity.
OBJECTIVES: JDM is an inflammatory myopathy characterized by prominent vasculopathy. AECAs are frequently detected in inflammatory and autoimmune diseases. We sought to determine whether AECAs correlate with clinical features of JDM, and thus serve as biomarkers to guide therapy or predict outcome. METHODS: Plasma samples from 63 patients with JDM, 49 patients with polyarticular JIA and 40 juvenile healthy controls were used to detect anti-heat shock cognate 71 kDa protein (HSC70) autoantibodies, a newly identified AECA, in ELISA assays. Clinical features were compared between JDM patients with and without anti-HSC70 autoantibodies. RESULTS: Anti-HSC70 autoantibodies were detected in 35% of patients with JDM, in 0% of patients with JIA (P < 0.0001) and in 0% of healthy donors (P < 0.0001). Both the presence of cutaneous ulcers (59% vs 17%, P < 0.002) and the use of wheelchairs and/or assistive devices (64% vs 27%, P < 0.007) were strongly associated with anti-HSC70 autoantibodies in JDM. High scores on the severity of myositis damage measures at the time of measurement of anti-HSC70 autoantibodies and an increased number of hospitalizations were also associated with anti-HSC70 autoantibodies. Intravenous immunoglobulin therapy was used more often in anti-HSC70 autoantibody-positive patients. CONCLUSION: Anti-HCS70 autoantibodies are detected frequently in children with JDM and are novel myositis-associated autoantibodies correlating with disease severity.
Authors: Ross E Petty; Taunton R Southwood; Prudence Manners; John Baum; David N Glass; Jose Goldenberg; Xiaohu He; Jose Maldonado-Cocco; Javier Orozco-Alcala; Anne-Marie Prieur; Maria E Suarez-Almazor; Patricia Woo Journal: J Rheumatol Date: 2004-02 Impact factor: 4.666
Authors: Sara Sabbagh; Iago Pinal-Fernandez; Lisa G Rider; Andrew Lee Mammen; Takayuki Kishi; Ira N Targoff; Frederick W Miller Journal: Ann Rheum Dis Date: 2019-04-24 Impact factor: 19.103
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