Rinsho Shinkeigaku (Clinical Neurology)

Case Report

A case of anti-Th/To antibody-positive systemic sclerosis with muscle symptoms and interstitial pneumonia

Shiori Kikuchi, M.D.1), Jun Sawada, M.D., Ph.D.1), Tsukasa Saito, M.D., Ph.D.1), Takayuki Katayama, M.D., Ph.D.1), Daisuke Fujishiro, M.D.2), Ichizo Nishino, M.D., Ph.D.3), Naoyuki Hasebe, M.D., Ph.D.1)

1) Internal Medicine, Cardiovascular, Respiratory and Neurology Division, Asahikawa Medical University
2) Division of Metabolism and Biosystemic Science, Department of Medicine, Asahikawa Medical University
3) Department of Neuromuscular Research, National Institute of Neuroscience, National Center of Neurology and Psychiatry (NCNP)

A 62-year-old Japanese man with swollen fingers and walking difficulty due to myalgia and muscle weakness in proximal limb muscles was admitted to our hospital. Serum creatine kinase was remarkably increased (7,380 U/l) and rapidly progressing interstitial pneumonia developed. Muscle biopsy showed necrotic and regenerating fibers without mononuclear infiltration and fibrosis. Anti-Th/To antibodies were detected in the serum, and anti-Th/To antibody-positive systemic sclerosis was diagnosed. Anti-Th/To antibody-positive sclerosis-associated myopathy has not yet been reported in the literature. The present case suggests that anti-Th/To antibody-positive systemic sclerosis can be accompanied by immune-mediated necrotizing myopathy and be effectively treated with immunotherapy comprising corticosteroids, tacrolimus and immunoglobulin.
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(CLINICA NEUROL, 61: 228−233, 2021)
key words: anti-Th/To antibody, systemic sclerosis, necrotizing myopathy, muscle biopsy

(Received: 1-Jul-20)