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• Vol.59 No.7
• CLINICA NEUROL, 59: 431−435, 2019
• Vol.59 No.7 contents

Case Report

A case of anti-titin antibody positive nivolumab-related necrotizing myopathy with myasthenia gravis

Aiko Isami, M.D.¹⁾, Ayaka Uchiyama, M.D.¹⁾, Yuichi Shimaoka, M.D., Ph.D.²⁾, Shigeaki Suzuki, M.D., Ph.D.³⁾, Izumi Kawachi, M.D., Ph.D.⁴⁾ and Nobuya Fujita, M.D., Ph.D.¹⁾

¹⁾Department of Neurology, Nagaoka Red Cross Hospital
²⁾Department of Respiratory medicine, Nagaoka Red Cross Hospital
³⁾Department of Neurology, Keio University School of Medicine
⁴⁾Department of Neurology, Brain Research Institute, Niigata University

A 53-year-old man suffering from squamous cell lung cancer presented with bilateral ptosis and bulbar palsy a month after initial treatment with the immune checkpoint inhibitor nivolumab. The symptoms showed worsening from midday, suggesting myasthenia gravis (MG), although anti-AChR antibody was negative. Although no muscle weakness was detected, the CK level was elevated to 5,255 IU/l, and MRI of the thigh revealed inflammation of the bilateral rectus femoris muscle. A muscle biopsy showed signs of necrotizing myopathy with expression of sarcolemmal HLA class I and accumulation of macrophages, CD4, CD8, and CD20-positive lymphocytes. Positivity for anti-titin antibody, one of the anti-striated muscle antibodies, was evident. The patient was diagnosed as having nivolumab-related necrotizing myopathy with myasthenia gravis, an immune-related adverse event (irAE). Treatment with prednisolone rapidly ameliorated the symptoms, and the serum CK level normalized. There have been several reports of nivolumab-related myositis with MG. On the basis of the muscle pathology and antibody data, we were able to clarify that necrotizing myopathy was related to the pathogenesis of this case.
Full Text of this Article in Japanese PDF (1011K)

(CLINICA NEUROL, 59: 431−435, 2019)
key words: nivolumab, immune-related adverse events (irAE), myasthenia gravis, myopathy, anti-striated muscle antibody

(Received: 11-Jan-19)