Pembrolizumab on pre-existing inclusion body myositis: a case report
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BMC Rheumatology
CASE REPORT
Open Access
Pembrolizumab on pre-existing inclusion body myositis: a case report Naohiro Uchio1, Atsushi Unuma1, Toshiyuki Kakumoto2, Masao Osaki2, Yoshitaka Zenke3, Kenichi Sakuta4, Akatsuki Kubota1, Yoshikazu Uesaka2, Tatsushi Toda1 and Jun Shimizu1,5*
Abstract Background: Cases of exacerbation of pre-existing neuromuscular diseases induced by immune checkpoint inhibitors (ICIs) have rarely been reported because patients with autoimmune diseases have generally been excluded from ICI therapy due to the increased risk of exacerbation. We describe the first case of an elderly patient who experienced exacerbation of a previously undiagnosed sporadic inclusion body myositis (sIBM), the most common myopathy in the geriatric population, which was triggered by anti-programmed cell death-1 therapy. Case presentation: A 75-year-old man who was receiving pembrolizumab presented with limb weakness. Three years prior, he had noticed slowly progressive limb weakness, but he received no diagnosis. After the first infusion of pembrolizumab, his creatine kinase (CK) levels had increased. The neurological examination and muscle biopsy findings confirmed the diagnosis of sIBM and suggested exacerbation of sIBM induced by pembrolizumab. After the patient’s CK levels decreased, pembrolizumab was restarted. The tumor progressed after its treatment with pembrolizumab. The patient died after 15 months of follow-up. Conclusions: In patients with slowly progressive limb weakness, sIBM should be explored before ICI therapy. In addition, if patients show high CK levels after ICI introduction, it is necessary to confirm whether they have sIBM in order to avoid unnecessary immunosuppressive therapies and assess whether they can tolerate ICI reintroduction. Keywords: Inclusion body myositis, Immune checkpoint, Pembrolizumab, Immune-related adverse events
Background Immune checkpoint inhibitors (ICIs) potentiate T-cell activity and show dramatic efficacy in treating cancers, but they may also induce immune-related adverse events (irAEs) resembling autoimmune diseases [1–3]. Several reports have documented ICI-induced exacerbation of pre-existing autoimmune diseases [2, 4, 5]. Regarding neuromuscular irAEs, a case with myasthenia gravis [4] and another with interferon-alpha-induced myositis [2] showing ICI-induced exacerbation have been reported. * Correspondence: [email protected] 1 Department of Neurology, Graduate School of Medicine, The University of Tokyo, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8655, Japan 5 Department of Physical Therapy, Tokyo University of Technology, 5-23-22, Nishikamata, Ota-ku, Tokyo 144-8535, Japan Full list of author information is available at the end of the article
Because of the rarity of ICI introduction in pre-existing autoimmune diseases, whether each ICI type exacerbates particular autoimmune diseases remains unclear. Herein, we report a case of presumed exacerbation of previously undiagnosed sporadic inclusion body myositis (sIBM) in an elderly patient treated with anti-pro
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