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Japanese

A Case of Immune-Mediated Necrotizing Myopathy Associated with Primary Sjögren Syndrome Nobutaka Takahashi 1 , Akihiro Nishida 1 , Jun Tsugawa 1 , Motoshige Okajima 1 , Shinsuke Fujioka 1 , Yoshio Tsuboi 1 1Department of Neurology, Fukuoka University Faculty of Medicine Keyword: 免疫介在性壊死性ミオパチー , 原発性シェーグレン症候群 , 抗SS-A抗体 , immune-mediated necrotizing myopathy , IMNM , primary Sjögren syndrome , SjS , anti-SS-A antibody pp.183-187
Published Date 2021/2/1
DOI https://doi.org/10.11477/mf.1416201731
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Abstract

A 66-year-old woman visited our hospital complaining of shortness of breath during exertion and progressive weakness in all her limb muscles. On admission, we noted muscle weakness in her trunk and in her proximal limb muscles, although, her muscle MRI showed no remarkable findings. However, her serum CK level (2,747 U/L) was above the normal range. Histopathological examination of muscle biopsy, performed from the left biceps brachii muscle, revealed immune-mediated necrotizing myopathy (IMNM). Her serum samples were negative for myositis-associated autoantibodies (MAAs), anti-SRP, and HMGCR antibodies. However, as the anti-SS-A antibody level in her serum was high (53.2 U/mL), we conducted the salivary gland biopsy and the Schirmer test on her eyes. We found lymphocytes infiltration in her salivary gland tissue, and thus, she was diagnosed with primary Sjögren syndrome (SjS). We also observed necrotizing myopathy associated with the SjS. Following her treatment with oral steroids, her symptoms and CK level improved. Although, inflammatory myositis frequently occurs in association with general collagen diseases, necrotizing myopathy has rarely been observed secondary to SjS. We report here this rare case study along with the review of the relevant literature.

(Received June 24, 2020; Accepted October 12, 2020; Published February 1, 2021)


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電子版ISSN 1344-8129 印刷版ISSN 1881-6096 医学書院

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