ID | 59975 |
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Yamamura, Yuriko
Department of Nephrology, Rheumatology, Endocrinology and Metabolism Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Matsumoto, Yoshinori
Department of Nephrology, Rheumatology, Endocrinology and Metabolism Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Tadokoro, Koh
Department of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
Ohta, Yasuyuki
Department of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Sato, Kota
Department of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Yamashita, Toru
Department of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Yamamura, Masahiro
Center for Rheumatology, Okayama Saiseikai General Hospital
Sada, Ken-Ei
Department of Nephrology, Rheumatology, Endocrinology and Metabolism Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Abe, Koji
Department of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Wada, Jun
Department of Nephrology, Rheumatology, Endocrinology and Metabolism Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences
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Abstract | Background: Statin-associated necrotizing myopathy (SANM) is a rare autoimmune disorder caused by administration of statins. SANM is characterized by weakness due to necrosis and regeneration of myofibers. Here we report the first case of SANM with acute respiratory failure treated with noninvasive pressure support ventilation in addition to immunosuppressants.
Case presentation: A 59-year-old woman who had been treated with 2.5 mg/day of rosuvastatin calcium for 5 years stopped taking the drug 4 months before admission to our hospital due to elevation of creatine kinase (CK). Withdrawal of rosuvastatin for 1 month did not decrease the level of CK, and she was admitted to our hospital due to the development of muscle weakness of her neck and bilateral upper extremities. Anti-3-hydroxy-3-methylglutaryl coenzyme A reductase antibodies were positive. Magnetic resonance imaging showed myositis, and muscle biopsy from the right biceps brachii muscle showed muscle fiber necrosis and regeneration without inflammatory cell infiltration, suggesting SANM. After the diagnosis, she received methylprednisolone pulse therapy (mPSL, 1 g/day × 3 days, twice) and subsequent oral prednisolone therapy (PSL, 30 mg/day for 1 month, 25 mg/day for 1 month and 22.5 mg/day for 1 month), leading to improvement of her muscle weakness. One month after the PSL tapering to 20 mg/day, her muscle weakness deteriorated with oxygen desaturation (SpO2: 93% at room air) due to hypoventilation caused by weakness of respiratory muscles. BIPAP was used for the management of acute respiratory failure in combination with IVIG (20 g/day × 5 days) followed by mPSL pulse therapy (1 g/day × 3 days), oral PSL (30 mg/day × 3 weeks, then tapered to 25 mg/day) and tacrolimus (3 mg/day). Twenty-seven days after the start of BIPAP, she was weaned from BIPAP with improvement of muscle weakness, hypoxemia and hypercapnia. After she achieved remission with improvement of muscle weakness and reduction of serum CK level to a normal level, the dose of oral prednisolone was gradually tapered to 12.5 mg/day without relapse for 3 months. Conclusions: Our report provides new insights into the role of immunosuppressants and biphasic positive airway pressure for induction of remission in patients with SANM. |
Keywords | Noninvasive pressure support ventilation
Statin-associated necrotizing myopathy
BIPAP
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Published Date | 2020-06-03
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Publication Title |
BMC Pulmonary Medicine
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Volume | volume20
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Issue | issue1
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Publisher | BMC
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Start Page | 156
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ISSN | 1471-2466
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Content Type |
Journal Article
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language |
English
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OAI-PMH Set |
岡山大学
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Copyright Holders | © The Author(s). 2020
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File Version | publisher
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Related Url | isVersionOf https://doi.org/10.1186/s12890-020-01195-7
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License | http://creativecommons.org/licenses/by/4.0/
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