State University of New York, Upstate Medical University, Syracuse, NY, USA.
J Investig Med High Impact Case Rep. 2020 Jan-Dec;8:2324709620950113. doi: 10.1177/2324709620950113.
A 42-year-old woman with juvenile idiopathic arthritis was treated with anakinra, corticosteroids, and hydroxychloroquine when she developed chronic hypoxic respiratory myopathy. She was admitted to the intensive care unit for acute hypercapnic respiratory failure and required prolonged intubation, subsequent tracheostomy, and long-term ventilatory support due to multiple failed spontaneous breathing trials after discontinuation of anakinra and steroids. Muscle biopsy revealed type II fiber atrophy with the accumulation of autophagosomes and vacuoles presenting as curvilinear bodies, elevated MHC class I antigen expression, and infiltration by CD68+ macrophages and CD8+ T cells. Type II fiber atrophy was attributed to corticosteroid use and curvilinear bodies due to blockade of autophagy by hydroxychloroquine. After hydroxychloroquine was discontinued, the patient recovered to her prehospitalization baseline.
一位 42 岁的女性患有幼年特发性关节炎，在出现慢性低氧性呼吸肌病时接受了阿那白滞素、皮质类固醇和羟氯喹治疗。她因急性高碳酸血症呼吸衰竭而住进重症监护病房，需要长期插管，随后进行气管切开术，并长期接受通气支持，因为在停用阿那白滞素和皮质类固醇后多次尝试自主呼吸失败。肌肉活检显示 II 型纤维萎缩，伴有自噬体和空泡的积累，表现为曲线形体，MHC Ⅰ类抗原表达升高，CD68+巨噬细胞和 CD8+T 细胞浸润。II 型纤维萎缩归因于皮质类固醇的使用，而曲线形体则归因于羟氯喹阻断自噬。停用羟氯喹后，患者恢复到住院前的基线水平。
