G.P.67 Favorable response to low dose oral methotrexate in acute sarcoid myositis
Review articleOpen access
N.Y. Jung - No affiliation found
2012/10/01 Abstract DOI: 10.1016/j.nmd.2012.06.173
Journal: Neuromuscular Disorders
AbstractSarcoidosis is a multisystemic granulomatous disease of unknown etiology. Diagnosis depends on clinico-radiologic features, histologic proof of non-caseating granulomas, and exclusion of other granulomatous diseases. Patients commonly present with bilateral hilar lymphadenopathy, pulmonary infiltrates, and ocular and skin lesions. Asymptomatic muscle involvement with systemic sarcoidosis occurs in 50–80% of patients, while symptomatic myositis, mostly as chronic form, is in less than 3%. Acute myopathy as an initial manifestation is extremely rare. We recently experienced a rapidly progressive case of acute sarcoid myositis, which showed a favorable response to low dose of oral methotrexate. A 49-year-old woman was admitted with progressive limb weakness for 2 months. Her symptom began with difficulties in climbing stairs. Her weakness extended to arm and neck in 1 month. She reported weight loss and mild dyspnea over months. Her weakness was on proximal arms and legs, showing waddling gait and Gower’s sign. Serum angiotensin-converting enzyme and creatine kinase level was elevated; ESR and CRP were also slight elevated. Needle electromyography showed active myopathic patterns. Muscle biopsy disclosed many non-caseating granulomas and some lymphohistiocytic infiltaration in perimysial space together with multiple necrotic and regenerating fibers. A systemic search for granuloma revealed bilateral hilar and mediastinal lymphnode enlargement on chest computed tomography and whole-body fluorodeoxyglucose positron emission tomography. Corticosteroid therapy is considered the mainstay of treatment for sarcoid myopathy. However, weakness got worse in this case, despite high-dose methylprednisolone followed by oral prednisolone. We added 15 mg/week of oral methotrexate and finally she began to improve slowly with notable improvement in her muscle strength. We recommend methotrexate as an effective second-line treatment option in patients with acute sarcoid myositis.
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