Department of Medicine, Division of Internal Medicine, University of Florida, Gainesville, Florida, USA
Department of Medicine, Division of Internal Medicine, University of Florida, Gainesville, Florida, USA.
BMJ Case Rep. 2021 Mar 2;14(3):e240828. doi: 10.1136/bcr-2020-240828.
Sarcoidosis is characterised by the formation of noncaseating granulomas classically affecting lungs, lymph nodes and skin. Osteoarticular involvement affects up to 15% of patients; however, acro-osteolysis, destruction involving distal phalanges of fingers and toes, associated with sarcoidosis, is extremely rare. A 44-year-old woman with a history of biopsy-proven sarcoidosis managed with prednisone and methotrexate presented with swelling and pain in the distal fingers of her right hand without skin manifestations. Radiographic imaging showed erosion of distal phalanges on second, third and fifth fingers and bone resorption in bilateral toes. A biopsy of the finger lesions showed noncaseating granulomas consistent with sarcoidosis. She was diagnosed with sarcoid acro-osteolysis and started on adalimumab with clinical and radiographic improvement. While most cases of osteoarticular sarcoidosis are asymptomatic and respond to standard immunosuppression, we present a case with progressive and refractory clinical course. This is the first reported case of sarcoid acro-osteolysis affecting the toes.
结节病的特征是形成非干酪性肉芽肿，经典地影响肺、淋巴结和皮肤。骨关节受累影响多达 15%的患者；然而，与结节病相关的肢端骨溶解，即累及手指和脚趾远端指骨的破坏，极为罕见。一位 44 岁的女性，有活检证实的结节病病史，曾接受泼尼松和甲氨蝶呤治疗，出现右手远端手指肿胀和疼痛，无皮肤表现。影像学检查显示第二、第三和第五手指的远端指骨侵蚀和双侧脚趾的骨吸收。手指病变的活检显示非干酪性肉芽肿，符合结节病。她被诊断为结节病性肢端骨溶解症，并开始接受阿达木单抗治疗，临床和影像学均有改善。虽然大多数骨关节结节病病例是无症状的，且对标准免疫抑制有反应，但我们报告了一个进行性和难治性临床病程的病例。这是首例报道的累及脚趾的结节病性肢端骨溶解症病例。
