Sakamoto A, Oda Y, Nagayoshi Y, Iwakiri K, Tamiya S, Iwamoto Y, Tsuneyoshi M
Second Department of Pathology, Faculty of Medicine, Kyushu University, Fukuoka, Japan.
Arch Orthop Trauma Surg. 2001;121(1-2):104-8. doi: 10.1007/s004020000187.
A 47-year-old woman suffered from gait disturbance due to back pain and muscle weakness. Laboratory data showed serum hypophosphatemia, elevated alkaline phosphatase, and a normal level of ionized calcium. Radiological examinations revealed multiple pathologic fractures in the ribs and pubic rami. She had had no episode of familial or any other notable disorder, and so she was initially treated with medication for adult-onset osteomalacia. However, 19 years later (when she was 66 years old), she noticed a soft-tissue tumor in her buttock. The tumor was excised. The histological features were those of glomangiopericytoma characterized by both glomus tumor-like and hemangiopericytoma-like structures. After removal of the tumor, her symptoms disappeared immediately. Laboratory data normalized 8 months later. To our knowledge, this is the first report of oncogenic osteomalacia caused by glomangiopericytoma.
一名47岁女性因背痛和肌肉无力而出现步态障碍。实验室检查数据显示血清低磷血症、碱性磷酸酶升高,以及离子钙水平正常。影像学检查发现肋骨和耻骨支多处病理性骨折。她没有家族性或任何其他显著疾病史,因此最初接受了成人型骨软化症的药物治疗。然而,19年后(她66岁时),她注意到臀部有一个软组织肿瘤。该肿瘤被切除。组织学特征为血管球性血管外皮细胞瘤,其特点是具有血管球瘤样和血管外皮细胞瘤样结构。肿瘤切除后,她的症状立即消失。8个月后实验室检查数据恢复正常。据我们所知,这是首例由血管球性血管外皮细胞瘤引起的致癌性骨软化症的报告。