Toriu Naoya, Ueno Toshiharu, Mizuno Hiroki, Sekine Akinari, Hayami Noriko, Hiramatsu Rikako, Sumida Keiichi, Yamanouchi Masayuki, Hasegawa Eiko, Suwabe Tatsuya, Hoshino Junichi, Sawa Naoki, Takaichi Kenmei, Fujii Takeshi, Hasegawa Tomoka, Amizuka Norio, Yanagita Motoko, Ubara Yoshifumi
Nephrology Center, Department of Rheumatology, Toranomon Hospital, Tokyo, Japan.
Okinaka Memorial Institute for Medical Research, Tokyo, Japan.
Bone Rep. 2018 Dec 10;10:100187. doi: 10.1016/j.bonr.2018.100187. eCollection 2019 Jun.
We report a 48-year-old Japanese man with a brown tumor of the right distal tibia. At the age of 25 years, hemodialysis was initiated due to nail-patella syndrome. Severe secondary hyperparathyroidism and osteoporosis progressed over time, so parathyroidectomy was performed at age 45. Spontaneous fracture of the right distal tibia occurred suddenly at age 48. Imaging studies revealed a bone tumor-like lesion and surgery was performed. The resected specimen was a brown mass consisting of multinucleated giant cells on a fibrous tissue background, and these findings were consistent with a diagnosis of brown tumor. Immunohistochemistry revealed that multinucleated giant cells near areas of bone matrix were positive for tartrate-resistant acid phosphatase and cathepsin K, but the majority of the giant cells in the lesion were negative for these markers. Even after parathyroidectomy, brown tumor should be considered in the differential diagnosis of bone tumor-like lesions in patients on long-term dialysis. This case suggests that osteoclast activation may not contribute to development of brown tumors, although these lesions are generally considered to arise from subperiosteal bone resorption related to osteoclast overactivity in patients with hyperparathyroidism.
我们报告一例48岁的日本男性，患有右胫骨远端棕色瘤。该患者25岁时因指甲-髌骨综合征开始接受血液透析。随着时间的推移，严重的继发性甲状旁腺功能亢进和骨质疏松症逐渐进展，因此在45岁时进行了甲状旁腺切除术。48岁时，右胫骨远端突然发生自发性骨折。影像学检查发现一个骨肿瘤样病变，并进行了手术。切除的标本是一个棕色肿块，在纤维组织背景上可见多核巨细胞，这些表现符合棕色瘤的诊断。免疫组织化学显示，骨基质附近的多核巨细胞对酒石酸抗酸性磷酸酶和组织蛋白酶K呈阳性，但病变中的大多数巨细胞对这些标志物呈阴性。即使在甲状旁腺切除术后，对于长期透析患者的骨肿瘤样病变进行鉴别诊断时，也应考虑棕色瘤。该病例表明，尽管棕色瘤通常被认为是由甲状旁腺功能亢进患者破骨细胞过度活跃导致的骨膜下骨吸收引起的，但破骨细胞激活可能与棕色瘤的发生无关。
