Lee Jun-Ku, Kang Yun Kyung, Wang Pei Wei, Hong Soo Min
Department of Orthopaedic Surgery, Seoul Paik Hospital, Inje University College of Medicine, Seoul, Korea.
Department of Pathology, Seoul Paik Hospital, Inje University College of Medicine, Seoul, Korea.
J Bone Metab. 2020 Feb;27(1):71-75. doi: 10.11005/jbm.2020.27.1.71. Epub 2020 Feb 29.
In terms of management of Paget's disease of bone (PDB), early diagnosis and proper management achieving remission is essential with lifelong specialist follow-up. We present the case of a 40-year-old woman with PDB affecting mainly the distal extremities (ankle and wrist). The patient visited our hospital in 2012 with heel pain. Plain radiography revealed osteoporosis, and a bone scan revealed hot uptake. Initial laboratory investigations showed normal serum calcium, 25-hydroxy-vitamin D, and parathyroid hormone levels; however, osteocalcin, C-terminal telopeptide of type I collagen, and bone alkaline phosphatase levels were elevated. A bone mineral density scan showed T- and Z-scores of -2.5 and -2.7, respectively, and bisphosphonate treatment was initiated. Biopsy performed on the calcaneal lateral wall revealed inconclusive findings. Follow-up biopsy on the left distal radius was performed 7 years later to investigate wrist pain, and this examination led to a final diagnosis as PDB. We suggest inconclusive biopsy result during the early phase of PDB and highly recommend follow-up evaluation in osteoporosis with atypical behavior.
就骨佩吉特病(PDB)的管理而言,早期诊断和实现缓解的适当管理以及终身专科随访至关重要。我们报告一例40岁女性的病例,其PDB主要累及远端肢体(踝关节和腕关节)。该患者于2012年因足跟疼痛就诊于我院。X线平片显示骨质疏松,骨扫描显示放射性摄取增加。初始实验室检查显示血清钙、25-羟基维生素D和甲状旁腺激素水平正常;然而,骨钙素、I型胶原C末端肽和骨碱性磷酸酶水平升高。骨密度扫描显示T值和Z值分别为-2.5和-2.7,遂开始双膦酸盐治疗。对跟骨外侧壁进行活检,结果不明确。7年后对左桡骨远端进行随访活检以调查腕部疼痛,此次检查最终确诊为PDB。我们提示PDB早期活检结果不明确,并强烈建议对具有非典型表现的骨质疏松症进行随访评估。