Department of Orthopaedic Surgery, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand.
Division of Endocrinology and Metabolism, Department of Medicine, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand.
BMC Musculoskelet Disord. 2021 May 14;22(1):443. doi: 10.1186/s12891-021-04326-1.
Osteitis fibrosa cystica is the classic manifestation of primary hyperparathyroidism (PHPT), occurs after prolonged exposure of bone to high serum parathyroid hormone (PTH) level. It has become increasingly rare due to early detection of PHPT.
A 37-year-old woman was referred to our institution for fixation of multiple fractures of upper and lower extremities that had been reoccurring in the past 5 years. Her medical history showed right-shoulder, left-elbow, and right-femur fractures after a fall 5 years previously. One month ago, she sustained fractures of the right distal humerus, left tibia, and left femur without history of trauma. Upon arrival to our hospital, a thorough review of her plain radiographs demonstrated brown tumors at multiple sites, along with a salt-and-pepper appearance of the skull and a rugger-jersey spine, compatible with osteitis fibrosa cystica. Patient was diagnosed with PHPT, confirmed by high-corrected serum calcium (13.6 [8.6-10.0] mg/dl), low serum phosphate (2.2 [2.5-4.5] mg/dL), high serum alkaline phosphatase (1482 [35-105] U/L), and significantly elevated parathyroid hormone (PTH 3850 [15-65] pg/mL). A histologically confirmed, 2.5-cm parathyroid adenoma was removed by parathyroidectomy. Ten days later, closed reduction and internal fixation of the left proximal femoral shaft was performed. Pain and ambulation were significantly improved 6 months postoperatively. At the 1.5-year follow-up, fracture unions and complete mineralization of brown tumors were noted; the patient could ambulate with neither pain nor an assistive device.
PHPT has become more asymptomatic in countries where routine calcium screening is performed. Nevertheless, the classic skeletal involvement, osteitis fibrosa cystica, should not be overlooked, particularly in young patients who present with a low-energy fracture.
纤维囊性骨炎是甲状旁腺功能亢进症（PHPT）的典型表现，发生在骨长时间暴露于高甲状旁腺激素（PTH）水平后。由于 PHPT 的早期发现，这种情况变得越来越少见。
一名 37 岁女性因过去 5 年来反复发生的上下肢多处骨折而被转至我院。她的病史显示，5 年前因跌倒导致右肩、左肘和右股骨骨折。一个月前，她无外伤史发生右肱骨远端、左胫骨和左股骨骨折。当她到达我院时，对其平片进行全面审查显示多处棕色瘤，以及颅骨的椒盐状外观和 Rugger-Jersey 脊柱，符合纤维囊性骨炎的表现。患者被诊断为 PHPT，通过高校正血清钙（13.6 [8.6-10.0] mg/dl）、低血清磷酸盐（2.2 [2.5-4.5] mg/dL）、高血清碱性磷酸酶（1482 [35-105] U/L）和明显升高的甲状旁腺激素（PTH 3850 [15-65] pg/mL）得到证实。通过甲状旁腺切除术切除了一个 2.5 厘米的甲状旁腺腺瘤，组织学得到证实。甲状旁腺切除术后 10 天，进行左股骨近端干闭合复位内固定。术后 6 个月疼痛和活动度明显改善。在 1.5 年的随访中，观察到骨折愈合和棕色瘤完全矿化；患者无需疼痛或辅助设备即可行走。
在常规进行钙筛查的国家，PHPT 变得更加无症状。然而，不应忽视经典的骨骼受累，即纤维囊性骨炎，尤其是在出现低能量骨折的年轻患者中。
