Whitson Heather E, Lobaugh Bruce, Lyles Kenneth W
GRECC, VA Medical Center, Durham, NC 27710, USA.
Bone. 2006 Oct;39(4):954-8. doi: 10.1016/j.bone.2006.04.032. Epub 2006 Jun 12.
Bisphosphonate therapy is a common and effective treatment for Paget's disease of bone, osteoporosis, hypercalcemia of malignancy and cancer metastatic to bone. Clinically significant hypocalcemia has not been reported in patients with Paget's disease of bone and normal parathyroid function treated with an aminobisphosphonate. We treated a 52-year-old woman with polyostotic Paget's disease of bone (serum alkaline phosphatase level-1971 IU/L [normal 31-110 IU/L]), who had not previously received bisphosphonates, with daily oral 30 mg risedronate, oral 1000 mg elemental calcium and oral 400 IU cholecalciferol. After 10 days of treatment, she developed severe hypocalcemia (5.4 mg/dL [normal 8.7-10.2 mg/dL]), requiring hospitalization and support with 5 days of intravenous calcium gluconate. On the day risedronate treatment began, her PTH was low normal at 14 pg/mL (normal 12-72 pg/mL), consistent with a relatively suppressed PTH axis due to high bone turnover. Her vitamin D level was within normal limits (serum 25(OH)D 19 ng/mL [normal 8-38 ng/mL]), although possibly not optimally repleted. We hypothesize that this case represents an example of hungry bone syndrome in a patient with extensive Paget's disease of bone who received risedronate, causing acute suppression of bone resorption while elevated bone formation rates continued. In the year following her recovery, the patient was successfully treated with slowly titrated anti-resorptive therapy (subcutaneous calcitonin followed by titrated doses of risedronate), and is now clinically well. Physicians should be aware of the potential for hypocalcemia when patients with polyostotic Paget's disease and markedly elevated indicators of bone remodeling are initiated on powerful anti-resorptive therapy.
双膦酸盐治疗是骨Paget病、骨质疏松症、恶性肿瘤高钙血症及骨转移癌的一种常用且有效的治疗方法。在骨Paget病且甲状旁腺功能正常的患者中，使用氨基双膦酸盐治疗尚未报告有临床意义的低钙血症。我们对一名52岁的多骨型骨Paget病女性患者（血清碱性磷酸酶水平为1971 IU/L[正常范围31 - 110 IU/L]）进行治疗，该患者此前未接受过双膦酸盐治疗，给予每日口服30 mg利塞膦酸盐、口服1000 mg元素钙和口服400 IU胆钙化醇。治疗10天后，她出现了严重低钙血症（5.4 mg/dL[正常范围8.7 - 10.2 mg/dL]），需要住院并静脉输注葡萄糖酸钙支持治疗5天。在开始利塞膦酸盐治疗当天，她的甲状旁腺激素（PTH）处于正常低限，为14 pg/mL（正常范围12 - 72 pg/mL），这与由于高骨转换导致的相对受抑制的PTH轴一致。她的维生素D水平在正常范围内（血清25(OH)D 19 ng/mL[正常范围8 - 38 ng/mL]），尽管可能补充得并不理想。我们推测该病例代表了一名患有广泛骨Paget病的患者在接受利塞膦酸盐治疗后发生饥饿骨综合征的例子，即骨吸收被急性抑制而骨形成率持续升高。在她康复后的一年里，该患者通过缓慢滴定的抗吸收治疗（皮下注射降钙素，随后滴定剂量的利塞膦酸盐）成功治愈，目前临床状况良好。当多骨型骨Paget病且骨重塑指标明显升高的患者开始接受强效抗吸收治疗时，医生应意识到发生低钙血症的可能性。
