两例症状性家族性高钙血症:对拟骨化疗法的治疗反应。

JCEM case reports Pub Date : 2024-06-03 eCollection Date: 2024-06-01 DOI:10.1210/jcemcr/luae096
Jonathan Shakesprere, Ramsha Shafiq, Inderpreet Madahar, Hugh B Quinn, Yashan Thakkar, Adnan Haider
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引用次数: 0

摘要

家族性低钙尿症(FHH)以轻度至中度高钙血症、血清 PTH 水平正常升高和相对低钙尿为特征。据报道,钙离子拮抗剂西那卡西酮(Cinacalcet)可减轻 FHH 的症状和血清钙水平。我们报告了两名患有慢性高钙血症的成年男性,他们最初被怀疑患有原发性甲状旁腺功能亢进症。尿钙筛查和基因检测证实这两名患者均患有 FHH。第一例患者在服用西那卡塞后QTc缩短恢复正常,第二例患者血清钙和PTH水平下降,但无症状性高钙血症。可为 FHH 患者提供降钙治疗,尤其是有高钙血症症状、血清钙水平高于正常值 1 mg/dL (0.25 mmol/L) 或有心律失常风险的患者。Cinacalcet 治疗总体上耐受性良好,并能随着时间的推移显著降低两名成年 FHH 患者的血清钙和 PTH 水平。在控制 FHH 患者的持续性高钙血症和潜在不良事件方面,降钙治疗已显示出良好的前景。潜在的障碍包括治疗的不确定性、费用和可能的不良反应。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Two Cases of Symptomatic Familial Hypocalciuric Hypercalcemia: Treatment Response to Calcimimetic Therapy.

Familial hypocalciuric hypercalcemia (FHH) is marked by mild to moderate hypercalcemia, normal-elevated serum PTH levels, and relative hypocalciuria. Cinacalcet, a calcimimetic therapy, has been reported to reduce symptom burden and serum calcium levels in FHH. We report 2 adult males with chronic hypercalcemia, with initial concerns for primary hyperparathyroidism. Urine calcium screening and genetic testing confirmed FHH in both patients. Shortened QTc normalized while on cinacalcet in the first patient and reductions in serum calcium and PTH levels without symptomatic hypercalcemia were noted in the second patient. Calcimimetic therapy can potentially be offered to FHH patients, particularly those with hypercalcemia symptoms, serum calcium levels >1 mg/dL (0.25 mmol/L) above normal or at risk of cardiac arrhythmias. Cinacalcet treatment was overall well tolerated and significantly reduced serum calcium and PTH levels in 2 adult FHH patients over time. Calcimimetic therapy has shown promise in managing persistent hypercalcemia and potential adverse events in FHH patients. Potential barriers include indefinite treatment, cost, and possible adverse effects.

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