Shakesprere Jonathan, Shafiq Ramsha, Madahar Inderpreet, Quinn Hugh B, Thakkar Yashan, Haider Adnan
Department of Internal Medicine, West Virginia University School of Medicine, Morgantown, WV 26505, USA.
Section of Endocrinology and Metabolism, Department of Internal Medicine, West Virginia University School of Medicine, Morgantown, WV 26505, USA.
JCEM Case Rep. 2024 Jun 3;2(6):luae096. doi: 10.1210/jcemcr/luae096. eCollection 2024 Jun.
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.
家族性低钙血症性高钙血症(FHH)的特征是轻度至中度高钙血症、血清甲状旁腺激素(PTH)水平正常或升高以及相对低钙尿症。据报道,拟钙剂西那卡塞可减轻FHH患者的症状负担并降低血清钙水平。我们报告了2例患有慢性高钙血症的成年男性,最初怀疑为原发性甲状旁腺功能亢进。尿钙筛查和基因检测证实两名患者均患有FHH。第一例患者在服用西那卡塞期间QTc缩短恢复正常,第二例患者血清钙和PTH水平降低且无高钙血症症状。拟钙剂治疗可能适用于FHH患者,尤其是那些有高钙血症症状、血清钙水平高于正常1 mg/dL(0.25 mmol/L)或有心律失常风险的患者。西那卡塞治疗总体耐受性良好,随着时间推移,显著降低了2例成年FHH患者的血清钙和PTH水平。拟钙剂治疗在管理FHH患者的持续性高钙血症和潜在不良事件方面显示出前景。潜在障碍包括治疗期限不确定、费用以及可能的不良反应。