Leunbach T L, Hansen A T, Madsen M, Cipliene R, Christensen P S, Schou A J
Department of Pediatrics, Aalborg University Hospital, Reberbansgade 15, 9000 Aalborg, Denmark.
H.C. Andersen Children's Hospital, Odense University Hospital, Sdr. Boulevard 29, 5000 Odense, Denmark.
Bone Rep. 2021 Mar 4;14:100761. doi: 10.1016/j.bonr.2021.100761. eCollection 2021 Jun.
We report a boy with hypercalcemia due to neonatal severe hyperparathyroidism (NSHPT) caused by a compound heterozygous mutation in the calcium sensing receptor (CaSR) managed successfully on a type II calcimimetic drug. The hypercalcemia was temporarily treated by hyperhydration, bisphosphonate and calcium depleted milk. At 29 days of age cinacalcet was introduced. The starting dose was 0.5 mg/kg/day and was subsequently titrated to the point of efficacy (5.2 mg/kg/day) when a persuasive reduction in parathyroid hormone and calcium concentrations was observed. We propose a trial of type II calcimimetics in newborns with NSHPT irrespective of the genetic mutation and advocate that residual functionality of the CaSR predict the drug efficacy.
我们报告了一名因钙敏感受体(CaSR)复合杂合突变导致新生儿严重甲状旁腺功能亢进症(NSHPT)而出现高钙血症的男孩,其通过使用II型拟钙剂成功得到治疗。高钙血症通过补液、双膦酸盐和低钙牛奶进行了临时治疗。在29日龄时开始使用西那卡塞。起始剂量为0.5mg/kg/天,随后在观察到甲状旁腺激素和钙浓度有明显降低时,滴定至有效剂量(5.2mg/kg/天)。我们建议对患有NSHPT的新生儿进行II型拟钙剂试验,无论其基因突变情况如何,并主张CaSR的残余功能可预测药物疗效。