Treatment of hyperinsulinemic hypoglycemia because of diffuse nesidioblastosis with nifedipine after surgical therapies in a newborn
Treatment of hyperinsulinemic hypoglycemia because of diffuse nesidioblastosis with nifedipine after surgical therapies in a newborn
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DOI：
10.1515/jpem-2013-0091
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发表时间：
2013-11-01
影响因子：
1.4
通讯作者：
Keskin, Mehmet
中科院分区：
文献类型：
--
作者：
Koklu, Esad;Ozkan, Keramettin Ugur;Keskin, Mehmet
最近的研究表明，钙通道阻滞剂在治疗新生儿持续性高胰岛素血症中具有一定的作用。我们报告一例持续高胰岛素血症的新生儿，手术治疗后仅用口服硝苯地平成功治疗。一名4天大的男婴被诊断为顽固性低血糖和自闭症。只有以每分钟20 mg/kg的速度静脉滴注葡萄糖才能维持正常血糖。新生儿持续性高胰岛素低血糖是由于低血糖时血浆胰岛素浓度不适当升高(44mU/L)所致。药物治疗只导致静脉血糖需求的轻微减少；进行了85-90%的胰腺切除术，组织学上证实为弥漫性新生血管母细胞增生症。然而，尽管在第一次胰腺切除后进行了所有的药物治疗，高胰岛素低血糖仍然存在，第二次进行了95%的胰腺切除。在第二次胰腺切除术后，持续的高胰岛素血症用生长抑素和二氮嗪治疗，但导致静脉血糖需求没有减少。我们报告一例婴儿，在两次胰腺次全切除后出现持续性低血糖，但随后用硝苯地平(每天2毫克/公斤)治疗后血糖恢复正常。患者口服硝苯地平后已出院。钙通道阻滞剂可安全有效地治疗复发性高胰岛素血症。
Recent studies have demonstrated a role for calcium channel blocking agents in the treatment of persistent hyperinsulinemic hypoglycemia of newborns. We report a newborn infant with persistent hyperinsulinemic hypoglycemia whom we successfully treated with oral nifedipine alone after surgical therapies. A 4-day-old male infant was referred with intractable hypoglycemia and seziures. Normoglycaemia could be maintained only by the intravenous infusion of glucose at a rate of 20 mg/kg per minute. Persistent hyperinsulinemic hypoglycemia of newborn was diagnosed from an inappropriately raised plasma insulin concentration (44 mU/L) at the time of hypoglycemia. Medical treatments led to only a mild reduction in the intravenous glucose requirement; an 85-90% pancreatectomy was performed and histological "diffuse nesidioblastosis" was confirmed. However, despite all the medical treatments after the first pancreatectomy, the hyperinsulinemic hypoglycemia persisted and a second 95% pancreatectomy was performed. After the second pancreatectomy, persistent hyperinsulinemic hypoglycemia was treated with somatostatin and diazoxide, but led to no reduction in the intravenous glucose requirement. We report the case of an infant who had persistent hypoglycemia after two subtotal pancreatic resections but subsequently became normoglycemic on treatment with nifedipine (2 mg/kg per day). The patient was discharged home on oral nifedipine. Calcium channel blocking agents cuold be used with efficacy and safety in recurrent persistent hyperinsulinemic hypoglycemia.