病例报告:歌舞伎综合征和新生儿持续性低血糖。

IF 1.1 Q4 PRIMARY HEALTH CARE Journal of Family Medicine and Primary Care Pub Date : 2024-12-01 Epub Date: 2024-12-09 DOI:10.4103/jfmpc.jfmpc_674_24
Osama Y Safdar, Miral M Abddulghfar, Renad N Saaty, Zahrah Bernawi, Reem Babteen, Osama M Felemban
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引用次数: 0

摘要

歌舞伎综合征(KS)是一种罕见的先天性疾病,有两种不同的类型,KS1和KS2,分别具有表观遗传基因KMT2D和KDM6A的变异。它与多种异常有关,如发育迟缓、非典型面部特征、心脏异常、轻微骨骼异常、泌尿生殖系统异常和轻中度智力残疾。这种综合征可导致新生儿低血糖,由高胰岛素血症和电解质异常引起。我们描述了18个月大的歌舞伎综合征2型埃及女孩,自出生以来出现持续低血糖,并伴有尿浓度异常,血气显示学术。她接受了治疗。诊断是在他五个月大的时候做出的。在患者的生长过程中出现生长衰竭和发育迟缓。我们发现KS2携带杂合基因突变KDM6A,与新生儿严重低血糖有关。主要是由于先天性高胰岛素血症,有待进一步研究。我们旨在提高对与新生儿低血糖相关的各种遗传疾病的认识,早期识别和治疗,以预防神经发育并发症,改善预后。
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Case report: Kabuki syndrome and persistent hypoglycemia in neonates.

The Kabuki syndrome (KS) is a rare congenital disease that has two different types, KS1 and KS2, with variant in epigenetic gene KMT2D and KDM6A, respectively. It is associated with multiple abnormalities such as (developmental delay, atypical facial features, cardiac anomalies, minor skeleton anomalies, genitourinary anomalies, and mild to moderate intellectual disability). This syndrome can lead to neonatal hypoglycemia that results from hyperinsulinemia and electrolyte abnormalities. We described 18-month-old Egyptian girl with Kabuki syndrome type 2 presented with persistent hypoglycemia since birth and accompanied by an abnormality in urine concentration, and blood gases showed academia. She was treated medically. The diagnosis was made at the age of five months. Growth failure and developmental delay were noted during the patient's growth. We found that KS2 with heterozygous gene mutation KDM6A, which had related with severe hypoglycemia in neonates. Mostly due to congenital hyperinsulinism for further investigations. We aim to increase the awareness of different genetic diseases that are related to neonatal hypoglycemia and early recognition and treatment to prevent the neurodevelopmental complication and improve the outcome.

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