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新生儿致命性神经肌肉糖原贮积病Ⅳ型。

Neonatal presentation of lethal neuromuscular glycogen storage disease type IV.

机构信息

Medical Genetics and Neurodevelopmental Pediatric Center-Peyton Manning Children Hospital, Indianapolis, IN 46260, USA.

出版信息

J Perinatol. 2012 Oct;32(10):810-3. doi: 10.1038/jp.2011.178.

Abstract

A total of 11 types of glycogen storage disorders have been recognized with variable clinical presentations. Type IV, also known as Andersen disease, represents a rare subtype that can induce severe clinical findings early in life. We report on a patient with early fetal onset of symptoms with severe neuromuscular findings at birth. The pregnancy was further complicated by polyhydramnios and depressed fetal movement. At birth severe hypotonia was noticed requiring active resuscitation and then mechanical ventilation. His lack of expected course for hypoxic ischemic encephalopathy prompted genetic testing, including a muscle biopsy, which confirmed the diagnosis of glycogen storage disease IV (GSD IV). Mutation analysis of the glycogen branching enzyme 1 gene demonstrated a previously unrecognized mutation. We review recent information on early presentation of GSD IV with particular interest in the presentation of the neonatal lethal neuromuscular form of this rare disorder.

摘要

总共有 11 种糖原贮积症,其临床表现各不相同。第四型糖原贮积症,又称安德森病,是一种罕见的亚型,可导致婴儿早期出现严重的临床表现。我们报告了一例患者,其在胎儿期即出现症状,出生时即有严重的神经肌肉表现。妊娠还伴有羊水过多和胎动减少。出生时,患儿明显出现严重的低张力,需要积极复苏,然后进行机械通气。患儿没有出现预期的缺氧缺血性脑病表现,促使我们进行了基因检测,包括肌肉活检,从而确诊为糖原贮积症四型(GSD IV)。糖原分支酶 1 基因突变分析显示了一种以前未被识别的突变。我们回顾了最近关于 GSD IV 早期表现的信息,特别关注这种罕见疾病的新生儿致死性神经肌肉型的表现。

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