Literature DB >> 23014386

Neonatal presentation of lethal neuromuscular glycogen storage disease type IV.

L F Escobar1, S Wagner, M Tucker, J Wareham.   

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.

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Year:  2012        PMID: 23014386     DOI: 10.1038/jp.2011.178

Source DB:  PubMed          Journal:  J Perinatol        ISSN: 0743-8346            Impact factor:   2.521


  3 in total

1.  A Rare Cause of Elevated Chitotriosidase Activity: Glycogen Storage Disease Type IV.

Authors:  Hayriye Hizarcioglu-Gulsen; Aysel Yuce; Zuhal Akcoren; Burcu Berberoglu-Ates; Yusuf Aydemir; Erdal Sag; Serdar Ceylaner
Journal:  JIMD Rep       Date:  2014-08-26

2.  Novel method for detection of glycogen in cells.

Authors:  Alexander V Skurat; Dyann M Segvich; Anna A DePaoli-Roach; Peter J Roach
Journal:  Glycobiology       Date:  2017-05-01       Impact factor: 4.313

3.  Prospective cohort study for identification of underlying genetic causes in neonatal encephalopathy using whole-exome sequencing.

Authors:  Theodora U J Bruun; Caro-Lyne DesRoches; Diane Wilson; Vann Chau; Tadashi Nakagawa; Masahiro Yamasaki; Shinya Hasegawa; Toshiyuki Fukao; Christian Marshall; Saadet Mercimek-Andrews
Journal:  Genet Med       Date:  2017-08-17       Impact factor: 8.822

  3 in total

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