Literature DB >> 8884087

Homozygous deletion of exon 18 leads to degradation of the lysosomal alpha-glucosidase precursor and to the infantile form of glycogen storage disease type II.

M G Ausems1, M A Kroos, M Van der Kraan, J A Smeitink, W J Kleijer, H K Ploos van Amstel, A J Reuser.   

Abstract

We describe two unrelated Dutch patients with typical symptoms of infantile glycogen storage disease type II (GSD II) and virtual absence of acid alpha-glucosidase activity in leukocytes and cultured skin fibroblasts. The patients were identified as homozygotes for a deletion of exon 18 of the acid alpha-glucosidase gene (GAA). The in-frame deletion manifests at the protein level in a characteristic way: the enzyme precursor is smaller than normal and degraded in the endoplasmic reticulum or Golgi complex. These case present an evident example of a genotype-phenotype correlation in glycogen storage disease type II.

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Year:  1996        PMID: 8884087     DOI: 10.1111/j.1399-0004.1996.tb03801.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  2 in total

1.  Predicting cross-reactive immunological material (CRIM) status in Pompe disease using GAA mutations: lessons learned from 10 years of clinical laboratory testing experience.

Authors:  Deeksha S Bali; Jennifer L Goldstein; Suhrad Banugaria; Jian Dai; Joanne Mackey; Catherine Rehder; Priya S Kishnani
Journal:  Am J Med Genet C Semin Med Genet       Date:  2012-01-17       Impact factor: 3.908

2.  Pompe disease results in a Golgi-based glycosylation deficit in human induced pluripotent stem cell-derived cardiomyocytes.

Authors:  Kunil K Raval; Ran Tao; Brent E White; Willem J De Lange; Chad H Koonce; Junying Yu; Priya S Kishnani; James A Thomson; Deane F Mosher; John C Ralphe; Timothy J Kamp
Journal:  J Biol Chem       Date:  2014-12-08       Impact factor: 5.157

  2 in total

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