Clinical and GAA gene mutation analysis in mainland Chinese patients with late-onset Pompe disease: identifying c.2238G > C as the most common mutation

BMC Med Genet. 2014 Dec 20:15:141. doi: 10.1186/s12881-014-0141-2.

Abstract

Background: Pompe disease is an autosomal recessive lysosomal glycogen storage disorder that has been reported in different ethnic populations which carry different common mutations of the acid alpha-glucosidase (GAA) gene. The GAA mutation pattern in mainland Chinese patients with late-onset Pompe disease is still not well understood.

Methods: We presented the clinical and genetic characteristics of 27 mainland Chinese late-onset Pompe patients from 24 families.

Results: GAA mutation analysis revealed 26 different mutations, including 10 that were novel. The allelic frequency of c.2238G > C (p.W746C) was found to be 27.08% in this patient group. Respiratory dysfunction was diagnosed in 10 of 11 patients who underwent pulmonary function evaluation, although only four required ventilator support at night.

Conclusions: Our findings indicate that c.2238G > C (p.W746C) is the most common mutation in mainland Chinese late-onset Pompe patients, as observed in Taiwanese patients. The novel mutations identified in this study expand the genetic spectrum of late-onset Pompe disease, and the prevalence of respiratory dysfunction highlights the importance of monitoring pulmonary function in late-onset Pompe patients.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Age of Onset
  • Asian People / genetics*
  • Child
  • Child, Preschool
  • China
  • Genetic Association Studies
  • Genetic Predisposition to Disease
  • Glycogen Storage Disease Type II / genetics*
  • Glycogen Storage Disease Type II / physiopathology
  • Humans
  • Infant
  • Male
  • Pedigree
  • Point Mutation*
  • Respiratory Function Tests
  • Young Adult
  • alpha-Glucosidases / genetics*

Substances

  • GAA protein, human
  • alpha-Glucosidases