Novel SLC37A4 Mutations in Korean Patients With Glycogen Storage Disease Ib

Ann Lab Med. 2017 May;37(3):261-266. doi: 10.3343/alm.2017.37.3.261.

Abstract

Background: Molecular techniques are fundamental for establishing an accurate diagnosis and therapeutic approach of glycogen storage diseases (GSDs). We aimed to evaluate SLC37A4 mutation spectrum in Korean GSD Ib patients.

Methods: Nine Korean patients from eight unrelated families with GSD Ib were included. SLC37A4 mutations were detected in all patients with direct sequencing using a PCR method and/or whole-exome sequencing. A comprehensive review of previously reported SLC37A4 mutations was also conducted.

Results: Nine different pathogenic SLC37A4 mutations were identified in the nine patients with GSD Ib. Among them, four novel mutations were identified: c.148G>A (pGly50Arg), c.320G>A (p.Trp107*), c.412T>C (p.Trp138Arg), and c.818G>A (p.Gly273Asp). The most common mutation type was missense mutations (66.7%, 6/9), followed by nonsense mutations (22.2%, 2/9) and small deletion mutations (11.1%, 1/9). The most common mutation identified in the Korean population was c.443C>T (p.Ala148Val), which comprised 39.9% (7/18) of all tested alleles. This mutation has not been reported in GSD Ib patients in other ethnic populations.

Conclusions: This study expands knowledge of the SLC37A4 mutation spectrum in Korean patients with GSD Ib.

Keywords: GSD Ib; Glycogen storage disease; Korean population; SLC37A4; mutation.

Publication types

  • Case Reports

MeSH terms

  • Alleles
  • Antiporters / genetics*
  • Asian People / genetics*
  • Child
  • Exons
  • Female
  • Genotype
  • Glycogen Storage Disease Type I / diagnosis
  • Glycogen Storage Disease Type I / genetics*
  • Humans
  • Infant
  • Male
  • Monosaccharide Transport Proteins / genetics*
  • Mutation, Missense
  • Polymorphism, Genetic
  • Republic of Korea
  • Sequence Deletion

Substances

  • Antiporters
  • Monosaccharide Transport Proteins
  • SLC37A4 protein, human

Supplementary concepts

  • Glycogen Storage Disease IB