Clinical and molecular characteristics of patients with Gaucher disease in Southern China

Blood Cells Mol Dis. 2018 Feb:68:30-34. doi: 10.1016/j.bcmd.2016.10.026. Epub 2016 Nov 3.

Abstract

Gaucher disease (GD) is a common lysosomal storage disorder caused by the deficiency of acid β-glucosidase, due to mutations in the GBA gene. To explore the clinical and molecular characteristics of GD patients from Southern China, GBA gene were analyzed by nest PCR and direct Sanger-sequencing. Novel missense mutations were transiently transfected in COS-7 cells by plasmid system for functional verification. Among the 22 GD patients, 19 patients were classified as type 1 and three as type 2. Over 60% of the type 1 patient had the onset before two years of age and about 42% of them died before three years of age. Six type 1 patients with L444P homozygous genotype, presented with early onset and severe hepatosplenomegaly. Four novel mutations Y22C, F109L, L149F and c.983_990delCCCACTGG were identified. The GBA activities in vitro of novel mutants Y22C, F109L and L149F were 20.2%, 6.9% and 6.5% of the wild-type, respectively. L444P mutation accounted for 47.7% of the mutant alleles. Our results revealed that type 1 GD tends to present with a severe phenotype among southern Chinese. L444P was the most prevalent mutation and L444P homozygous genotype was associated with severe type 1 GD. Three novel missense mutations identified were pathogenic.

Keywords: Expression study; GBA gene; Gaucher disease; Molecular analysis.

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Animals
  • Asian People / genetics
  • COS Cells
  • Child
  • Child, Preschool
  • China / epidemiology
  • Chlorocebus aethiops
  • Female
  • Gaucher Disease / epidemiology
  • Gaucher Disease / genetics*
  • Genotype
  • Glucosylceramidase / chemistry
  • Glucosylceramidase / genetics*
  • Homozygote
  • Humans
  • Infant
  • Male
  • Middle Aged
  • Models, Molecular
  • Mutation*
  • Mutation, Missense
  • Point Mutation
  • Protein Conformation
  • Young Adult

Substances

  • Glucosylceramidase