JAGN1 Deficient Severe Congenital Neutropenia: Two Cases from the Same Family

J Clin Immunol. 2015 May;35(4):339-43. doi: 10.1007/s10875-015-0156-2. Epub 2015 Apr 8.

Abstract

Recently autosomal recessively inherited mutations in the gene encoding Jagunal homolog 1 (JAGN1) was described as a novel disease-causing gene of severe congenital neutropenia (SCN) JAGN1-mutant neutrophils were characterized by abnormality in endoplasmic reticulum structure, absence of granules, abnormal N-glycosylation of proteins and susceptibility to apoptosis. These findings imply the role of JAGN1 in neutrophil survival. Here, we report two siblings with a homozygous mutation in JAGN1 gene, exhibiting multisystemic involvement.

Publication types

  • Case Reports

MeSH terms

  • Child, Preschool
  • Congenital Bone Marrow Failure Syndromes
  • DNA Mutational Analysis
  • Exons
  • Female
  • Homozygote
  • Humans
  • Infant
  • Male
  • Membrane Proteins / deficiency
  • Membrane Proteins / genetics*
  • Mutation*
  • Mutation, Missense
  • Neutropenia / congenital*
  • Neutropenia / diagnosis
  • Neutropenia / genetics
  • Pedigree
  • Phenotype
  • Siblings

Substances

  • JAGN1 protein, human
  • Membrane Proteins

Supplementary concepts

  • Neutropenia, Severe Congenital, Autosomal Recessive 3