A novel deletion mutation in IL2RG gene results in X-linked severe combined immunodeficiency with an atypical phenotype

Immunogenetics. 2017 Jan;69(1):29-38. doi: 10.1007/s00251-016-0949-3. Epub 2016 Aug 26.

Abstract

Severe combined immunodeficiency (SCID) is the most serious disorder among primary immunodeficiency diseases threatening children's life. Atypical SCID variant, presenting with mild reduced T cells subsets, is often associated with infection susceptibility but poor clinical diagnosis. The atypical X-SCID patient in the present study showed a mild clinical presentation with a TlowNK+B+ immunophenotype. The patient has reduced T- cell subpopulations with a subdued thymic output measured by sjTRECs. Further analysis showed that T cells maintained a normal proliferation and a broad Vβ repertoire. NK cells, however, exhibited a skewed development toward immature CD3-CD16+CD56- cells. Genetic analysis revealed a novel deletion at nucleotide 52 in exon 1 of IL2RG gene. Sequence alignment predicted a truncated IL2RG protein missing signal peptide derived from a possible alternative reading frame. The novel mutation in IL2RG gene identified in our study may help the early diagnosis of atypical X-SCID.

Keywords: Atypical SCID; Frameshift; IL2RG gene; Primary immunodeficiency diseases; Signal peptide; X-linked SCID.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Amino Acid Sequence
  • Cell Differentiation
  • Cell Proliferation
  • Female
  • Flow Cytometry
  • Humans
  • Interleukin Receptor Common gamma Subunit / genetics*
  • Male
  • Pedigree
  • Phenotype
  • Polymerase Chain Reaction
  • Sequence Deletion / genetics*
  • Sequence Homology, Amino Acid
  • Severe Combined Immunodeficiency / genetics*
  • Severe Combined Immunodeficiency / pathology
  • T-Lymphocytes
  • X-Linked Combined Immunodeficiency Diseases / genetics*
  • X-Linked Combined Immunodeficiency Diseases / pathology

Substances

  • IL2RG protein, human
  • Interleukin Receptor Common gamma Subunit

Supplementary concepts

  • Severe combined immunodeficiency, atypical