Novel and recurrent STAT3 mutations in hyper-IgE syndrome patients from different ethnic groups

Mol Immunol. 2008 Nov;46(1):202-6. doi: 10.1016/j.molimm.2008.07.001. Epub 2008 Aug 15.

Abstract

We performed clinical, immunological and genetic studies of 12 hyper-IgE syndrome (HIES) patients from 4 Hungarian, 2 Lebanese, one Russian, one Polish, and one Swedish families with autosomal dominant (AD) or sporadic forms of the disease to reveal cross-ethnicity of recurrent and novel mutations in the signal transducer and activator of transcription-3 gene (STAT3). Four patients from 3 Hungarian families, and one Russian, and one Swedish patient carried the heterozygous R382W germline mutation at the DNA-binding site of STAT3. The recurrent V637M mutation affecting the SRC homology 2 (SH2) domain was detected in one Lebanese and one Polish family, and the V463del deletion located in the DNA-binding domain was unveiled in another Lebanese family. A novel H332Y mutation affecting the DNA-binding site of STAT3 in three Hungarian patients from a Gypsy family was also found. The segregation of this mutation with HIES, restriction fragment length polymorphism analysis of STAT3 from patients and controls and the negligible production upon IL-6 stimulation of monocyte chemotactic protein-1 by the patient's blood mononuclear cells suggested that the H332Y mutation was disease-causing. These data suggest, that dominant negative mutations of the DNA-binding and SH2 domains of STAT3 cause AD and sporadic cases of HIES in different ethnic groups with R382W as the predominant mutation found in 5 of the 9 families. Functional and genetic data support that the novel H332Y mutation may result in the loss of function of STAT3 and leads to the HIES phenotype.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Child
  • Child, Preschool
  • DNA / metabolism
  • Demography
  • Ethnicity / genetics*
  • Female
  • Humans
  • Job Syndrome / genetics*
  • Job Syndrome / immunology*
  • Male
  • Mutation / genetics*
  • Protein Binding
  • Protein Structure, Tertiary
  • Restriction Mapping
  • STAT3 Transcription Factor / chemistry
  • STAT3 Transcription Factor / genetics*

Substances

  • STAT3 Transcription Factor
  • STAT3 protein, human
  • DNA