Unique spectrum of MEFV mutations in Iranian Jewish FMF patients--clinical and demographic significance

Rheumatology (Oxford). 2007 Nov;46(11):1718-22. doi: 10.1093/rheumatology/kem228. Epub 2007 Oct 15.

Abstract

Objectives: To determine the spectrum of mutations in the Mediterranean fever gene (MEFV) of Iranian Jews with familial Mediterranean fever (FMF) and to analyse their clinical manifestations.

Methods: FMF patients with both parents of Iranian-Jewish (IJ) extraction or with one IJ parent (IJ-other, 10 of each) were characterized for clinical manifestations, and the B30.2 (PRYSPRY) domain of their MEFV was sequenced for mutations.

Results: Only one rare mutation, R653H, and one new mutation, G632S were present in the IJ group (in 2/10 patients), whereas the new, and common mutations were present in the IJ-other patients (8/10 patients). The new mutation was traced thrice to an IJ ancestor, and although carried asymptomatically by family members, it was over-represented in the patients (3/28 unrelated IJ alleles) compared non-affected IJ subjects (1/126 alleles, P = 0.03) or with non-Jewish Iranians (0/108 alleles, P = 0.001). The mutation was associated with a distinct phenotype regarding sites involved in the attack (P = 0.001), mild severity, sole expression of febrile episodes (P = 0.01) and a male bias (P = 0.01). In two 3D PRYSPRY models the G632S mutation was localized to a surface loop and close to a putative binding site.

Conclusions: Iranian Jews with FMF have a unique spectrum of mutations including a newly described mutation with a non-typical phenotype.

MeSH terms

  • Adolescent
  • Adult
  • Base Sequence
  • Child
  • Cytoskeletal Proteins / chemistry
  • Cytoskeletal Proteins / genetics*
  • Familial Mediterranean Fever / genetics*
  • Female
  • Haplotypes
  • Humans
  • Jews / genetics*
  • Male
  • Middle Aged
  • Mutation*
  • Pedigree
  • Protein Structure, Tertiary
  • Pyrin
  • Severity of Illness Index

Substances

  • Cytoskeletal Proteins
  • MEFV protein, human
  • Pyrin