HLA DRB1*DQB1* haplotype in HTLV-I-associated familial infective dermatitis may predict development of HTLV-I-associated myelopathy/tropical spastic paraparesis

Am J Med Genet. 1996 Jan 2;61(1):37-41. doi: 10.1002/(SICI)1096-8628(19960102)61:1<37::AID-AJMG7>3.0.CO;2-U.

Abstract

A possible causal association between infective dermatitis and HTLV-I infection was reported in 1990 and confirmed in 1992. We now report familial infective dermatitis (ID) occurring in a 26-year-old mother and her 9-year-old son. The mother was first diagnosed with ID in 1969 at the age of 2 years in the Dermatology Unit at the University Hospital of the West Indies (U.H.W.I.) in Jamaica. The elder of her 2 sons was diagnosed with ID at the age of 3 years, also at U.H.W.I. Both mother and son are HTLV-I-seropositive. A second, younger son, currently age 2 years, is also HTLV-I-seropositive, but without clinical evidence of ID. Major histocompatibility complex (MHC), class II, human leucocyte antigen (HLA) genotyping documented a shared class II haplotype, DRB1*DQB1* (1101-0301), in the mother and her 2 sons. This same haplotype has been described among Japanese patients with HTLV-I-associated myelopathy/tropical spastic paraparesis (HAM/TSP), and has been associated with a possible pathologically heightened immune response to HTLV-I infection. The presence of this haplotype in these familial ID cases with clinical signs of HAM/TSP may have contributed to their risk for development of HAM/TSP. The unaffected, HTLV-I-seropositive younger son requires close clinical follow-up.

Publication types

  • Case Reports
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Adult
  • Child
  • Child, Preschool
  • Dermatitis / etiology*
  • Dermatitis / genetics
  • Dermatitis / immunology
  • Female
  • Genotype
  • HLA-DQ Antigens / genetics*
  • HLA-DQ beta-Chains
  • HLA-DR Antigens / genetics*
  • HLA-DRB1 Chains
  • HTLV-I Infections / complications
  • HTLV-I Infections / genetics
  • HTLV-I Infections / immunology*
  • Haplotypes
  • Histocompatibility Testing
  • Humans
  • Jamaica
  • Male
  • Paraparesis, Tropical Spastic / epidemiology
  • Paraparesis, Tropical Spastic / immunology*
  • Pedigree
  • Predictive Value of Tests
  • Skin Diseases, Infectious / etiology*
  • Skin Diseases, Infectious / genetics
  • Skin Diseases, Infectious / immunology

Substances

  • HLA-DQ Antigens
  • HLA-DQ beta-Chains
  • HLA-DQB1 antigen
  • HLA-DR Antigens
  • HLA-DRB1 Chains