Girl with combined cellular immunodeficiency, pancytopenia, malformations, deletion 11q23.3 --> qter, and trisomy 8q24.3 --> qter

Am J Med Genet. 2002 Apr 1;108(4):322-6. doi: 10.1002/ajmg.10284.

Abstract

We describe here a 3-year-old girl demonstrating combined cellular immunodeficiency of B- and T-cells, pancytopenia, multiple anomalies, and severe mental retardation. Cytogenetic analysis and fluorescent in situ hybridization (FISH) indicated an unbalanced translocation of chromosomes 8q and 11q, resulting in monosomy 11q23.3-qter and trisomy 8q24.3-qter. The association of cellular immunodeficiency and partial deletion 11q and/or partial trisomy 8q has not been described previously; however, the 11q deletion has been reported with humoral immunodeficiency or pancytopenia. Some one-third to one-half of patients with partial monosomy 11q were reported to have pancytopenia, which has been related to the absence of the 11q23-q24 region. Our case narrows down the critical interval for thrombo- or pancytopenia to 11q23.3-q24 and excludes both the ATM (which resides on 11q23.1) and the MLL genes as possible candidate genes. We are proposing that haploinsufficiency of the NFRKB gene on 11q24-q25 and/or the ETS-1 proto-oncogene on 11q24 may have caused or contributed to the immunodeficiency (decreased levels of B- and T-lymphocytes) in our patient.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple / genetics
  • Abnormalities, Multiple / pathology
  • Child, Preschool
  • Chromosome Deletion*
  • Chromosomes, Human, Pair 11 / genetics*
  • Chromosomes, Human, Pair 8 / genetics*
  • Female
  • Humans
  • In Situ Hybridization, Fluorescence
  • Microsatellite Repeats
  • Pancytopenia / genetics*
  • Pancytopenia / pathology
  • Proto-Oncogene Mas
  • Severe Combined Immunodeficiency / genetics*
  • Severe Combined Immunodeficiency / pathology
  • Trisomy*