ICF syndrome: high variability of the chromosomal phenotype and association with classical Hodgkin lymphoma

Am J Med Genet A. 2007 Sep 1;143A(17):2052-7. doi: 10.1002/ajmg.a.31885.

Abstract

We report on two sibs with ICF syndrome (immunodeficiency, centromeric heterochromatin instability, and facial anomalies) diagnosed in the elder brother based on the typical chromosomal abnormalities present in 56% of metaphases from cultured lymphocytes. In a previous cytogenetic analysis this diagnosis had been missed due to low manifestation of the ICF chromosomal phenotype. Hypomethylation of classical satellites 2 and 3, and of alpha-satellite DNA was shown in the lymphocytes of the younger sister. At 7 years of age the boy presented with hemiplegia due to tumerous invasion of the right brachial plexus. Histopathology revealed classical Hodgkin lymphoma, a neoplasia which might have been facilitated by the underlying genetic defect.

Publication types

  • Case Reports

MeSH terms

  • Centromere / genetics
  • Child
  • Child, Preschool
  • Chromosomal Instability / genetics*
  • Facial Bones / abnormalities*
  • Female
  • Genetic Variation*
  • Heterochromatin / genetics
  • Hodgkin Disease / diagnosis
  • Hodgkin Disease / genetics*
  • Humans
  • Immunologic Deficiency Syndromes / diagnosis
  • Immunologic Deficiency Syndromes / genetics*
  • Male
  • Phenotype*

Substances

  • Heterochromatin