Expanding phenotype of XNP mutations: mild to moderate mental retardation

Am J Med Genet. 2002 Jul 1;110(3):243-7. doi: 10.1002/ajmg.10446.

Abstract

Mutations in the XNP gene have been reported in alpha thalassemia/mental retardation (MR) syndrome (ATR-X) and other severe X-linked MR conditions with facial dysmorphisms. In this report, we describe a missense mutation in exon 18 in a family with borderline to moderate MR. Like other disorders associated with an XNP mutation, skewed X-inactivation was found in all carrier females in this family. Only retrospective examination revealed childhood facial hypotonia and HbH inclusions in some of the affected males. These results expand the spectrum of clinical phenotypes known to be due to mutations in the XNP gene, and indicate that XNP mutation analysis should not be restricted to patients with severe MR and characteristic facial features.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • DNA / chemistry
  • DNA / genetics
  • DNA Helicases*
  • DNA Mutational Analysis
  • Family Health
  • Female
  • Humans
  • Intellectual Disability / genetics*
  • Intellectual Disability / pathology
  • Male
  • Middle Aged
  • Mutation
  • Mutation, Missense
  • Nuclear Proteins / genetics*
  • Pedigree
  • Phenotype
  • X-linked Nuclear Protein

Substances

  • Nuclear Proteins
  • DNA
  • DNA Helicases
  • ATRX protein, human
  • X-linked Nuclear Protein

Associated data

  • OMIM/301040
  • OMIM/309580
  • OMIM/309590