Effect of mutations in XPD(ERCC2) on pregnancy and prenatal development in mothers of patients with trichothiodystrophy or xeroderma pigmentosum

Eur J Hum Genet. 2012 Dec;20(12):1308-10. doi: 10.1038/ejhg.2012.90. Epub 2012 May 23.

Abstract

The XPD(ERCC2) gene encodes a DNA helicase involved in DNA repair and transcription. Patients with mutations in XPD may have different autosomal recessive phenotypes including trichothiodystrophy (TTD) or xeroderma pigmentosum (XP). TTD patients have sulfur-deficient, brittle hair, short stature and developmental delay. In contrast, XP patients have freckle-like pigmentation and a greatly increased risk of sun-induced skin cancers. Mothers of TTD patients have been reported to have a high frequency of pregnancy and neonatal complications. We performed a molecular epidemiological study of 15 mothers of 17 TTD patients and 13 mothers of 17 XP patients, all with XPD mutations. We found that 94% (16/17) of the TTD pregnancies had pre-term delivery, pre-eclampsia, hemolysis, elevated liver enzymes and low platelets (HELLP) syndrome, prematurity or low birth weight. None of the 17 XP pregnancies had these complications (P<0.001). As mutations in XPD may have differential effects on DNA repair and transcription, these observations should provide insights into the role of XPD in human pregnancy and fetal development.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Female
  • Fetal Development / genetics*
  • Fetal Diseases / diagnosis
  • Fetal Diseases / genetics
  • HELLP Syndrome / diagnosis
  • HELLP Syndrome / genetics
  • Humans
  • Infant, Low Birth Weight
  • Infant, Newborn
  • Male
  • Mutation*
  • Pre-Eclampsia / diagnosis
  • Pre-Eclampsia / genetics
  • Pregnancy
  • Premature Birth
  • Trichothiodystrophy Syndromes / complications
  • Trichothiodystrophy Syndromes / genetics*
  • Xeroderma Pigmentosum / diagnosis
  • Xeroderma Pigmentosum / genetics*
  • Xeroderma Pigmentosum Group D Protein / genetics*

Substances

  • Xeroderma Pigmentosum Group D Protein
  • ERCC2 protein, human