Genetic deafness in a preterm infant with a critical postnatal course

Pediatr Crit Care Med. 2006 May;7(3):270-2. doi: 10.1097/01.PCC.0000216679.47571.DA.

Abstract

Objective: We present a case of deafness in a preterm infant with several predisposing factors of an acquired hearing impairment that, however, turned out to have a genetic cause. We describe the severe postnatal course and review the relevant literature.

Design: Case report.

Setting: University-based tertiary neonatal intensive care unit.

Patient: Preterm infant (gestational age, 26/37; wks).

Measurements and main results: A preterm infant exhibited hearing impairment after a complicated clinical course with pneumothoraces, a hemodynamically relevant patent ductus arteriosus, treatment with potentially ototoxic drugs, intraventricular hemorrhage, and periventricular leukomalacia. Despite the absence of a family history for deafness, genetic testing was performed. Surprisingly, genetic analysis revealed the presence of two compound heterozygous mutations in the patient's GJB2 gene as the cause for his early-onset nonsyndromic deafness.

Conclusion: To elucidate the nature of a hearing disorder, it is worthwhile to consider a genetic cause, despite the fact that it may seem unlikely in a severely sick preterm infant with numerous risk factors for a postnatally acquired hearing impairment and without a positive family history.

Publication types

  • Case Reports

MeSH terms

  • Connexin 26
  • Connexins / genetics*
  • DNA Mutational Analysis
  • Deafness / genetics*
  • Genetic Testing
  • Humans
  • Infant, Newborn
  • Infant, Premature
  • Male
  • Risk Factors

Substances

  • Connexins
  • GJB2 protein, human
  • Connexin 26