Multiple coronary artery aneurysms in a child with neurofibromatosis type 1

Eur J Pediatr. 2000 Jul;159(7):477-80. doi: 10.1007/s004310051313.

Abstract

A number of frequently unrecognised vascular manifestations have been described in patients with neurofibromatosis type 1 (NF1), including involvement of the great vessels, cerebral, visceral and renal arteries. Rarely, changes in the coronary arteries have been reported in adults with NF1. We report on a 16-year-old boy affected by NF1 with dysmorphic features and three aneurysms in the mid-portion of the left descending coronary artery disclosed by chance during investigation for a malignant peripheral nerve sheath tumour. Molecular analysis detected a gross de novo deletion in the NF1 gene. The boy had had no previous cardiac symptoms but died suddenly after developing signs and symptoms suggestive of myocardial infarction.

Conclusion: To the best of our knowledge, this represents the first report of multiple lesions in the coronary arteries in a child affected by neurofibromatosis type 1 with a known deletion of the neurofibromatosis type 1 gene.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple / genetics
  • Adolescent
  • Adult
  • Chromosome Deletion
  • Coronary Aneurysm / genetics*
  • Death, Sudden, Cardiac / etiology
  • Genes, Neurofibromatosis 1 / genetics*
  • Humans
  • Male
  • Neurofibromatosis 1 / genetics*
  • Risk Factors