Familial thoracic aortic aneurysms and dissections: identification of a novel locus for stable aneurysms with a low risk for progression to aortic dissection

Circ Cardiovasc Genet. 2011 Feb;4(1):36-42. doi: 10.1161/CIRCGENETICS.110.958066. Epub 2010 Dec 16.

Abstract

Background: Thoracic aortic aneurysms leading to acute aortic dissections are the major diseases that affect the thoracic aorta. Approximately 20% of patients with thoracic aortic aneurysms and dissections (TAAD) have a family history of TAAD, and these patients present younger with more rapidly enlarging aneurysms than patients without a family history of aortic disease.

Methods and results: A large family with multiple members with TAAD inherited in an autosomal-dominant manner was identified. The ascending aortic aneurysms were associated with slow enlargement, a low risk of dissection, and decreased penetrance in women. Genome-wide linkage analysis was performed, and a novel locus on chromosome 12 was identified for the mutant gene causing disease in this family. Of the 12 male members who carry the disease-linked microsatellite haplotype, 9 had ascending aortic aneurysms with an average diameter of 4.7 cm at an average age of 52.4 years (range, 32 to 76 years) at the time of diagnosis; only 1 individual had progressed to acute aortic dissection, and no other members with aortic dissections were identified. Women harboring the disease-linked haplotype did not have thoracic aortic disease, including 1 aged 84 years. Sequencing of 9 genes within the critical interval at the chromosome 12 locus did not identify the mutant gene.

Conclusions: Mapping a locus for ascending thoracic aortic aneurysms associated with a low risk of aortic dissection supports our hypothesis that genes leading to familial disease can be associated with less-aggressive thoracic aortic disease.

Publication types

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

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Aorta / pathology*
  • Aortic Aneurysm, Thoracic / complications*
  • Aortic Aneurysm, Thoracic / genetics*
  • Aortic Dissection / complications*
  • Aortic Dissection / genetics*
  • Chromosomes, Human, Pair 12 / genetics
  • Disease Progression*
  • Family
  • Female
  • Genetic Association Studies
  • Genetic Loci / genetics*
  • Genetic Predisposition to Disease
  • Genome, Human / genetics
  • Haplotypes / genetics
  • Humans
  • Lod Score
  • Male
  • Middle Aged
  • Pedigree
  • Risk Factors
  • Sequence Analysis, DNA