Ascending aortic remodelling in Fabry disease after long-term enzyme replacement therapy

Swiss Med Wkly. 2017 Nov 9:147:w14517. doi: 10.4414/smw.2017.14517. eCollection 2017.

Abstract

Background: Previous cross-sectional studies reported a high prevalence of ascending aorta dilations/aneurysms in male adults with Fabry disease, independently of cardiovascular risk factors.

Aims of the study: To characterise the remodelling of the ascending aorta in classic Fabry disease under long-term enzyme replacement therapy.

Methods: Diameter of the ascending aorta was measured with magnetic resonance imaging at the sino-tubular junction (STJ), and proximal (pAsAo), and distal ascending aorta (dAsAo) at baseline, and after 5 and 10 years of enzyme replacement therapy in 15 adult Fabry patients (10 males; 5 females).

Results: Over a mean follow-up of 9.5 years, the annual expansion rates measured in 10 males with Fabry disease were 0.41 ± 0.16, 0.36 ± 0.25 and 0.41 ± 0.26 mm/y at the STJ, pAsAo and dAsAo, respectively. Expansion rate at the pAsAo level in male patients was significantly higher than the expected expansion projected from theoretical normal values: 0.36 ± 0.25 vs 0.13 ± 0.05, p = 0.017. In 5 females, the annual expansion rates at the STJ, pAsAo and dAsAo were 0.14 ± 0.11, 0.21 ± 0.18 and 0.26 ± 0.24 mm/y, respectively. There was no significant difference from the projected normal expansion rate at the level of the pAsAo: 0.21 ± 0.18 vs 0.13 ± 0.04, p = 0.39.

Conclusion: Our data suggest that the remodelling of the ascending aorta is more pronounced in male patients with Fabry disease under long-term enzyme replacement therapy compared with the progression observed in a large population study.

MeSH terms

  • Adult
  • Aorta* / diagnostic imaging
  • Aorta* / pathology
  • Aortic Aneurysm / pathology
  • Dilatation
  • Disease Progression
  • Enzyme Replacement Therapy / methods*
  • Fabry Disease / complications*
  • Fabry Disease / genetics
  • Female
  • Humans
  • Longitudinal Studies
  • Male
  • Prospective Studies
  • Risk Factors
  • Sex Factors