Follow-up of pulmonary right-to-left shunt in hereditary haemorrhagic telangiectasia

Eur Respir J. 2016 Jun;47(6):1750-7. doi: 10.1183/13993003.01588-2015. Epub 2016 Mar 10.

Abstract

Pulmonary arteriovenous malformations (PAVMs) are associated with severe neurological complications in hereditary haemorrhagic telangiectasia (HHT). Transthoracic contrast echocardiography (TTCE) is recommended for screening of pulmonary right-to-left shunts (RLS). Although growth of PAVMs is shown in two small studies, no studies on follow-up with TTCE exist.All HHT patients underwent a second TTCE 5 years after initial screening. Patients with a history of PAVM embolisation were excluded. Pulmonary RLS grade on TTCE after 5 years was compared to the grade at screening.200 patients (53.5% female, mean±sd age at screening 44.7±14.1 years) were included. Increase in RLS grade occurred in 36 (18%) patients, of whom six (17%) underwent embolisation. The change in grade between screening and follow-up was not more than one grade. Of patients with nontreatable pulmonary RLS at screening (n=113), 14 (12.4%) underwent embolisation. In patients without pulmonary RLS at initial screening (n=87), no treatable PAVMs developed during follow-up.Within 5 years, no treatable PAVMs developed in HHT patients without pulmonary RLS at initial screening. Increase in pulmonary RLS grade occurred in 18% of patients, and never increased by more than one grade. Of patients with nontreatable pulmonary RLS at initial screening, 12% underwent embolisation.

MeSH terms

  • Adult
  • Arteriovenous Fistula / diagnostic imaging*
  • Arteriovenous Fistula / physiopathology*
  • Arteriovenous Malformations
  • Contrast Media / chemistry
  • Echocardiography
  • Embolization, Therapeutic
  • Female
  • Follow-Up Studies
  • Humans
  • Lung / physiopathology*
  • Male
  • Middle Aged
  • Predictive Value of Tests
  • Pulmonary Artery / abnormalities*
  • Pulmonary Artery / diagnostic imaging
  • Pulmonary Artery / physiopathology
  • Pulmonary Veins / abnormalities*
  • Pulmonary Veins / diagnostic imaging
  • Pulmonary Veins / physiopathology
  • Telangiectasia, Hereditary Hemorrhagic / complications
  • Telangiectasia, Hereditary Hemorrhagic / diagnostic imaging*
  • Telangiectasia, Hereditary Hemorrhagic / physiopathology*
  • Tomography, X-Ray Computed
  • Treatment Outcome

Substances

  • Contrast Media

Supplementary concepts

  • Pulmonary Arteriovenous Fistulas