Health screening behaviors among adults with hereditary hemorrhagic telangiectasia in North America

Genet Med. 2017 Jun;19(6):659-666. doi: 10.1038/gim.2016.161. Epub 2016 Oct 13.

Abstract

Purpose: This study aimed to identify factors that influence screening behaviors of adults with hereditary hemorrhagic telangiectasia (HHT).

Methods: Participants with a self-reported diagnosis of HHT were recruited from the HHT Foundation International, Inc.; the "HHT Awareness" Facebook group; and six HHT clinics. A cross-sectional mixed methods survey was administered to investigate the relationships among the Health Belief model constructs, the domains of illness representations, and HHT-specific screening behaviors consistent with recommended guidelines.

Results: A total of 320 participants reported rates of cerebral arteriovenous malformation (AVM) screenings, pulmonary AVM screenings, and HHT annual checkups that were 82.0, 67.1, and 56.5%, respectively. Logistical regression analysis showed that perceived barriers (β = -0.114, P < 0.001), perceived susceptibility (β = 0.117, P < 0.05), treatment control (β = 0.078, P < 0.05), and emotional representations (β = 0.067, P < 0.05) were significant predictors of HHT screening. Open-ended responses revealed perceived barriers to screening, including a lack of health-care providers (HCPs) familiar with and/or knowledgeable about HHT.

Conclusion: Our results reveal suboptimal screening rates among adults with HHT and identify several factors influencing these behaviors. We suggest that there is a need for increased provider education regarding HHT as well as approaches that providers can use to improve screening adherence.Genet Med advance online publication 13 October 2016.

Publication types

  • Research Support, N.I.H., Intramural

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Cross-Sectional Studies
  • Female
  • Health Knowledge, Attitudes, Practice
  • Humans
  • Male
  • Mass Screening*
  • Middle Aged
  • Patient Acceptance of Health Care*
  • Patient Compliance
  • Telangiectasia, Hereditary Hemorrhagic / diagnosis*
  • Telangiectasia, Hereditary Hemorrhagic / psychology
  • Young Adult