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F12 Research participation: the view of persons at risk and persons with premanifest huntington’s disease
  1. Filipa Júlio1,2,
  2. Ruth Blanco1,3,
  3. Josè Perez Casanova1,3,
  4. Barbara D’Alessio1,4,
  5. Beatrice De Schepper1,5,
  6. Dina De Sousa1,6,
  7. Paul De Sousa1,6,
  8. Cristina Ferreira1,2,
  9. Hans Gommans1,7,
  10. Rob Haselberg1,7,
  11. Emilie Hermant1,8,
  12. Danuta Lis1,9,
  13. Sabrina Maffi10,
  14. Svein Olaf Olsen1,11,
  15. Marios Papantoniou1,12,
  16. Ferdinando Squitieri10,
  17. Marina Tretyakova1,13,
  18. Zaynab Umakhanova1,13,
  19. Vladimír Václavík1,14,
  20. Michaela Winkelmann1,15,
  21. Astri Arnesen1,11,
  22. European Huntington Association
  1. 1European Huntington Association, Moerbeke (Waas), Belgium
  2. 2Associação Portuguesa dos Doentes de Huntington, Lisboa, Portugal
  3. 3Asociación Corea de Huntington Española, Madrid, Spain
  4. 4Fondazione Lega Italiana Ricerca Huntington, Rome, Italy
  5. 5Huntington Liga, Moerbeke (Waas), Belgium
  6. 6Scottish Huntington’s Association, Paisley, UK
  7. 7Vereniging Van Huntington, Amersfoort, the Netherlands
  8. 8L’Association Huntington France, Paris, France
  9. 9Polskie Stowarzyszenie Choroby Huntingtona, Warsaw, Poland
  10. 10Huntington and Rare diseases Unit, IRCCS Casa Sollievo della Sofferenza Research Hospital, San Giovanni Rotondo, Italy
  11. 11Landsforeningen for Huntingtons sykdom, LHS v/Geir Viksund, Erdal, Norway
  12. 12Huntington’s Disease Association of Cyprus, Limassol, Cyprus
  13. 13Orphan People, Russia
  14. 14Spoločnosť Pre Pomoc Pri Huntingtonovej Chorobe, Gajary, Slovakia
  15. 15Deutsche Huntington Hilfe E.V., Duisburg, Germany


Background The development of effective therapies for Huntington’s disease (HD) requires an active, informed, and lasting commitment from HD families to research. Specifically, those traditionally less involved in studies should be heard and engaged in this process: persons at risk for HD (HDRisk) and persons with premanifest HD (PreHD).

Aim To determine which factors affect the willingness of persons with HDrisk and PreHD to participate in research and check for differences related to HD status.

Methods The European Huntington Association (EHA) created an online survey to assess the perceptions and experiences about research participation among persons with HDRisk and PreHD. The two groups were compared on their answers to questions about research experience and knowledge, sources of information about research, the importance of reasons for getting involved and not getting involved in studies, and factors preventing and facilitating study participation.

Results/Outcome The survey was filled by 263 persons with HDRisk and 262 persons with PreHD. While the overall motivation of both groups to engage in research was high, respondents with PreHD reported significantly more research experience and better research knowledge than respondents with HDRisk. Respondents with HDRisk identified more barriers to research participation and the need for more support to engage in studies compared to respondents with PreHD.

Conclusions Motivation to take part in studies is high, despite limited research experience and literacy. This motivation is strongly influenced by HD status. Our findings highlight the relevance of planning tailored interventions to better support an informed participation of specific HD groups in research.

  • clinical research
  • clinical trials
  • premanifest HD
  • survey

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