Research Participation in Huntington’s Disease: Perceptions and Experiences of the Dutch Community

Filipa Júlio1,2, Ruth Blanco1,3, Josè Perez Casanova1,3, Barbara D'Alessio1,4, Beatrice De Schepper1,5, Dina De Sousa1,6, Paul De Sousa1,6, Cristina Ferreira1,2, Hans Gommans1,7, Rob Haselberg1,7, Emilie Hermant1,8, Danuta Lis1,9, Sabrina Maffi1, Svein Olaf Olsen1, Marios Papantoniou1, Ferdinando Squitieri10, Marina Tretyakova1, Zaynab Umakhanova1,13, Vladimír Václavík1, Michaela Winkelmann1 and Astri Arnesen1,11 on behalf of the European Huntington Association

1 European Huntington Association, Belgium
2 Associação Portuguesa dos Doentes de Huntington, Portugal
3 Asociación Corea de Huntington Española, Spain
4 Fondazione Lega Italiana Ricerca Huntington, Italy
5 Huntington Liga, Belgium
6 Scottish Huntington's Association, UK
7 Vereniging Van Huntington, The Netherlands
8 L'Association Huntington France, France
9 Polskie Stowarzyszenie Choroby Huntingtona, Poland
10 Huntington and Rare Diseases Unit, IRCCS Casa Sollievo della Sofferenza Research Hospital, Italy
11 Landsforeningen for Huntingtons sykdom, Norway
12 Huntington's Disease Association of Cyprus, Cyprus
13 Orphan People, Russia
14 Spoločnost' Pre Pomoc Pri Huntingtonovej Chorobe, Slovakia
15 Deutsche Huntington Hilfe E.V., Germany

Despite the multiple achievements in Huntington’s disease (HD) research over the last years, effective disease-modifying treatments are still lacking. Because clinical progresses imply a strong commitment from HD families, they should be heard in this process. Our aim was to establish which factors affect the willingness of persons at risk for HD (HDRisk) and persons with premanifest HD (PreHD) to take part in studies. Specifically, this work investigated the view of the Dutch HDRisk and PreHD communities regarding research participation.

The European Huntington Association created a multi-language online survey to capture the research perceptions and experiences of persons with HDRisk and PreHD. Questions assessed research experience and knowledge, information sources, reasons for involvement and noninvolvement in research, and factors preventing and facilitating study participation. The Dutch survey responses were examined and compared with the global survey results.

The survey was filled by 525 persons with HDRisk and PreHD. We found that the Dutch survey respondents (DSR, n=87) relied more on their family and on television to get research information than the global survey respondents (GSR, n=438). Overall, the reasons and factors enabling study involvement were less important for the DSR than the GSR. The need to travel to the study site and invasive procedures were greater obstacles to participation for the DSR than the GSR. Importantly, the information and support provided by others had a reduced influence on the DSR study engagement compared to GSR. Lastly, negative emotions were less important obstacles to DSR research participation.

The Dutch survey responses generally met the global survey results. The DSR show high motivation to take part in studies, despite having limited research experience and knowledge. This motivation is affected by tangible factors such as site distance and procedure invasiveness, but not care burden, suggesting that the DSR consider their formal/informal healthcare assistance model to be effective. Notably, the DSR motivation is less influenced by external factors and emotional status compared to GSR, suggesting that the Dutch HDRisk and PreHD communities present an intrinsic motivation to participate in research that may be enhanced through family education and multimedia campaigns.

Clinical Research; Survey; Motivation