title: Preconceptional ancestry-based carrier couple screening for cystic fibrosis and haemoglobinopathies: what determines the intention to participate or not and actual participation?
abstract: This paper explores determinants of the intention to participate or not and of actual participation in preconceptional ancestry-based carrier couple screening for cystic fibrosis (CF) and haemoglobinopathies (HbPs). In total, 9453 individuals from a multi-ethnic population were invited. Invitees who had a partner and who were planning a pregnancy were the target population (33-36%). Test participation was conditional on survey participation. Those who refrained from test participation were asked to participate in the survey only. The questionnaire was based on the Theory of Planned Behaviour, which explains behaviour through intention. It was completed by 418 survey participants: 171 who intended to participate in the testing, and 247 who refrained from test participation. Both test intenders and offer decliners generally had a positive attitude towards test participation, and perceived high behavioural control. This applied to Western and non-Western survey participants equally. Offer decliners, however, perceived less control in terms of the time and effort needed for participation. Still, 68% of them intended to participate in the future if the screening would be offered routinely. Test intenders more often would draw reproductive consequences from test results, perceived a higher risk of being a carrier, more benefits and less adverse psychological outcomes. Feelings of stigmatisation were not an important issue, but 14% thought that there would be discrimination against carriers: among them more were non-Western survey participants. Preconceptional ancestry-based CF and HbPs carrier screening was evaluated as positive and desirable among Western and non-Western survey participants. The effort and time needed for participation were important reasons for declining participation, which might be overcome by improving access to the screening.
authors.affiliation: Department of Clinical Genetics, VU University Medical Center, Amsterdam, The Netherlands. p.lakeman@vumc.nl
authors.forename: Phillis
authors.initials: P
authors.lastname: Lakeman
authors.forename: Anne Marie Catharina
authors.initials: AM
authors.lastname: Plass
authors.forename: Lidewij
authors.initials: L
authors.lastname: Henneman
authors.forename: Pieter Dirk
authors.initials: PD
authors.lastname: Bezemer
authors.forename: Martina Cornelia
authors.initials: MC
authors.lastname: Cornel
authors.forename: Leo Pieter
authors.initials: LP
authors.lastname: ten Kate
mesh_terms: D000328:Adult; D000465:Algorithms; D001294:Attitude to Health; D003550:Cystic Fibrosis / Q000235:genetics*; D003625:Data Collection; D005191:Family Characteristics; D005260:Female; D006580:Genetic Carrier Screening; D005820:Genetic Testing / Q000379:methods / Q000523:psychology; D006453:Hemoglobinopathies / Q000235:genetics*; D006801:Humans; D033182:Intention; D008297:Male; D008954:Models, Biological; D010358:Patient Participation; D016742:Preconception Care / Q000379:methods*; D011795:Surveys and Questionnaires; D055815:Young Adult