The ethics conundrum in Recall by Genotype (RbG) research: Perspectives from birth cohort participants

Minion, Joel; Butcher, Frances; Timpson, Nicholas J.; Murtagh, Madeleine J.

doi:10.1101/124636

Cited by 6 publications

(9 citation statements)

References 22 publications

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“…The ethical papers were mostly focused on the perspective of participants to RbG: authors investigated participants' understanding of the rationale of RbG-designed studies [15], the experience of participation in genetic research [15,19,20], the expectations associated with and the experience of recruitment in RbG studies [19,21], and the communication of individual genetic results (meanings of genetic results, disclosure) [15,[20][21][22][23] (Table 1).…”

Section: Identification Of Relevant Rbg Ethical Issuesmentioning

confidence: 99%

“…On the other hand, in population studies where RbG was not foreseen at the outset, re-call could be performed without revealing the main reason for it. While such an approach may appear unacceptable on principle, currently, there are no recognized ethical and legal standards for RbG in cohorts where RbG was not planned and which was therefore not included in the original informed consent [2,6,15,16].…”

Section: Introductionmentioning

confidence: 99%

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Balancing scientific interests and the rights of participants in designing a recall by genotype study

et al. 2021

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Recall by genotype (RbG) studies aim to better understand the phenotypes that correspond to genetic variants of interest, by recruiting carriers of such variants for further phenotyping. RbG approaches pose major ethical and legal challenges related to the disclosure of possibly unwanted genetic information. The Cooperative Health Research in South Tyrol (CHRIS) study is a longitudinal cohort study based in South Tyrol, Italy. Demand has grown for CHRIS study participants to be enrolled in RbG studies, thus making the design of a suitable ethical framework a pressing need. We here report upon the design of a pilot RbG study conducted with CHRIS study participants. By reviewing the literature and by consulting relevant stakeholders (CHRIS participants, clinical geneticists, ethics board, GPs), we identified key ethical issues in RbG approaches (e.g. complexity of the context, communication of genetic results, measures to further protect participants). The design of the pilot was based on a feasibility assessment, the selection of a suitable test case within the ProtectMove Research Unit on reduced penetrance of hereditary movement disorders, and the development of appropriate recruitment and communication strategies. An empirical study was embedded in the pilot study with the aim of understanding participants’ views on RbG. Our experience with the pilot study in CHRIS allowed us to contribute to the development of best practices and policies for RbG studies by drawing recommendations: addressing the possibility of RbG in the original consent, implementing tailored communication strategies, engaging stakeholders, designing embedded empirical studies, and sharing research experiences and methodology.

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Section: Identification Of Relevant Rbg Ethical Issuesmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Balancing scientific interests and the rights of participants in designing a recall by genotype study

et al. 2021

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“…Our ndings here report on the participants' experiences and thoughts on how CO90s engaged with them and the resultant relationship and trust they developed for it. This kind of trust that participants develop for their longitudinal cohort studies has been mentioned elsewhere [37] including in other studies [38] such as the Irish Longitudinal Study of Ageing (TILDA) which reported that their participants felt they had a bond with the study and a special rapport with data collectors and that this impacted on participant retention [39]. Consistent with previous writings [40] we have evidenced here that cohort study participation should not be considered a unidirectional activity, the interviews reported here demonstrate that the perceived reciprocity contributed to sustaining the long-term commitment required for a longitudinal study.…”

Section: Discussionmentioning

confidence: 92%

What does engagement mean to participants in longitudinal cohort studies? A qualitative study

Ochieng

Minion

Turner

et al. 2021

Preprint

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Background Engagement is important within cohort studies for a number of reasons. It is argued that engaging participants within the studies their involved in may promote their recruitment and retention within the studies. It is also thought that participant input can improve study designs, make them more acceptable for uptake by participants and aid in contextualising research communication to participants. Ultimately it is also argued that engagement needs to provide an avenue for participants to feedback to the cohort study and that this is an ethical imperative. This study sought to explore the participants’ experiences and thoughts of their engagement with their birth cohort study. Methods Participants were recruited from the Children of the 90s (CO90s) study. Qualitative semi-structured interviews were conducted with 42 participants. The interviews were transcribed verbatim, and uploaded onto Nvivo software. They were then analysed via thematic analysis with a constant comparison technique. Results Participants’ experiences of their engagement with CO90s were broadly based on three aspects: communication they received from CO90s, experiences of ethical conduct from CO90s and receiving rewards from CO90s. The communication received from CO90s, ranged from newsletters explaining study findings and future studies, to more personal forms like annual greeting cards posted to each participant. Ethical conduct from CO90s mainly involved participants understanding that CO90s would keep their information confidential, that it was only involved in ‘good’ ethical research and their expectation that CO90s would always prioritise participant welfare. Some of the gifts participants said they received at CO90s included toys, shopping vouchers, results from clinical tests, and time off from school to attend data collection (Focus) days. Participants also described a temporality in their engagement with CO90s and the subsequent trust they had developed for the cohort study. Conclusions The experiences of engagement described by participants were theorized as being based on reciprocity which was sometimes overt -in receiving gifts from CO90s and other times more nuanced- for example through CO90s ethical conduct. This perceived reciprocity aligns well with Maussian conception of gifting and in this case espoused participants’ trust in the cohort resulting in a unique participant-cohort study relationship.

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“…ALSPAC's strict procedures of anonymity and confidentiality are made explicitly clear to participants [ 31 ]. Qualitative research reported participants were trusting and had faith in ALSPAC and ‘considered themselves part of the ALSPAC team’ [ 10 ]. This may help to promote more accurate and honest drug use self‐reporting.…”

Section: Discussionmentioning

confidence: 99%

“…The Avon Longitudinal Study of Parents and Children (ALSPAC), a large United Kingdom (UK) birth cohort, has a long‐standing relationship between participants and researchers. Participants report a high level of trust in the study [ 10 ], which may facilitate accurate reporting of illicit drug use. We compare illicit drug use data from the ALSPAC birth cohort with the population‐based household Crime Survey for England and Wales (CSEW).…”

Section: Introductionmentioning

confidence: 99%

Testing the validity of national drug surveys: comparison between a general population cohort and household surveys

et al. 2021

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Background and Aims There are concerns that national population‐based estimates of illicit drug use are underestimated. We investigated this by comparing estimates of illicit substance use at age 24 from the Crime Survey for England and Wales (CSEW) with a birth cohort (Avon Longitudinal Study of Parents and Children, ALSPAC) and by comparing the Smoking and Alcohol Toolkit Studies (STS/ATS) to ALSPAC. Design Cross‐sectional household survey and cross‐sectional data from one wave of a longitudinal birth cohort. Setting England and Wales. Participants Young adults aged 23–25 reporting on substance use in 2017 to CSEW (n = 1165), ALSPAC (n = 3389) and STS/ATS (n = 950). Measurements Lifetime and past‐year illicit drug use, smoking status and hazardous drinking at age 24. Findings The 2017 CSEW estimate of lifetime illicit drug use was 40.6%, compared with 62.8% in ALSPAC (risk difference % [RD%] = 22.2%; 95% CI = 18.9–25.5%; P ≤ 0.001). The RD in lifetime use between ALSPAC and the CSEW was 23.2% (95% CI = 20.0–26.4%) for cannabis, 16.9% (95% CI = 14.4–19.4%) for powder cocaine and 24.8% (95% CI = 22.6–27.0%) for amphetamine. Past‐year drug use was 16.4% in CSEW, compared with 36.7% in ALSPAC (RD% = 20.3%; 95% CI = 17.6–23.0%; P ≤ 0.001). For past‐year substance use, the RD between ALSPAC and the CSEW was 15.4% (95% CI = 12.9–17.9%) for cannabis, 14.8% (95% CI = 13.0%–16.6%) for powder cocaine and 15.9% (95% CI = 14.5–17.4%) for amphetamine. Levels of current smoking were similar between STS (27.4%) and ALSPAC (29.4%). Hazardous drinking was substantially higher in ALSPAC (60.3%) than the ATS (32.1%; RD% = 28.2%; 95% CI = 24.8–31.6%; P ≤ 0.001). Conclusions The Avon Longitudinal Study of Parents and Children provides one source of validation for measurements of drug use in government household surveys and indicates that illicit drug use may be underestimated in the Crime Survey for England and Wales.

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The ethics conundrum in Recall by Genotype (RbG) research: Perspectives from birth cohort participants

Cited by 6 publications

References 22 publications

Balancing scientific interests and the rights of participants in designing a recall by genotype study

Balancing scientific interests and the rights of participants in designing a recall by genotype study

What does engagement mean to participants in longitudinal cohort studies? A qualitative study

Testing the validity of national drug surveys: comparison between a general population cohort and household surveys

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