Can a decision aid enable informed decisions in neonatal nursery recruitment for a fragile X newborn screening study?

Bailey, Donald B.; Bann, Carla; Bishop, Ellen; Guarda, Sonia; Barnum, Leah; Roche, Myra I.

doi:10.1038/gim.2012.135

Cited by 6 publications

(8 citation statements)

References 37 publications

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“…The research community is dependent on the integrity of the investigator to obtain informed consent and yet, the current format of the informed consent document complicates this process (Bailey et al, 2013). These concerns are magnified by the increasing length and complexity of consent forms, which further contributes to the lack of participant understanding (Henry et al, 2009).…”

Section: Discussionmentioning

confidence: 99%

A Randomized Controlled Trial of an Electronic Informed Consent Process

Rothwell

Wong

Rose

et al. 2014

Journal of Empirical Research on Human Research Ethics

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A pilot study assessed an electronic informed consent model within a randomized controlled trial (RCT). Participants who were recruited for the parent RCT project were randomly selected and randomized to either an electronic consent group (n = 32) or a simplified paper-based consent group (n = 30). Results from the electronic consent group reported significantly higher understanding of the purpose of the study, alternatives to participation, and who to contact if they had questions or concerns about the study. However, participants in the paper-based control group reported higher mean scores on some survey items. This research suggests that an electronic informed consent presentation may improve participant understanding for some aspects of a research study.

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Section: Discussionmentioning

confidence: 99%

A Randomized Controlled Trial of an Electronic Informed Consent Process

Rothwell

Wong

Rose

et al. 2014

Journal of Empirical Research on Human Research Ethics

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“…66,72,79 Asian participants or participants for whom English was not their first language were especially likely to prefer information provision by a health professional. 66 This option has some merit as it has been found to be the most effective way to convey complex information 96 and parents had a higher recall after verbal conversations 45 with midwives. 72 In addition, in a study that reported some of the highest levels of awareness across the literature, the majority of mothers in the sample (34%) had received their information from midwives.…”

Section: Content Of Relevant Informationmentioning

confidence: 99%

“…63 Some argue that providing information after birth does not give parents sufficient time to absorb the information. 96 Thus, there appears to be no doubt that there may be barriers to interacting with information, 18,66 with communication policies seeming to recognise this. For example, the AAP Newborn Screening Task Force suggests that information on NBS be provided in the third trimester.…”

Section: Retentionmentioning

confidence: 99%

Provision of information about newborn screening antenatally: a sequential exploratory mixed-methods project

Ulph

Wright

Dharni

et al. 2017

Health Technol Assess

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BackgroundParticipation in the UK Newborn Bloodspot Screening Programme (NBSP) requires parental consent but concerns exist about whether or not this happens in practice and the best methods and timing to obtain consent at reasonable cost.ObjectivesTo collate all possible modes of prescreening communication and consent for newborn (neonatal) screening (NBS); examine midwives’, screening professionals’ and users’ views about the feasibility, efficiency and impact on understanding of each; measure midwives’ and parents’ preferences for information provision; and identify key drivers of cost-effectiveness for alternative modes of information provision.DesignSix study designs were used: (1) realist review – to generate alternative communication and consent models; (2) qualitative interviews with parents and health professionals – to examine the implications of current practice for understanding and views on alternative models; (3) survey and observation of midwives – to establish current costs; (4) stated preference surveys with midwives, parents and potential future parents – to establish preferences for information provision; (5) economic analysis – to identify cost-effectiveness drivers of alternative models; and (6) stakeholder validation focus groups and interviews – to examine the acceptability, views and broader impact of alternative communication and consent models.SettingProviders and users of NBS in England.ParticipantsStudy 2: 45 parents and 37 health professionals; study 3: 22 midwives and eight observations; study 4: 705 adults aged 18–45 years and 134 midwives; and study 6: 12 health-care professionals and five parents.ResultsThe realist review identified low parental knowledge and evidence of coercive consent practices. Interview, focus group and stated preference data suggested a preference for full information, with some valuing this more than choice. Health professionals preferred informed choice models but parents and health professionals queried whether or not current consent was fully informed. Barriers to using leaflets effectively were highlighted. All studies indicated that a ‘personalised’ approach to NBS communication, allowing parents to select the mode and level of information suited to their learning needs, could have added value. A personalised approach should rely on midwife communication and should occur in the third trimester. Overall awareness was identified as requiring improvement. Starting NBS communication by alerting parents that they have a choice to make and telling them that samples could be stored are both likely to enhance engagement. The methods of information provision and maternal anxiety causing additional visits to health-care professionals were the drivers of relative cost-effectiveness. Lack of data to populate an economic analysis, confirmed by value of information analysis, indicated a need for further research.LimitationsThere are some limitations with regard to the range of participants used in studies 2 and 3 and so caution should be exercised when interpreting some of the results.ConclusionsThis project highlighted the importance of focusing on information receipt and identified key communication barriers. Health professionals strongly preferred informed consent, which parents endorsed if they were made aware of sample storage. Uniform models of information provision were perceived as ineffective. A choice of information provision was supported by health professionals and parents, which both enhances cost-effectiveness and improves engagement, understanding and the validity of consent. Remaining uncertainties suggest that more research is needed before new communication modes are introduced into practice. Future research should measure the impact of the suggested practice changes (informing in third trimester, information toolkits, changed role of midwife).Trial registrationCurrent Controlled Trials ISRCTN70227207.FundingThis project was funded by the NIHR Health Technology Assessment programme and will be published in full inHealth Technology Assessment; Vol. 21, No. 55. See the NIHR Journals Library website for further project information.

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“…We previously reported acceptance rates and reasons for accepting or declining screening, 12 prevalence of FMR1 premutation expansions, 34 fathers' participation in the consent process, 35 examples of how the identification of a target child can lead to identifying other family members, 36 and the development and evaluation of a brochure to support informed decision-making about study participation. 37,38 Here we report findings from an assessment of maternal reactions to the disclosure of their child's FMR1 carrier status after newborn screening. Our primary goal was to determine whether these mothers experienced adverse mental health outcomes (stress, anxiety, depressive symptoms, low quality of life), whether they regretted the decision to participate, and how adaptation over time varied as a function of the child's age or the availability of spousal support.…”

Section: What This Study Addsmentioning

confidence: 99%

“…Those who expressed interest were given much more detailed information by the recruiter, including the comprehensive consent form. Approximately halfway through the project a new brochure was developed to support informed decision-making 37,38 and was used at all 3 sites. The brochure included a section on what it means to be a "fragile X carrier," addressed the implications of carrier status for both newborns and parents, and made it clear that carrier detection was a much more likely outcome than the detection of children with FXS.…”

Section: Setting and Proceduresmentioning

confidence: 99%

Maternal Consequences of the Detection of Fragile X Carriers in Newborn Screening

et al. 2015

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The possibility of newborn screening for fragile X syndrome is complicated by the potential for identifying premutation carriers. Although knowing the child's carrier status has potential benefits, the possibility of late-onset disorders in carrier children and their parents raises concerns about whether such information would be distressing to parents and potentially more harmful than helpful. This study sought to answer this question by offering voluntary fragile X screening to new parents and returning results for both the full mutation and premutation FMR1 gene expansions. We tested the assumption that such information could lead to adverse mental health outcomes or decision regret. We also wanted to know if child age and spousal support were associated with the outcomes of interest.METHODS: Eighteen mothers of screen-positive infants with the premutation and 15 comparison mothers completed a battery of assessments of maternal anxiety, postpartum depression, stress, family quality of life, decision regret, and spousal support. The study was longitudinal, with an average of 3 assessments per mother. RESULTS:The premutation group was not statistically different from the comparison group on measures of anxiety, depression, stress, or quality of life. A subset of mothers experienced clinically significant anxiety and decision regret, but factors associated with these outcomes could not be identified. Greater spousal support was generally associated with more positive outcomes.CONCLUSIONS: Although we did not find evidence of significant adverse events, disclosure of newborn carrier status remains an important consideration in newborn screening policy.WHAT'S KNOWN ON THIS SUBJECT: Parents generally adapt well to newborn screening results, but reactions to carrier status for X-linked conditions are unknown. WHAT THIS STUDY ADDS:Results suggest that detection and disclosure of FMR1 newborn carrier status may not result in significant adverse events for mothers.

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Can a decision aid enable informed decisions in neonatal nursery recruitment for a fragile X newborn screening study?

Cited by 6 publications

References 37 publications

A Randomized Controlled Trial of an Electronic Informed Consent Process

A Randomized Controlled Trial of an Electronic Informed Consent Process

Provision of information about newborn screening antenatally: a sequential exploratory mixed-methods project

Maternal Consequences of the Detection of Fragile X Carriers in Newborn Screening

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