Parents' Decision-Making in Newborn Screening: Opinions, Choices, and Information Needs

Lipstein, Ellen A.; Nabi-Burza, Emara; Perrin, James M.; Luff, Donna; Browning, Marsha F.; Kuhlthau, Karen

doi:10.1542/peds.2010-0217

Cited by 28 publications

(37 citation statements)

References 27 publications

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“…Several studies have found that having adequate information improved parents' experiences of positive screens. 31,35,37 In our study, such parents recounted confusion and distress, with some having no memory that their child had even been screened. Providing key information well before delivery could help alleviate later distress by preparing parents for the possibility of positive screening results, and reinforcing the difference between screening and diagnosis.…”

Section: Discussionmentioning

confidence: 80%

“…17,29 Results are consistent with a growing literature revealing very supportive parental views of NBS. 27,29,[35][36] It is important to appreciate that the majority of parents in NBS programs are 'satisfied customers': 37 most will receive screen negative results, and in the case of acceptance of screening, their understanding is not put to the test. It highlights the importance, in research and quality assurance studies, of seeking out those participants who are most likely to provide a discordant view, however infrequent this is.…”

Section: Discussionmentioning

confidence: 99%

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Consent for newborn screening: parents’ and health-care professionals’ experiences of consent in practice

et al. 2016

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Consent processes for newborn bloodspot screening (NBS) are variable, with a lack of descriptive research that depicts how the offer of NBS is made to parents. We explored the experience, in practice, of consent for NBS. Semistructured interviews in two Canadian provinces were held with: (1) parents of children offered NBS (n = 32); and (2) health-care professionals involved in the NBS process (n = 19). Data on recollections of NBS, including consent processes, were utilized to identify emerging themes using the method of constant comparison. Three themes were relevant to NBS consent: (1) The 'offer' of NBS; (2) content and timing of information provision; and (3) the importance of parental experiences for consent decisions. Recollections of consent for NBS were similar between jurisdictions. Excepting midwives and their patients, NBS was viewed as a routine part of giving birth, with little evidence of an informed consent process. Although most parents were satisfied, all respondents suggested information about NBS be provided long before the birth. Accounts of parents who declined screening highlight the influence of parental experiences with the heel prick process in screening decisions. Findings further our understanding of consent in practice and highlight areas for improvement in parent-provider interactions.

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Section: Discussionmentioning

confidence: 80%

Section: Discussionmentioning

confidence: 99%

Consent for newborn screening: parents’ and health-care professionals’ experiences of consent in practice

et al. 2016

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“…being included in the review, 8,12,14,15,17,19,20,25,26,[28][29][30]33, covering NBS in a number of different countries: England (n = 11 8,14,25,62,66,70,72,[78][79][80]83 ), Wales (n = 2 85,86 ), France (n = 1 89 ), Germany (n = 1 15 ), the Netherlands (n = 1 29 ), USA (n = 20 17,19,26,30,33,61,63,64,71,[74][75][76]81,82,84,87,88,90,91,93 ), Canada (n = 4 12,65,…”

Section: Theories Of Informed Consent: Developing An Analytical Framementioning

confidence: 99%

“…Although numerous papers focused on parents' information needs, these were sometimes inferred from guidelines or from health professionals' views rather than by asking parents directly. Studies using parent samples found that the following topics were valued: l testing -how this is carried out 14,66,70,76,77 l results -when they are provided and how 14,26,63,70,76,77 l the purpose of screening 62,63,70,77 l choice 14,62,66,70,72 l conditions screened for: 14,70,76 ¢ detailed 76 ¢ basic and implications 12,14 l storage 30,68,77 l treatment 63,70,76 l carriers 29,64,78 l the importance of having a child screened/benefits 62,63 l risks -infections 70,78 l the need for further samples/how to respond if in the USA 26,78 70 l information about the fact that screening uses deoxyribonucleic acid (DNA) 70 l heritability 76 l screening alternatives (e.g. testing later in life) …”

Section: Content Of Relevant Informationmentioning

confidence: 99%

“…26,[28][29][30]33,68,93 Some parents were aware of this and admitted that they had made a decision about NBS without knowing information about symptoms, treatment, severity and hereditability. 76 Other studies, however, suggested that parents were not aware of their limited knowledge. For example, in one of the studies reporting the highest levels of awareness of the purpose of NBS (82%; n = 712), only one-third of parents knew that it was possible to receive a false-positive result, 77 an important part of preparing parents for the outcomes of NBS.…”

Section: Understandingmentioning

confidence: 99%

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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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Diagnostic experiences of Duchenne families and their preferences for newborn screening: A mixed‐methods study

Crossnohere

Armstrong

Fischer

et al. 2022

American J of Med Genetics Pt C

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Duchenne muscular dystrophy is the most common form of muscular dystrophy diagnosed in childhood but is not routinely screened for prenatally or at birth in the United States. We sought to characterize the diagnostic experiences of families and describe their preferences for newborn screening (NBS). We conducted a registrybased survey of families with Duchenne and Becker muscular dystrophy that included open-and closed-ended questions regarding the journey to a diagnosis, preferences for when to learn of a diagnosis, and how knowledge of a diagnosis would impact life decisions. Open-ended responses were analyzed thematically, and closed-ended responses were analyzed descriptively. Sixty-five families completed the survey. The average ages of first concern and diagnosis were 2 and 4 years, respectively. One-third of families (30%) indicated that they would prefer to receive a diagnosis in the newborn period irrespective of treatment options available, and nearly all of the remaining families (93%) indicated that they would want to learn about a diagnosis if there were treatments that worked well during the newborn period. All families (100%) indicated that a diagnosis in the newborn period would impact life decisions. We identified three overarching themes, which described the stages of the diagnostic journey, including having concerns about the child, seeking answers, and receiving the diagnosis. NBS can facilitate improved health outcomes through early access to care, and inform families on major health and nonhealth decisions. The preferences and experiences of families and other stakeholders should be considered when determining the potential value and benefit of expanding NBS programs.

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Parents' Decision-Making in Newborn Screening: Opinions, Choices, and Information Needs

Cited by 28 publications

References 27 publications

Consent for newborn screening: parents’ and health-care professionals’ experiences of consent in practice

Consent for newborn screening: parents’ and health-care professionals’ experiences of consent in practice

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

Diagnostic experiences of Duchenne families and their preferences for newborn screening: A mixed‐methods study

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