Dharni, Nimarta

Gray, Ewan

Payne, Katherine

Ulph, Fiona

Wright, SJ

English

Purpose: Current approaches to information provision in the context of national Newborn Bloodspot Screening Programmes (NBSP) are not sufficient in terms of maximising parents’ ability to make an informed decision. The challenge of information provision will be exacerbated by the proposed expansion of NBSP to include more conditions. This study aimed to design a stated choice experiment (sCE) to understand preferences for information provision in an expanded NBSP in the UK. Method: Mixed methods were used to define the (i) choice question (ii) type of sCE (iii) attributes and levels. Four methods were used: (i) systematic review of published (2001-2014) discrete choice experiments (DCEs) eliciting preferences for screening, diagnostic or genetic tests or maternity-related services; (ii) rapid review of NHS-NBSP policy and practice documents and training materials; (iii) consultation with NHS-NBSP experts (2 senior academic midwives; 4 screening co-ordinators; 2 laboratory scientists; a patient-support charity); (iv) semi-structured interviews with 6 parents and 7 regional quality assurance co-ordinators. Key attributes and levels identified from published DCEs and the policy/practice/training documents were tabulated. Qualitative data from experts and semi-structured interviews were analysed using thematic analysis together with data collected from follow-up cognitive (think aloud) interviews with parents and midwives to test attribute/level framing. Result: Emerging qualitative themes indicated: (i) the complexity of the choice question; (ii) the relevant study population (new parents; members of the public of child-bearing age; midwives); and (iii) three aspects needed in the sCE design: (a) the extent and type of information attributes (n=12) to facilitate ability to make an informed decision and (b) process attributes related to approaches to information provision (n=4) relative to the ability to make an informed decision (outcome attribute) and (c) capturing the impact of expansion in number of conditions tested by using two survey versions. These results suggested that a hybrid-conjoint analysis (part A) with DCE (part B) was the appropriate type of sCE. The identified published DCEs (n=58 studies) and the document review provided the candidate attributes for parts A and B. Additional background questions on respondents characteristics together with a structured tool to quantify the degree to which respondents seek information should be included in the survey. Conclusion: This study developed a hybrid sCE to provide a practical solution to understanding preferences in complex situations

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The University of Manchester ResearchDeveloping a Stated Choice Experiment to UnderstandPreferences for Information Provision in an ExpandedNewborn Bloodspot Screening ProgrammeLink to publication record in Manchester Research ExplorerCitation for published version (APA):Wright, SJ., Payne, K., Gray, E., Dharni, N., & Ulph, F. (2014). Developing a Stated Choice Experiment toUnderstand Preferences for Information Provision in an Expanded Newborn Bloodspot Screening Programme.Poster session presented at Society for Medical Decision Making 36th Annual North American Meeting, Miami.Citing this paperPlease note that where the full-text provided on Manchester Research Explorer is the Author Accepted Manuscriptor Proof version this may differ from the final Published version. If citing, it is advised that you check and use thepublisher's definitive version.General rightsCopyright and moral rights for the publications made accessible in the Research Explorer are retained by theauthors and/or other copyright owners and it is a condition of accessing publications that users recognise andabide by the legal requirements associated with these rights.Takedown policyIf you believe that this document breaches copyright please refer to the University of Manchester’s TakedownProcedures [http://man.ac.uk/04Y6Bo] or contact uml.scholarlycommunications@manchester.ac.uk providingrelevant details, so we can investigate your claim.Download date:12. Nov. 2022Developing a Stated Choice Experiment to Understand Preferences for Information Provision in an Expanded Newborn Bloodspot Screening ProgrammeStuart Wright1, Katherine Payne1, Ewan Gray1, Nimarta Dharni2 and Fiona Ulph2 1Manchester Centre for Health Economics, The University of Manchester, Manchester, UK.    2Manchester Centre for Health Psychology, The University of Manchester, Manchester, UK.Key messagesA hybrid stated choice experiment was designed to elicit parent and midwife preferences for newborn bloodspot screening (NBS) information provision.The survey comprised a conjoint analysis linked with a discrete choice experimentThe outcome of information provision ‘being able to make an informed decision’ was used as the linking attributeThe next phase is to pilot the survey design in a larger sample of parents (current and future) and practising midwivesIntroductionNewborn bloodspot screening (NBS) is a public health programme which aims to detect severe inherited illnesses in newborn children. By identifying children with such conditions at an early age, treatment can be given which can help to prevent or reduce the health implications to the child. In the UK, participation in the NBS should only occur after parent(s) have provided informed consent. However, evidence suggests that many parents have a poor understanding of NBS, with many not knowing “specifically what the testing had been for”1. Such lack of information can cause future problems such as parental anxiety and unnecessary health care resource use2. Given that the UK NBS is due to expand from the current five conditions to screening for nine conditions, these problem may only grow in magnitude. Understanding parents and midwives’ preferences for aspects of newborn bloodspot screening would allow this information to be tailored so as to allow it to be better taken on board. In theory, appropriately tailored information provision would reduce parental anxiety experienced on receipt of positive results or when a repeat test is needed. The experience and opinions of midwives must also be incorporated when designing such an intervention. This is particularly true as any additional time spent providing information will impact on their other duties or result in them working for longer hours. By balancing the preferences of both of these groups, a resource-effective programme of information provision can be constructed which prepares parents for screening and its possible consequences.AimTo design a stated choice experiment (sCE) to elicit parent and midwife preferences for information provision in an expanded NBS programme in the UK. MethodMixed methods were used to define the (i) choice question (ii) type of sCE (iii) attributes and levels. Four methods were used: A systematic review of published (2001-2014) discrete choice experiments (DCEs) eliciting preferences for screening, diagnostic or genetic tests or maternity-related services. The attributes and levels identified from published DCEs were tabulated and potentially relevant examples extracted, A rapid review of NHS-NBSP policy and practice documents and training materials. Key characteristics that a reported to be a requirement of information provision in NBS were listed.  Consultation with NHS-NBSP expert. The list of potentially relevant information attributes was discussed with experts.Semi-structured interviews with parents and regional quality assurance coordinators. Qualitative data elicited from interviews were analysed using thematic analysis. ResultsSystematic ReviewA total of 58 relevant DCEs were identified and unique attributes were tabulated for each type of intervention. Duplicate attributes were assimilated and the remaining unique attributes were grouped into 20 common themes. Themes which were not relevant to the policy context were then removed leaving 13 attributes, with 11 being represented in the conjoint analysis (part A of the survey) and 3 in the DCE (part B of the survey). Rapid ReviewThree key newborn screening documents were reviewed to aid the development of the sCE. These were:the current “Blood Spot Screening for You and Your Baby” leaflet, a set of postnatal notes which are given to parents and contain some information and a handbook designed for health professionals who take the bloodspot sample3. These documents were used to validate the attributes to be included in part A (reflecting types of information required) with the current screening leaflet representing current practice. The health professional handbook also helped to reinforce current practice.Qualitative interviews and Consultation with ExpertsSemi-structured interviews have also been used to inform the design of the sCE. Partici pants in these interviews included 20 parents and 7 regional quality assurance co-ordinators in newborn bloodspot screening.Two senior academic midwives, 4 screening co-ordinators, 2 laboratory scientists, and a patient-support charity also offered their opinions on the design of the experiment, although not within the same formal interviews.Emerging qualitative themes indicated: (i) the complexity of the choice question; (ii) the relevant study population (new parents; members of the public of child-bearing age; midwives); and (iii) three key aspects needed in the sCE design:The extent and type of information attributes to facilitate ability to make an informed decisionProcess attributes related to approaches to information provision relative to the ability to make an informed decision (outcome attribute)Capturing the impact of expansion in number of conditions tested by using two survey versions. These qualitative data suggested it was also necessary to use a measure to capture respondents’ views about shared decision making in the context of NBS. In this survey an adaptation of a published measure will be used4. Together the results suggested that a hybrid-conjoint analysis (part A – see Table 1) with DCE (part B – see Table 2) was the appropriate type of sCE. The two types of stated preference survey would be linked using the outcome of information provision ‘being able to make an informed decision’.Produced at the Graphics Support Workshop, Media Services, The University of Manchesterstuart.wright-2@manchester.ac.ukTable 1: Attributes included in the conjoint analysisa Each attribute is assigned two levels (yes – information is available to answer this question; no – information is not available to answer this question)Table 2: Attributes and levels in the discrete choice experiment Society for Medical Decision Making 36th Annual North American MeetingConclusionThis study developed a hybrid sCE to provide a practical solution to understanding preferences in complex situations. This was achieved by separating preferences for processes and outcomes into a conjoint analysis and sCE and including the individual’s preference rating for the former as an attribute in the latter.References1. Moody L. and Choudry K. Parental views on informed consent for expanded newborn   screening. Health Expectations 2011;16:3:239-2502. Waisbren SE, Albers S, Amato, S, Ampola M, Brewster TG, Demmer L, Eaton RB, Greenstein R, Korson M, Larson C, Marsden D, Msall M, Naylor EW, Pueschel S, Seashore M, Shih VE and Levy HL. Effect of Expanded Newborn Screening for Biochemical Genetic Disorders on Child Health Outcomes and Parental Stress. JAMA 2003;290:19:2564-25723. NHS Screening Programmes. Health Professional Handbook. December 2012. http://newbornbloodspot.screening.nhs.uk/health-professional-handbook (accessed October 3rd 2014)4. Bradley JG, Zia MJ, Hamilton N. Patient preferences for control in medical decision making:   a scenario-based approach. Fam. Med.  1996;28(7):496–501Acknowledgements The authors would like to thank the parents, midwives, and newborn screening experts who helped us to develop this stated preference survey. We are also grateful to Rodney Pollitt and Fiona Trotter for offering their comments as member of the scientific advisory group for the project. Finally we would like to thank the other members of the PINSA study team; Tina Lavender, Rebecca Bennett, Steve Roberts, Kieran Walshe and Suresh Victor for their contributions to the design of the experiment.This project was funded by the National Institute for Health Research HTA programme, project number 11-62-02DisclaimerThe views and opinions expressed therein are those of the authors and do not necessarily reflect those of the HTA programme, NIHR, NHS or the Department of Health.National Institute forHealth Research

Developing a Stated Choice Experiment to Understand Preferences for Information Provision in an Expanded Newborn Bloodspot Screening Programme

Wright, Stuart J

Developing a stated choice experiment to understand preferences for information provision in an expanded Newborn Bloodspot Screening Programme

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Developing a Stated Choice Experiment to Understand Preferences for Information Provision in an Expanded Newborn Bloodspot Screening Programme

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