Title: Rethinking Strategies for Positive Newborn Screening Result Delivery (ReSPoND): a process evaluation of co-designing interventions to minimise impact on parental emotional well-being and stress
Funder: Department of Health and Social Care

Abstract: Background: Annually, circa 10,000 parents of babies born in the United Kingdom (UK) are given a positive newborn screening (NBS+) result 2-8 weeks after birth(1,2). Despite guidance(3), NBS+ results are inconsistently delivered across UK-regions(4-7) and many parents are dissatisfied with how NBS+ results are communicated(5-12). Expansion of UK NBS to include nine conditions means added pressure for a cost effective approach to communication of NBS+ results. As most infants will be asymptomatic when parents receive the NBS+ result(1, 2), it is vital that communication is done carefully to avoid potential negative effects on future concordance with treatment and relationships with health professionals(4,5,11-13). Concordance and trust are important(14) to ensure timely uptake of confirmatory diagnostic testing and instigation of necessary treatment to maximise outcomes for the child(1,2). Poor communication can affect parent-child bonding and ongoing parental and social relationships(5,6,15). As such, family systems theory(16) will be the theoretical basis for this work. Study aim: To co-design, implement and evaluate interventions to improve delivery of NBS+ results to parents. Primary research question: Can parents and staff co-design interventions to improve delivery of NBS+ results to parents that can be successfully implemented into routine practice in a cost effective manner? Secondary research questions: - How are NBS+ results currently delivered to parents for the condition specific groups (CSGs) and what are the perceived benefits? - What are the current experiences of staff delivering, and parents receiving, NBS+ results for the CSGs? - What aspects of the new, co-designed interventions and local approaches to implementation are important in terms of improving the delivery of NBS+ results? - What are the costs associated with the delivery of the new, co-designed interventions and subsequent use of healthcare services and how does this compare with the costs associated with current strategies? - Which outcomes will be important to include in a subsequent evaluation study of the new, co-designed interventions from a stakeholder perspective? Study design: A mixed-methods study underpinned by family systems theory(16) using four phases with defined outputs: Phase 0: Establish project management arrangements and secure ethics and HRA approvals. Phase 1: A national survey using telephone interviews with all NBS-laboratories in England and representatives of clinical teams to explore current approaches for communication of NBS+ results and inform selection of two study sites for phases 2-3. Outputs: (i) description and cost of current communication practices (ii) selection of two study sites for Phases 2-3. Phase 2: Experience-based Co-design(17-23) will be used in the selected study sites to develop interventions for communicating NBS+ results to parents. Output: Interventions for the four CSGs. Phase 3: A parallel process evaluation(24,25), underpinned by Normalisation Process Theory(26,27) and economic analysis of the interventions in the two selected case study sites. Outputs: (i) process evaluation and economic analysis of the interventions in routine practice (ii) information about suitable outcome measures for a future study. Phase 4: Determine final interventions and design of a future evaluation study Outputs: (i) Design of a future evaluation study (ii) list of suitable outcome measures and relevant resource use and cost Dissemination Everyone directly involved in the study will be sent a summary of the research findings. Study findings will also be disseminated on the national NBS websites https://www.gov.uk/topic/population-screening-programmes/newborn-bloodspot,https://www.gov.uk/topic/population-screening-programmes/sickle-cell-thalassaemia). Additionally, the findings will be shared via the website of the relevant charities and support groups associated with these conditions (CF Trust, Sickle Cell Society, British Thyroid Foundation, National Society for Phenylketonuria, CLIMB). Finally, results will be disseminated by the usual academic routes at relevant national and international conferences and published in relevant international peer reviewed journals (including the NIHR HS&DR journal). References 1. Public Health England. Data Collection and Performance Analysis Report Newborn blood spot screening in the UK 2014/15. London; 2016. 2. Public Health England. NHS Sickle Cell and Thalassaemia Screening Programme Data Report 2014/15: Trends and performance analysis. London; 2016 3. UK Newborn Screening Programme Centre. Health Professional Handbook: A guide to newborn blood spot screening for healthcare professionals. London: UK Newborn Screening Programme Centre; 2012. 4. Parker H, Qureshi N, Ulph F, Kai J. Imparting carrier status results detected by universal newborn screening for sickle cell and cystic fibrosis in England: a qualitative study of current practice and policy challenges. BMC Health Serv Res. 2007;7:203. 5. Ulph F, Cullinan T, Qureshi N, Kai J. Parents' responses to receiving sickle cell or cystic fibrosis carrier results for their child following newborn screening. Eur J Hum Genet. 2015;23(4):459-65. 6. Chudleigh J, Buckingham S, Dignan J, O'Driscoll S, Johnson K, Rees D, et al. Parents' Experiences of Receiving the Initial Positive Newborn Screening (NBS) Result for Cystic Fibrosis and Sickle Cell Disease. J Genet Couns. 2016. 7. Finan C, Nasr SZ, Rothwell E, Tarini BA. Primary care providers' experiences notifying parents of cystic fibrosis newborn screening results. Clin Pediatr (Phila). 2015;54(1):67-75. 8. Parsons EP, Bradley DM. Psychosocial issues in newborn screening for cystic fibrosis. Paediatric respiratory reviews. 2003;4(4):285-92. 9. Tluczek A, Koscik RL, Farrell PM, Rock MJ. Psychosocial risk associated with newborn screening for cystic fibrosis: parents' experience while awaiting the sweat-test appointment. Pediatrics. 2005;115(6):1692-703. 10. Buchbinder M, Timmermans S. Newborn screening for metabolic disorders: parental perceptions of the initial communication of results. Clin Pediatr (Phila). 2012;51(8):739-44. 11. Collins JL, La Pean A, O'Tool F, Eskra KL, Roedl SJ, Tluczek A, et al. Factors that influence parents' experiences with results disclosure after newborn screening identifies genetic carrier status for cystic fibrosis or sickle cell hemoglobinopathy. Patient Educ Couns. 2012. 12. Salm A, Yetter E, Tluczek A. Informing parents about positive newborn screening results: Parents' recommendations Journal of Child Health Care. 2012;16(4):367-81. 13. Ulph F, Cullinan T, Qureshi N, Kai J. The impact on parents of receiving a carrier result for sickle cell or cystic fibrosis for their child via newborn screening. Eur J Hum Genet. 2014;22. 14. Steuten L, Buxton M. Economic evaluation of healthcare safety: which attributes of safety do healthcare professionals consider most important in resource allocation decisions? Quality & safety in health care. 2010;19(5):e6. 15. Tluczek A, Clark R, McKechnie AC, Brown RL. Factors affecting parent-child relationships one year after positive newborn screening for cystic fibrosis or congenital hypothyroidism. J Dev Behav Pediatr. 2015;36(1):24-34. 16. Segrin C, Flora J. Family Communication. 2nd ed. London: Routledge; 2011. 17. Bate SP, Robert G. Bringing user experience to health care improvement: the concepts, methods and practices of experience-based design. Oxford: Radcliffe Publishing 2007. 18. Tsianakas V, Robert G, Maben J, Richardson A, Dale C, Wiseman T. Implementing patient centred cancer care: using experience-based co-design to improve patient experience in breast and lung cancer services. Support Care Cancer. 2012;20(11):2639-47. 19. Robert G. Participatory action research: using Experience-based Co-design (EBCD) to improve the quality of health care services. In: Ziebland S, Coulter A, Calabrese J, Locock L, editors. Understanding and Using Health Experiences: Improving patient care. Oxford: Oxford University Press; 2013. 20. Locock L, Robert G, Boaz A, Vougioukalou S, Shuldham C, Fielden J, et al. Using a national archive of patient experience narratives to promote local patient-centered quality improvement: an ethnographic process evaluation of 'accelerated' experience-based co-design. J Health Serv Res Policy. 2014;19(4):200-7. 21. Robert G, Cornwell J, Locock L, Purushotham A, Sturmey G, Gager M. Patients and staff as codesigners of healthcare services. BMJ. 2015;350:g7714. 22. Donetto S, Pierri P, Tsianakas V, Robert G. Experience-based Co-design and healthcare improvement: realising participatory design in the public sector. The Design Journal. 2015;18(2):227-48. 23. Tsianakas V, Robert G, Richardson A, Verity R, Oakley C, Murrells T, et al. Enhancing the experience of carers in the chemotherapy outpatient setting: an exploratory randomised controlled trial to test impact, acceptability and feasibility of a complex intervention co-designed by carers and staff. Support Care Cancer. 2015. 24. 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Lay abstract: Each year about 800,000 babies in the UK have a blood test taken to screen for specific conditions, which if treated early will improve the child’s health and well-being (known as newborn screening (NBS)). In 2014-15, over 10,000 babies were identified as being affected or healthy carriers of a gene for one of the conditions screened for, which include sickle cell disease, cystic fibrosis, metabolic diseases and hypothyroidism. Parents’ agree to the blood test at the time of birth but many never expect a positive result. When a positive result occurs, a variety of ways are used to deliver the result but many parents complain about the manner in which the information is communicated to them. With the expansion of NBS in England, the importance of delivering this information appropriately to minimise any long-term negative health and psychological consequences is vital. Our Aim: For parents and health professionals to work together to design interventions to facilitate effective communication of positive NBS results to parents by health professionals. We are working with parent representatives for each screened condition covered by NBS who will form an advisory group to assist the research team, provide feedback on each phase of the project and monitor its progress. Charities whose work focuses on the conditions screened for have provided their support for this work. The research takes place in four phases: Phase 0: Gaining the necessary approvals to do the work Phase 1: What is happening now? We will find out how NBS results are communicated from laboratories to parents in all parts of England for the screened conditions. This will help us understand differences in terms of current practices and associated benefits. It will also help us decide which sites will be used for phases 2 and 3. Phase 2: Parents and health professionals working together to design the best way(s) to communicate positive NBS results We will watch how NBS results are delivered to parents and then ask parents and health professionals about their experiences in two study sites. Following this we will ask parents and health professionals to work together through a series of workshops to design approaches (interventions) for the effective and sensitive communication of positive NBS results to parents of affected children. Phase 3: Evaluating the different approaches We will start using the new approaches designed in Phase 2 in the two study sites and compare them to usual practice observed in Phase 2. We will watch how the new approaches are used and ask parents and health professionals which approach they think is best, how it affects them and work out how much each will cost, who needs to be involved and any factors that may affect how well the approaches work in practice such as language barriers. We will compare costs associated with the new, co-designed interventions with existing practices described in Phase 1. Phase 4: Future Directions Information from the first three phases will be reviewed and plans made for a future study to evaluate the chosen approach. Dissemination: The findings will be shared with support groups and charities, along with health professionals and academics.

Research activity codes: 8.1 ORGANISATION AND DELIVERY OF SERVICES
Health categories: REPRODUCTIVE HEALTH AND CHILDBIRTH