Ellinor K. Olander

1. The postnatal period is defined as the time immediately after childbirth (WHO 2022). Black women and birthing people's1 experiences during this time occur within intersecting racial, economic, a...

Background: In 2021, the UK National Screening Committee commissioned an evaluation of newborn bloodspot screening for severe combined immunodeficiency (SCID) in England. This paper describes the experiences of parents who received an SCID or non-SCID T-cell lymphopenia (non-SCID TCL) result for their baby during the evaluation. Methods: A qualitative exploratory design was employed using semi-structured interviews with 12 parents (n = 5 who had received an SCID outcome and n = 7 who had received a non-SCID TCL following SCID NBS). Results: The impact on parents whose baby was diagnosed with SCID was complex, reflecting the experience of receiving a presymptomatic diagnosis. Parents of babies who had been diagnosed with a non-SCID TCL viewed their baby’s result in terms of risk; while their baby might still have a serious immunological condition, it was not considered to be as serious as SCID. All parents reported that they valued their participation in the SCID screening evaluation. Conclusions: Support for families following a positive screening result for SCID needs to be considered. This includes tailored psychosocial support, given their experiences will not be the same as those of parents of non-screened babies with SCID.

The project aimed to explore the psychosocial impact on parents of receiving a false-positive outcome following a positive newborn bloodspot screening (NBS) result for SCID for their child. A mixed-methods design was employed using semi-structured interviews and standardised health-related questionnaires (EQ-5D-5L, ITQOL-47, and GAD-7). The participants were recruited from six National Health Service hospital trusts in England involved in the NHS England In-Service Evaluation of Screening for SCID. A total of 22 interviews were conducted with 28 parents. Health-related questionnaire data were collected from 26 of these parents. The interviews were analysed using a reflexive deductive approach to thematic analysis. For the health-related questionnaire data, a comparison of group means against population norms was undertaken using t-tests with unequal variances. The findings from the interviews showed that receiving a false-positive outcome following a positive NBS SCID result could cause parents to have an enhanced view of their child’s vulnerability in the short term. However, negative sequelae were largely mitigated as parents viewed their child’s exposure to ‘normal’ infections as evidence of a functional immune system. The health-related questionnaire data showed that the parents had significantly worse health than the population norm (as indicated by EQ-VAS: p = 0.0296); however, all the other measures were non-significant. More research is needed to explore the potential longer-term psychosocial impact of a false-positive screening result for SCID on parents beyond their child’s first year of life.

We examined interprofessional working in a newly implemented parent-infant mental health service team supporting families experiencing bonding and attachment difficulties. The aim was to identify forms of interprofessional work undertaken, barriers and facilitators of this work, and families’ and healthcare professionals’ perceptions of it. Semi-structured interviews were carried out with 21 stakeholders (5 parents, 4 team clinicians, 9 service referrers, 3 service commissioners) and were analyzed thematically. Interprofessional activities identified included building the service team’s cohesion and shared practice, building partner networks, interagency communication, coordination of roles, and raising awareness of infant mental health and parent-infant relationship needs. Enablers and barriers to interprofessional working were broadly consistent with findings from previous studies of related services, but with additional emphasis on consultative work as an enabler. Healthcare professionals reported benefiting from the case consultations and training on infant mental health provided by the service team. Parents reported that good interprofessional working enhanced satisfaction and engagement with the service. Findings indicate the centrality of interprofessional working for parent-infant mental health teams, with implications for future service implementation, service development, and understanding of mechanisms by which such services may influence family outcomes.

Background: Each year in England, almost 10,000 parents are informed of their child’s positive newborn bloodspot screening (NBS) results. This occurs approximately 2 to 8 weeks after birth depending on the condition. Communication of positive NBS results is a subtle and skillful task, demanding thought, preparation, and evidence to minimize potentially harmful negative sequelae. Evidence of variability in the content and the way the result is currently communicated has the potential to lead to increased parental anxiety and distress. Objective: This study focused on the development of co-designed interventions to improve the experiences of parents receiving positive NBS results for their children and enhance communication between health care professionals and parents. Methods: An experience-based co-design approach was used to explore experiences and co-design solutions with 17 health professionals employed in 3 National Health Service Trusts in England and 21 parents (13/21, 62% mothers and 8/21, 38% fathers) of 14 children recruited from the same 3 National Health Service Trusts. Experiences with existing services were gathered via semistructured interviews with health professionals. Filmed narrative interviews with parents were developed into a composite film. The co-design process identified priorities for improving communication of positive NBS results through separate parent and health professional feedback events followed by joint feedback events. In total, 4 interventions were then co-designed between the participants through a web-based platform. Results: Parents and health professionals provided positive feedback regarding the process of gathering experiences and identifying priorities. Themes identified from the parent interviews included impact of initial communication, parental reactions, attending the first clinic appointment, impact of health professionals’ communication strategies and skills, impact of diagnosis on family and friends, improvements to the communication of positive NBS results, and parents’ views on NBS. Themes identified from the health professional interviews included communication between health professionals, process of communicating with the family, parent- and family-centered care, and availability of resources and challenges to effective communication. In response to these themes, 4 interventions were co-designed: changes to the NBS card; standardized laboratory proformas; standardized communication checklists; and an email or letter for providing reliable, up-to-date, condition-specific information for parents following the communication of positive NBS results. Conclusions: Parents and health professionals were able to successfully work together to identify priorities and develop co-designed interventions to improve communication of positive NBS results to parents. The resulting co-designed interventions address communication at different stages of the communication pathway to improve the experiences of parents receiving positive NBS results for their children. International Registered Report Identifier (IRRID): RR2-10.1186/s40814-019-0487-5

Background Newborn bloodspot screening identifies presymptomatic babies who are affected by genetic or congenital conditions. Each year, around 10,000 parents of babies born in England are given a positive newborn bloodspot screening result for one of nine conditions that are currently screened for. Despite national guidance, variation exists regarding the approaches used to communicate these results to families; poor communication practices can lead to various negative sequelae. Objectives Identify and quantify approaches that are currently used to deliver positive newborn bloodspot screening results to parents (phase 1). Develop (phase 2), implement and evaluate (phase 3) co-designed interventions for improving the delivery of positive newborn bloodspot screening results. Quantify the resources required to deliver the co-designed interventions in selected case-study sites and compare these with costs associated with current practice (phase 3). Design This was a mixed-methods study using four phases, with defined outputs underpinned by Family Systems Theory. Setting All newborn bloodspot screening laboratories in England (n = 13). Participants Laboratory staff and clinicians involved in processing or communicating positive newborn bloodspot screening results, and parents of infants who had received a positive or negative newborn bloodspot screening result. Interventions Three co-designed interventions that were developed during phase 2 and implemented during phase 3 of the study. Main outcome measure Acceptability of the co-designed interventions for the communication of positive newborn bloodspot screening results. Results Staff were acutely aware of the significance of a positive newborn bloodspot screening result and the impact that this could have on families. Challenges existed when communicating results from laboratories to relevant clinicians, particularly in the case of congenital hypothyroidism. Clinicians who were involved in the communication of positive newborn bloodspot screening results were committed to making sure that the message, although distressing for parents, was communicated well. Despite this, variation in communication practices existed. This was influenced by many factors, including the available resources and lack of clear guidance. Although generally well received, implementation of the co-designed interventions in practice served to illuminate barriers to acceptability and feasibility. The interventions would not influence NHS expenditure and could be cost neutral when delivered by teleconsultations. Limitations Participants with a pre-existing interest in this topic may have been more likely to self-select into the study. The researchers are experienced in this field, which may have biased data collection and analysis. COVID-19 hindered implementation and related data collection of the co-designed interventions. Conclusions There was variation in the processes used to report positive newborn bloodspot screening results from newborn bloodspot screening laboratories to clinical teams and then to families. The various practices identified may reflect local needs, but more often reflected local resource. A more consistent ‘best practice’ approach is required, not just in the UK but perhaps globally. The co-designed interventions represent a starting point for achieving this. Future work Future work should include a national evaluation study with predefined outcomes, accompanied by an economic evaluation, to assess the acceptability, feasibility and usability of the co-designed interventions in practice nationally. Trial registration This trial is registered as ISRCTN15330120. Funding This project was funded by the National Institute for Health and Care Research (NIHR) Health and Social Care Delivery Research programme and will be published in full in Health and Social Care Delivery Research; Vol. 10, No. 19. See the NIHR Journals Library website for further project information.

Although the communication pathways of Newborn Bloodspot Screening (NBS) are a delicate task, these pathways vary across different conditions and are often not evidence-based. The ReSPoND interventions were co-designed by healthcare professionals alongside parents who had received a positive NBS result for their child. To calculate the cost of these co-designed strategies and the existing communication pathways, we interviewed 71 members of the clinical and laboratory staff of the 13 English NBS laboratories in the English National Health Service. Therefore, a scenario analysis was used to compare the cost of the existing communication pathways to the co-designed strategies delivered by (i) home-visits and (ii) telecommunications. On average, the existing communication pathway cost £447.08 per infant (range: £237.12 to £628.51) or £234,872.75 (£3635.99 to £1,932,986.23) nationally. Implementing the new interventions relying on home-visits exclusively would cost on average £521.62 (£312.84 to £646.39) per infant and £297,816.03 (£4506.37 to £2,550,284.64) nationally, or £447.19 (£235.79 to £552.03) and £231,342.40 (£3923.7 to £1,922,192.22) if implemented via teleconsultations, respectively. The new strategies delivered are not likely to require additional resources compared with current practice. Further research is needed to investigate whether this investment represents good value for money for the NHS budget.

Background: Patient and public involvement (PPI) has become increasingly important in the development, delivery and improvement of healthcare. PPI is used in healthcare innovation; yet, how it is used has been under‐reported. The aim of this scoping review is to identify and map the current available empirical evidence on the role of PPI during different stages of healthcare innovation.Methods: The scoping review was conducted in accordance with PRISMAScR and included any study published in a peer‐reviewed journal between 2004 and 2021 that reported on PPI in healthcare innovation within any healthcare setting or context in any country. The following databases were searched: Medline, EMBASE, CINAHL, PsycInfo, HMIC and Google Scholar. We included any study type, including quantitative, qualitative and mixed‐method studies. We excluded theoretical frameworks, conceptual, scientific or grey literature as well as discussion and opinion papers.Results: Of the 87 included studies, 81 (93%) focused on or were conducted by authors in developed countries. A wide range of conditions were considered, with more studies focusing on mental health (n = 18, 21%) and cancer care (n = 8, 9%). The vast majority of the studies focused on process and service innovations (n = 62, 71%). Seven studies focused on technological and clinical innovations (8%), while 12 looked at both technological and service innovations (14%). Only five studies examined systems innovation (5%) and one study looked across all types of innovations (1%). PPI is more common in the earlier stages of innovation, particularly problem identification and invention, in comparison to adoption and diffusion.Conclusion: Healthcare innovation tends to be a lengthy process. Yet, our study highlights that PPI is more common across earlier stages of innovation and focuses mostly on service innovation. Stronger PPI in later stages could support the adoption and diffusion of innovation.Patient or Public Contribution: One of the coauthors of the paper (S. S.) is a service user with extensive experience in PPI research. S. S. supported the analysis and writing up of the paper.

BACKGROUND Each year in England, almost 10,000 parents are informed of their child’s positive newborn bloodspot screening result around 2-8 weeks after birth, depending on the condition. Communication of positive newborn bloodspot screening results is a subtle and skilful task, which demands thought, preparation and evidence to minimise potentially harmful negative sequelae. Evidence exists of variability in the content and the way the result is currently communicated which has the potential to lead to increased parental anxiety and distress. OBJECTIVE The main objective was to co-design interventions to improve delivery of positive newborn bloodspot screening results to families. METHODS The principles of Experience-based Co-design were used with seventeen health care professionals employed in three National Health Service Trusts in England and 21 parents; 13 mothers and 8 fathers of 14 children recruited from the same three National Health Service Trusts. Staff experiences were gathered via semi-structured interviews. Filmed, narrative interviews with parents were developed into a composite film. These data were used to identify priorities for improving communication of positive newborn bloodspot screening results to parents during firstly, separate parent and heath care professionals feedback events followed by joint parent and heath care professionals feedback events. Following this, parents and heath care professionals worked together via online co-design working groups to develop co-designed solutions and additions to existing processes. RESULTS Themes identified from the parent’s interviews included: impact of initial communication; parental reactions; attending the first clinic appointment; impact of staff communication strategies and skills; impact of diagnosis on family and friends; improvements to the communication of positive NBS results; and parents views of NBS. Themes identified from the staff interviews included: communication between health care professionals; process of communicating with the family; parent and family- centred care; availability of resources and challenges to effective communication. Three online co-design working groups were developed, each attended by 12-18 participants who had taken part in the parental or health care professionals’ interviews. The priorities included: changes to the NBS card; standardised laboratory proformas; standardised communication checklists; and an email / letter for providing reliable up to date condition specific information for parents following communication of the positive NBS result. CONCLUSIONS Variation in communication practices for positive NBS results continues to exist. This was influenced by many factors and has the potential to lead to negative sequelae from a parental perspective. Parents and health care professionals were able to successfully work together to identify priorities and develop potential solutions to improve communication of positive NBS results to parents. The adaptation of EBCD to include virtual methods could reduce costs associated with this methodology while also enabling the approach to be more responsive to health care professionals’ and patients’/parents’ busy schedules. CLINICALTRIAL ISRCTN 15330120 INTERNATIONAL REGISTERED REPORT RR2-10.1186/s40814-019-0487-5