'There was nothing, just absolute darkness': Understanding the needs of those caring for children and young people with complex neurodisability in a diverse UK context: A qualitative exploration in the ENCOMPASS study.

BACKGROUND: Children and young people (CYP) with complex neurodisability experience multiple physical, communication, educational and social challenges, which require complex packages of multidisciplinary care. Part of the holistic care required includes supporting the families and parents/caregivers. The aim of the wider study was to introduce a new programme ('Ubuntu') to parents/caregivers and healthcare professionals (HCPs) in order to test the feasibility and acceptability of the concept and content, with the goal of potential adaptation for the UK in mind. Data collection and analysis uncovered rich data on caregiving journeys, navigation of health services, and perceived service gaps. This paper focuses solely on these topics. Further papers will report on the feasibility and adaptation data. METHODS: Two rounds of semi-structured interviews were conducted with 12 caregivers of CYP with complex neurodisability and six HCPs from a variety of disciplines, recruited from a community child health service in London Borough of Newham, UK in 2020. The interviews included open-ended questions to explore caregiving journeys, experiences of navigating health services and perceived service gaps. Transcripts were analysed using a data-driven inductive thematic analysis. RESULTS: Three themes were identified that related to the aim of understanding caregivers' experiences and unmet needs relating to current service provision. These were (1) Caregiver Mental Health, (2) The Information Gap and (3) The Need for Holistic Support. Mental health difficulties were reported, particularly around the period of diagnosis. Priority needs included the provision of clear information about the diagnosis and services offered, opportunities to forge peer support networks and for services across the community to collaborate. CONCLUSIONS: The delivery of health services for CYP with neurodisability should encompass the broad needs of the family as well as meeting the clinical needs of the CYP.

What aspects of health and wellbeing are most important to parent carers of children with disabilities?

INTRODUCTION: Parent carers of children with special educational needs or disabilities are at risk of poorer health and wellbeing outcomes because of the distinct and challenging circumstances they face. Evaluations of interventions promoting the health of parent carers should focus on measuring the aspects of health and wellbeing which are most relevant to this group. As part of a programme of research on parent carer-focused interventions, this study aimed to understand which aspects of health and wellbeing are perceived by parent carers as most meaningful and important. METHODS: A qualitative study using semistructured online interviews was conducted. A purposive sample of parent carers was interviewed about relevant health and wellbeing outcomes. Transcripts were analysed thematically. RESULTS: Thirty parent carers were interviewed, 19 of whom had experienced a health-promoting intervention, either as participants (n = 14) or facilitators (n = 5). Three main themes were identified: 'self, identity and beliefs'; 'social connections and support' and 'health-promoting practices and outcomes.' Each theme encompassed the challenges participants faced, and the changes that helped them overcome these challenges. 'Self-identity' challenges focused on the overwhelming nature of the parental care role and the emotional impact of this. Changes were brought about by developing a positive mindset, increasing confidence, and reconnecting with aspects of their identity which were important to them before they became parent carers. Challenges related to 'social connections' reflected parent carers' isolation. Change was brought about through increased peer support and peer interactions. Parent carers experienced challenges in terms of 'health-promoting activities' because they lacked free time and experienced poor physical health. Changes were brought about by engagement in health-promoting activities of various kinds. CONCLUSION: Parent carers view health and wellbeing in terms of overcoming the common challenges they face as a group. These challenges reflect the ways in which their physiological and psychological needs are often unmet. Researchers interested in measuring parent carer health and wellbeing should consider the specific challenges this group face, as well as theoretical frameworks which can make sense of these challenges, such as self-determination theory. PATIENT OR PUBLIC CONTRIBUTION: Our team carries out patient and public involvement (PPI) through a Family Faculty group facilitated by a Family Involvement Co-ordinator (A. McD.) who is herself a parent carer. A study-specific PPI working group was established which included members of the Family Faculty. The PPI group advised on various aspects of the research as reported in the paper. The manuscript was co-authored by the team's Family Involvement Co-ordinator (A. McD.).

Barriers and facilitators to parent-delivered interventions for children with or infants at risk of cerebral palsy. An integrative review informed by behaviour change theory.

PURPOSE: Empowering parents to deliver evidenced-based interventions improves outcomes for children with or infants at risk of cerebral palsy (CP), by integrating repetition and contextual learning into daily routines. We aimed to identify the barriers and facilitators to parent-delivered interventions and suggest practice improvements guided by behaviour change models. METHODS: Eight electronic databases were searched to identify studies presenting parent and therapist perspectives on parent-delivered interventions in CP. Included studies were critically appraised using validated checklists. Barriers and facilitators to parent-delivered interventions were identified and categorised into subcomponents of The Capability Opportunity and Motivation Model of Behaviour (COM-B), the Theoretical Domains Framework (TDF) and the Behaviour Change Wheel to formulate appropriate practice recommendations. RESULTS: Thirty-four studies were identified which mainly used qualitative or randomised control trial designs. Barriers to parent-delivery included insufficient parental knowledge, lack of confidence and time. Facilitators included staff continuity, empowering parents, efficient resource utilisation and flexible delivery. Practice recommendations emphasise realistic goal setting, tailored parental education and enhancing the coaching skills of therapists. CONCLUSIONS: Fostering parent-delivered interventions requires addressing knowledge gaps, skill and capacity of parents and therapists. Therapists forming strong alliances with parents and setting collaborative realistic goals are key to successful parent-delivered interventions.

Enhancing parents' skills and knowledge regarding CP interventions can increase their confidence (psychological capability) in actively participating in intervention delivery.Structuring services to accommodate family schedules and providing adequate resources (physical and social opportunity) reduce the burden on families and facilitate their engagement.Providing training and resources to therapists enhances their skills and knowledge (psychological capability) in coaching and educating parents effectively.Emphasizing the value of collaborative goal setting in achieving positive outcomes for the child and acknowledging progress (reflective and automatic motivation) motivates both parents and therapists to actively engage in intervention delivery.

How do parents frame their engagement experience in early intervention? A grounded theory study.

PURPOSE: Parent and therapist engagement and partnership are critical in early intervention physiotherapy and occupational therapy for infants with cerebral palsy to improve outcomes. The main aim of this study was to understand how parents perceive their engagement experience in early intervention over time. METHODS: Grounded theory methodology was used. Twenty parents of diverse backgrounds participated in 22 interviews (including some repeated longitudinally) to reflect on their engagement experience within the context of early intervention community services provided in the UK NHS. RESULTS: The findings highlight how parents' perspectives of their engagement in EI change according to critical circumstances, including their preceding neonatal trauma, the at-risk CP label, firmer diagnosis of CP and their child's response to intervention. We theorise that this disrupted transition experience to parenthood becomes part of parental framing (or sense-making) of their engagement in EI. Overlapping frames of uncertainty, pursuit and transformation capture and explain nuances in parents' engagement patterns within EI over time. CONCLUSION: This theorising has implications for early intervention therapists in how they engage in the lives of families and partner with parents to support healthier parental transition, wellbeing and subsequent improved infant outcomes.

New insights are provided into how recent advances to prognostic practices for infants with cerebral palsy affect parental perspectives and their engagement experience in early interventionThe paper's organising concepts support a clearer understanding for early intervention practitioners of this complex parent experienceEarly intervention practitioners are encouraged to reflect upon their practice as they engage in families' lives and partner with parents during this challenging period to optimise outcomes.

Parental engagement in early intervention for infants with cerebral palsy-A realist synthesis.

BACKGROUND: Emphasis on parental engagement strategies within occupational therapy and physiotherapy early intervention (EI) programmes for infants at high risk of cerebral palsy (CP) has increased. This reflects consensus that increasing parent participation enhances treatment efficacy, potentially improving infant and parent outcomes. However, evaluation of parental engagement in EI is complex. Despite the growing application of parental engagement strategies, aligned with family-centred care practice, theoretical evaluation is currently lacking within the literature. This realist synthesis aimed to identify component theories underlying EI strategies to support parental engagement and to use empirical findings to evaluate how these work in practice. METHODS: Realist synthesis: Databases Medline, Embase, Amed, CINAHL and PsychInfo were searched (from February 1985 - February 2020); further articles were sourced from reference lists. A data extraction form was used, and a Critical Appraisal Skills Programme tool was used to assess study rigour. RESULTS: Twenty-six articles were included. Quality of relationships, parent education and intervention co-design were the key themes related to parental engagement strategies. Findings indicate that constructive parent reasoning mechanisms of trust, belief, sense of control, perceived feasibility of home programme delivery and ultimately motivation are linked to the underlying intervention resources afforded by specific strategies (e.g., coaching pedagogy). These responses are precursors to engagement outcomes that include increased parental self-efficacy and adherence. Importantly, parental self-efficacy can initiate a process of change leading to improved parental confidence and anxiety. CONCLUSIONS: Sensitively designed programme strategies, centred on relational quality between parent, infant and therapist, are fundamental for effective parent connection, involvement and investment within EI for infants with CP.

Feasibility, safety and acceptability of select outcome measures in a physiotherapy study protocol for boys with haemophilia.

BACKGROUND: There is a lack of functional performance measures for children and young people with haemophilia (CYPwH) with associated control data from typically developing boys (TDB). The literature advocates development of a core set of outcome measures for different chronic conditions. As medical treatment improves, CYPwH are experiencing better outcomes; therefore, more challenging measures are required to monitor physical performance. Such testing is not performed routinely, due to practical and safety concerns. AIM: Evaluate the feasibility, safety and acceptability of select outcome measures as part of a study protocol testing CYPwH; including myometry, 10 metre incremental shuttle walk test (10-m ISWT), iSTEP (an incremental step test, with data from TDB), and 1 week of accelerometry-wear at home. METHODS: Sixty-six boys aged 6-15 years with mild, moderate or severe haemophilia A or B (including inhibitors) attending routine clinics at Great Ormond Street Hospital were approached to participate. Descriptive statistics and content analysis were used to assess outcomes of feasibility, safety and acceptability, which included recruitment/retention rates, protocol completion within routine appointment timeframes, performance testing without serious adverse events/reactions (SAE/SARs), and acceptability to CYPwH of high-level performance measures. RESULTS: Outcomes were met: 43 boys completed testing at clinic review (Jan-Nov 2018) within a 10-month timeframe, retention was 95% at completion of protocol and no SAE/SARs were reported throughout testing. CONCLUSION: Feasibility, safety and acceptability of the study protocol have been established in this population. Both high-level performance tests, iSTEP and 10-m ISWT, were an acceptable addition to boys' routine clinic appointments and could be safe, acceptable choices of outcome measure as part of a core set of tests for CYPwH. Further investigation of the psychometric properties for the iSTEP is now justified, in order for it to be used as a standardised, validated, reliable outcome measure in clinical or research settings. TRIAL REGISTRATION: Retrospectively registered on September 3, 2019, on ClinicalTrials.gov (ID: NCT04076306 ).

Early intervention programmes for infants at high risk of atypical neurodevelopmental outcome.

The purpose of this review is to present a new framework, EI SMART (early intervention: sensorimotor development, attention and regulation, relationships, and therapist support) for identifying key components that could contribute to more effective interventions for infants at high risk of atypical neurodevelopmental outcome. We present a clinical consensus of current challenges and themes in early intervention, based on multidisciplinary group discussions, including parents of high-risk infants, supported by a literature review. Components to include in early intervention programmes are: (1) promotion of self-initiated, developmentally appropriate motor activity; (2) supporting infant self-regulation and the development of positive parent-infant relationships; and (3) promotion of early communication skills, parent coaching, responsive parenting, and supporting parental mental well-being. Such multimodal programmes may need to be evaluated as a package. WHAT THIS PAPER ADDS: Early intervention programmes should address sensorimotor development, attention, self-regulation, and early communication skills. Therapist input to the programme should include parent coaching and support for parental mental well-being.

PROGRAMAS DE INTERVENCIÓN TEMPRANA PARA LACTANTES CON ALTO RIESGO DE TRATARNOS DEL DESARROLLO NEUROLÓGICO: El propósito de esta revisión es presentar un nuevo marco, EI SMART (intervención temprana: desarrollo sensoriomotor, atención y regulación, relaciones y apoyo del terapeuta) para identificar componentes clave que podrían contribuir a intervenciones más efectivas para los bebés con alto riesgo de desarrollar un trastorno del neurodesarrollo. Presentamos un consenso clínico de los desafíos y temas actuales en la intervención temprana, basados ​​en discusiones grupales multidisciplinares, incluidos los padres de bebés con alto riesgo, respaldados por una revisión de la literatura. Los componentes para incluir en los programas de intervención temprana son (1) la promoción de actividades motoras autoiniciadas y apropiadas para el desarrollo; (2) apoyar la autorregulación infantil y el desarrollo de relaciones positivas entre padres e infantes; (3) promoción de las habilidades de comunicación temprana, entrenamiento de padres, crianza responsable y apoyo al bienestar mental de los padres. Es posible que dichos programas multimodales deban evaluarse como un paquete terapéutico.

PROGRAMAS DE INTERVENÇÃO PRECOCE PARA CRIANÇAS EM ALTO RISCO DE RESULTADO ANORMAL DO DESENVOLVIMENTO: O propósito desta revisão é apresentar um novo formato: EI SMART (intervenção precoce: desenvolvimento sensóriomotor, atenção e regulação, relacionamentos, e apoio do terapeuta) para identificar componentes centrais que podem contribuir para intervenções mais efetivas em lactentes de alto risco. Apresentamos um consenso clínico dos desafios correntes e temas em intervenção precoce, com base em discussões interdisciplinares, incluindo pais de lactentes de alto risco, com apoio de uma revisão de literatura. Os componentes a serem incluídos em programas de intervenção precoce são 1) promoção de atividade motora auto-iniciada apropriada para o desenvolvimento; 2) suporte para a auto-regulação do lactente e desenvolvimento de relações pais-filhos positivas; 3) promoção de habilidades precoces de comunicação, suporte aos pais, parentalidade responsável, e suporte ao bem estar mental dos pais. Tais programas multimodais podem precisar ser avaliados em forma de um pacote.