A longitudinal, observational study of the features of transitional healthcare associated with better outcomes for young people with long-term conditions.

BACKGROUND: Most evidence about what works in transitional care comes from small studies in single clinical specialties. We tested the hypothesis that exposures to nine recommended features of transitional healthcare were associated with better outcomes for young people with long-term conditions during transition from child-centred to adult-oriented health services. METHODS: This is a longitudinal, observational cohort study in UK secondary care including 374 young people, aged 14-18.9 years at recruitment, with type 1 diabetes (n = 150), cerebral palsy (n = 106) or autism spectrum disorder with an associated mental health problem (n = 118). All were pre-transfer and without significant learning disability. We approached all young people attending five paediatric diabetes centres, all young people with autism spectrum disorder attending four mental health centres, and randomly selected young people from two population-based cerebral palsy registers. Participants received four home research visits, 1 year apart and 274 participants (73%) completed follow-up. Outcome measures were Warwick Edinburgh Mental Wellbeing Scale, Mind the Gap Scale (satisfaction with services), Rotterdam Transition Profile (Participation) and Autonomy in Appointments. RESULTS: Exposure to recommended features was 61% for 'coordinated team', 53% for 'age-banded clinic', 48% for 'holistic life-skills training', 42% for 'promotion of health self-efficacy', 40% for 'meeting the adult team before transfer', 34% for 'appropriate parent involvement' and less than 30% for 'written transition plan', 'key worker' and 'transition manager for clinical team'. Three features were strongly associated with improved outcomes. (1) 'Appropriate parent involvement', example association with Wellbeing (b = 4.5, 95% CI 2.0-7.0, p = 0.001); (2) 'Promotion of health self-efficacy', example association with Satisfaction with Services (b = - 0.5, 95% CI - 0.9 to - 0.2, p = 0.006); (3) 'Meeting the adult team before transfer', example associations with Participation (arranging services and aids) (odds ratio 5.2, 95% CI 2.1-12.8, p < 0.001) and with Autonomy in Appointments (average 1.7 points higher, 95% CI 0.8-2.6, p < 0.001). There was slightly less recruitment of participants from areas with greater socioeconomic deprivation, though not with respect to family composition. CONCLUSIONS: Three features of transitional care were associated with improved outcomes. Results are likely to be generalisable because participants had three very different conditions, attending services at many UK sites. Results are relevant for clinicians as well as for commissioners and managers of health services. The challenge of introducing these three features across child and adult healthcare services, and the effects of doing so, should be assessed.

A longitudinal, observational study examining the relationships of patient satisfaction with services and mental well-being to their clinical course in young people with Type 1 diabetes mellitus during transition from child to adult health services.

AIM: We hypothesized that participant well-being and satisfaction with services would be positively associated with a satisfactory clinical course during transition from child to adult health care. METHODS: Some 150 young people with Type 1 diabetes mellitus from five diabetes units in England were recruited to a longitudinal study of transition. Each young person was visited at home four times by a research assistant; each visit was 1 year apart. Satisfaction with services (Mind the Gap; MTG) and mental well-being (Warwick-Edinburgh Mental Well-being Scale; WEMWBS) were captured. Change in HbA1c , episodes of ketoacidosis, clinic and retinal screening attendance were used to assess clinical course. In total, 108 of 150 (72%) young people had sufficient data for analysis at visit 4. RESULTS: Mean age at entry was 16 years. By visit 4, 81.5% had left paediatric healthcare services. Median HbA1c increased significantly (P = 0.01) from 69 mmol/mol (8.5%) at baseline to 75 mmol/mol (9.0%) at visit 4. WEMWBS scores were comparable with those in the general population at baseline and were stable over the study period. MTG scores were also stable. By visit 4, some 32 individuals had a 'satisfactory' and 76 a 'suboptimal' clinical course. There were no significant differences in average WEMWBS and MTG scores between the clinical course groups (P = 0.96, 0.52 respectively); nor was there a significant difference in transfer status between the clinical course groups. CONCLUSIONS: The well-being of young people with diabetes and their satisfaction with transition services are not closely related to their clinical course. Investigating whether innovative psycho-educational interventions can improve the clinical course is a research priority.

How well do services for young people with long term conditions deliver features proposed to improve transition?

BACKGROUND: For young people with long-term conditions, transition from child to adult-oriented health services is a critical period which, if not managed well, may lead to poor outcomes. There are features of transition services which guidance and research suggest improve outcomes. We studied nine such features, calling them 'proposed beneficial features': age-banded clinic; meet adult team before transfer; promotion of health self-efficacy; written transition plan; appropriate parent involvement; key worker; coordinated team; holistic life-skills training; transition manager for clinical team. We aimed to describe the extent to which service providers offer these nine features, and to compare this with young people's reported experience of them. METHODS: A longitudinal, mixed methods study followed 374 young people as their care moved from child to adult health services. Participants had type 1 diabetes, cerebral palsy or autism spectrum disorder with additional mental health difficulties. Data are reported from the first two visits, one year apart. RESULTS: Three hundred four (81.3%) of the young people took part in the second visit (128 with diabetes, 91 with autism, 85 with cerebral palsy). Overall, the nine proposed beneficial features of transition services were poorly provided. Fewer than half of services stated they provided an age-banded clinic, written transition plan, transition manager for clinical team, a protocol for promotion of health self-efficacy, or holistic life-skills training. To varying degrees, young people reported that they had not experienced the features which services said they provided. For instance, the agreement for written transition plan, holistic life-skills training and key worker, was 30, 43 and 49% respectively. Agreement was better for appropriate parent involvement, age-banded clinic, promotion of health self-efficacy and coordinated team at 77, 77, 80 and 69% respectively. Variation in the meaning of the features as experienced by young people and families was evident from qualitative interviews and observations. CONCLUSIONS: UK services provide only some of the nine proposed beneficial features for supporting healthcare transition of young people with long term conditions. Observational studies or trials which examine the influence of features of transition services on outcomes should ensure that the experiences of young people and families are captured, and not rely on service specifications.

Are the health needs of young people with cerebral palsy met during transition from child to adult health care?

BACKGROUND: The transition from child to adult health care is a particular challenge for young people with cerebral palsy, who have a range of needs. The measurement of reported needs, and in particular unmet needs, is one means to assess the effectiveness of services. METHODS: We recruited 106 young people with cerebral palsy, before transfer from child services, along with their parents to a 3-year longitudinal study. Reported needs were measured with an 11-item questionnaire covering speech, mobility, positioning, equipment, pain, epilepsy, weight, control of movement, bone or joint problems, curvature of the back, and eyesight. Categorical principal component analysis was used to create factor scores for bivariate and regression analyses. RESULTS: A high level of reported needs was identified particularly for control of movement, mobility, and equipment, but these areas were generally being addressed by services. The highest areas of unmet needs were for management of pain, bone or joint problems, and speech. Analysis of unmet needs yielded two factor scores, daily living health care and medical care. Unmet needs in daily living health care were related to severity of motor impairment and to attending nonspecialist education. Unmet needs tended to increase over time but were not significantly (p > .05) related to whether the young person had transferred from child services. CONCLUSIONS: Reporting of unmet needs can indicate where service development is required, and we have shown that the approach to measurement can be improved. As the number of unmet health needs at the start of transition is considerable, unmet health needs after transition cannot all be attributed to poor transitional health care. The range and continuation of needs of young people with cerebral palsy argue for close liaison between adult services and child services and creation of models of practice to improve coordination.

Understanding frames: A UK survey of parents and professionals regarding the use of standing frames for children with cerebral palsy.

BACKGROUND: Standing frames are used for children with cerebral palsy (CP). They may improve body structure and function (e.g., reducing risk of hip subluxation, and improving bladder and bowel function), improving activity (e.g., motor abilities) and participation (e.g., interaction with peers), but there is little evidence that they do. We aimed to identify current UK standing frame practice for children with CP and to understand stakeholder views regarding their clinical benefits and challenges to use. METHOD: Three populations were sampled: clinicians prescribing standing frames for children with CP (n = 305), professionals (health and education) working with children with CP who use standing frames (n = 155), and parents of children with CP who have used standing frames (n = 91). Questionnaires were developed by the co-applicant group and piloted with other professionals and parents of children with CP. They were distributed online via clinical and parent networks across the UK. RESULTS: Prescribing practice was consistent, but achieving the prescribed use was not always possible. Respondents in all groups reported the perceived benefits of frames, which include many domains of the International Classification of Functioning Disability and Health for Children and Youth. Challenges of use are related to physical space and child-reported pain. CONCLUSIONS: These survey findings provide information from key stakeholders regarding current UK standing frame practice.

Understanding frames: A qualitative study of young people's experiences of using standing frames as part of postural management for cerebral palsy.

BACKGROUND: Consensus opinion supports standing frame use as part of postural management for nonambulant young people with cerebral palsy. Although the rationale for standing frame use and the associated challenges have been described, little attention has been given to the users' experiences. The aim of the current study was to explore young people's positive and negative experiences, and attitudes regarding standing frame use. METHODS: Framework analysis informed an open exploration of young people's opinions of standing frames. Using semistructured interviews, 12 young people with cerebral palsy (6 female) were interviewed, providing the data set for transcription and thematic analysis. FINDINGS: The first theme "attitudes to standing frames" describes the young people's understanding of why they use standing frames. Although standing frames can be painful, some young people believe they should be endured to improve their body structure and function. There were mixed views about the impact standing frames have socially, with some young people feeling excluded from their peers, and others feeling as though standing frames helped them "fit in." Some young people are not offered a choice about how and when they use their standing frame. The second theme "challenges of standing frame use" highlights the issues with standing frame use such as manual handling, interference from siblings, and the lack of aesthetically pleasing standing frame designs. CONCLUSIONS: Young people report benefits related to choice, pain relief, and participation but can also cause pain, discomfort, and reduced independence and participation. Healthcare professionals should have open, informative conversations about potential benefits and challenges of standing frames on all aspects of the young people's lives, including participation and activity.

The Questionnaire of Young People's Participation (QYPP): a new measure of participation frequency for disabled young people.

INTRODUCTION: The concept of participation was introduced by the World Health Organisation in the International Classification of Functioning, Disability and Health, 2001. Instruments to measure participation have been developed for children and adults, but none specifically for adolescents. Adolescence is a life stage with distinct patterns of participation, and previous research has shown that young people with disabilities have poorer participation than the general population. The aim of this study was to develop a measure of participation frequency, covering all major domains, for young people with cerebral palsy (CP). CP was chosen as an exemplar because it is a significant cause of disability in young people, with affected individuals experiencing a range of different impairments of varying severity. METHODS: A pool of 88 items was developed using the published literature, existing measures and qualitative data from young people. The item pool was revised following expert review by 17 experts. Cognitive interviews on the items were carried out with 12 young people and 12 carers; field-testing was then undertaken with 107 young people with CP aged 13-21 years, and 540 young people from the general population to enable item reduction and to examine reliability and construct validity. RESULTS: The content review resulted in a 92-item draft questionnaire, content validity index of 93%. Cognitive interviews led to further wording changes. Following field-testing, the questionnaire was shortened to 45 items. Known-groups validity was demonstrated by correlation with impairment severity. Test-retest reliability was satisfactory for all domains. Internal consistency varied between domains. CONCLUSION: This is the first instrument developed specifically to measure frequency of participation across multiple domains for young people with disability. Use of the questionnaire in research and clinical work will enable its properties to be better understood and its generalizability to wider groups to be clearer.

Pain in children with cerebral palsy: a cross-sectional multicentre European study.

AIM: To determine the prevalence and associations of self-reported and parent-reported pain in children with cerebral palsy (CP) of all severities. METHOD: Cross-sectional design using a questionnaire; analysis using ordinal regression. Children aged 8-12 years were randomly selected from population-based registers of children with CP in eight European regions; a further region recruited 75 children from multiple sources. Outcome measures were pain in the previous week among children who could self-report and parents' perception of their child's pain in the previous 4 weeks. RESULTS: Data on pain were available from 490 children who could self-report and parents of 806 children (those who could and could not self-report). The estimated population prevalence of self-reported pain in the previous week was 60% (95% CI: 54-65%) and that of parent-reported pain in the previous 4 weeks was 73% (95% CI: 69-76%). In self-reporting children, older children reported more pain but pain was not significantly associated with severity of impairment. In parent reports, severity of child impairment, seizures and parental unemployment were associated with more frequent and severe pain. CONCLUSION: Pain in children with CP is common. Clinicians should enquire about pain and consider appropriate physical, therapeutic or psychological management.

Psychological problems in children with hemiplegia: a European multicentre survey.

OBJECTIVE: To describe the prevalence and determinants of psychological problems in European children with hemiplegia. DESIGN: Cross-sectional survey. SETTING: Home visits in nine European regions by research associates who administered standard questionnaires to parents. PATIENTS: 279 children with hemiplegia aged 8-12 years were recruited from population-based case registers. OUTCOME MEASURE: Strengths and Difficulties Questionnaire comprising emotion, conduct, hyperactivity, peer problems and prosocial domains. An "impact score" (IS) measures the social and psychological impact of the child's difficulties. RESULTS: Children with hemiplegia had higher mean scores on the total difficulties score (TDS) compared with a normative sample (p<0.001). 48% and 57% of children, respectively, had borderline-abnormal TDS and IS. Significant, independent associations were observed between intellectual impairment and an increased risk for hyperactivity (odds ratio; OR 8.4, 95% CI 3.4 to 20.8), peer problems (OR 3.1, 95% CI 1.7 to 5.5), psychological and social impact (OR 3.0, 95% CI 1.6 to 5.6) when children with an intellectual quotient (IQ) <50 were compared with those with an IQ >70. Boys had an increased risk for conduct (OR 2.1, 95% CI 1.2 to 3.7) and hyperactivity disorders (OR 2.5, 95% CI 1.4 to 4.6). Poor self-esteem was associated with an increased risk for peer problems (OR 5.8, 95% CI 2.5 to 13.4) and poor prosocial skills (OR 7.5, 95% CI 2.4 to 23.2) compared with those with high self-esteem. Other determinants of psychological adjustment were impaired communication, severe pain and living with a single parent. CONCLUSIONS: Many of the psychological problems identified are amenable to treatment. Special attention should be given to those at highest risk of developing psychological difficulties.

Dasl(n)e: the challenge of developing a regional database for autism spectrum disorder.

The setting up of a database of children with autism spectrum disorder (ASD) in the north east of England is described. Best practice has been followed and included involving parents in planning and implementation at all stages, oversight by a multi-agency group, management by a multidisciplinary steering group, and independent administration of the database. From a potential listing of 986 children with ASD aged 3-12 years, the parents of 511 have so far responded (51.8%), although response rate varies considerably by local authority. Data checking has shown the information to be valid and case ascertainment broadly representative. The uses to which the data are being put and the continuing challenges are outlined.

Health status of children with cerebral palsy living in Europe: a multi-centre study.

AIM: The aim of this report is to describe the health status of 8-12-year-old children with cerebral palsy (CP) of all severities in Europe using the Child Health Questionnaire (CHQ). METHOD: A total of 818 children with CP from nine centres in defined geographical areas participated. CP type, gross and fine motor function, additional impairments were classified and family data were obtained. The CHQ was used to measure the parent's perception of their child's physical (PHY) and psychosocial (PSY) health. RESULTS: PHY scores were lower than the reference samples with a median of 46. The severity of gross motor function influenced the CHQ scores significantly in the PHY scale with the lowest scores for children with least gross motor function. There were significant differences between the CP types in PHY with the higher scores for children with unilateral spastic and the lowest scores for children with bilateral spastic and dyskinetic CP type. Fine motor function severity significantly affected both the PHY and PSY scales. The severity of intellectual impairment was significantly associated with CHQ scores in most dimensions with higher scores for higher IQ level in PHY and PSY. Children with seizures during the last year had a significantly lower health compared with children without seizures. The results of the multivariate regression analyses (forward stepwise regression) of CHQ scores on CP subtype, gross and fine motor function, cognitive function, additional impairments, seizures, parental education and employment revealed gross motor function, cognitive level and type of school attended were significant prognostic factors. CONCLUSION: This report is based on the largest sample to date of children with CP. Health status as measured using the CHQ was affected in all children and was highly variable. Gross motor function level correlates with health from the PHY well-being perspective but the PSY and emotional aspects do not appear to follow the same pattern.

A qualitative study of the physical, social and attitudinal environments influencing the participation of children with cerebral palsy in northeast England.

PURPOSE: The social model of disability considers participation to be determined by the social, attitudinal and physical environments experienced by an individual. This study aims to ascertain from families of children with cerebral palsy the features of such environments which facilitate or restrict participation. METHOD: Thirteen in-depth interviews using a topic guide were conducted with the parents of children with cerebral palsy. Interviews were tape-recorded, transcribed and analysed with NVivo software. RESULTS: The main themes emerging from the interviews were the importance of mobility, transport, support by and to parents and attitudes of individuals and institutions towards children. Most parents did not raise the policies and legislation determining participation barriers, although these are also likely to be influential. CONCLUSIONS: This study confirms the importance of the environment for the participation of children with cerebral palsy. Statutory agencies need to attend the attitudes and policies in their organization in order to plan the inclusive environments which parents report will facilitate their child's participation. This study also contributes to the development of a tool to quantify the environment to allow the development of models to determine the environments which maximize children's participation.

Participation of disabled children: how should it be characterised and measured?

PURPOSE: The aim of the paper is to explore the issues involved in measuring children's participation. METHOD: The concept of participation as encapsulated in the International Classification of Functioning, Disability and Health (ICF) is discussed as it applies to children. The essential components of any measure of children's participation are outlined, including participation essential for normal development and survival, leisure activities, and educational participation. Some existing instruments are briefly reviewed in terms of their coverage of the essential components and the adequacy of their approach to measurement. RESULTS: Key issues regarding the content of an adequate measure of participation include the need to consider the child's dependency on the family, and their changing abilities and autonomy as they grow older. Instruments may be most appropriate where they ask the child directly, implying use of visual as well as verbal presentation. Their focus should be on 'performance' such as whether and how often an activity is taken part in, and not incorporate degree of assistance within the measurement scaling. CONCLUSIONS: Currently available measures of children's participation all have some limitations in terms of their applicability across impairment groupings, whether the child can directly respond, and in the ICF components covered. The feasibility of developing measurement instruments of children's participation at different ages is discussed.

Discussion groups with parents of children with cerebral palsy in Europe designed to assist development of a relevant measure of environment.

BACKGROUND: An instrument to measure environmental factors relevant to physically impaired children is being developed in a European context. Preliminary work in England had identified some potentially important themes. Further inquiry was needed to identify issues important in other European countries. OBJECTIVE: To inform the content of a questionnaire relevant to the environment of children with cerebral palsy (CP) living in Europe. DESIGN: A qualitative study using discussion groups. PARTICIPANTS: Parents of 28 children with CP from five countries; Denmark, France, Italy, Ireland and Sweden. One discussion group was held in each country with an average of seven parents per group. RESULTS: The four themes identified in the preliminary work done in England were strongly confirmed across Europe - namely: Mobility, Transport, Support by and to parents, and Attitudes of individuals and institutions towards children. Two new themes identified in the discussion groups were Bureaucracy and Access to information about rights and entitlements. CONCLUSIONS: The environmental factors that cause concern to parents of children with CP are similar across Europe. A prototype environmental questionnaire has been developed based on these findings. The environmental questionnaire is in use in a study in nine European centres.

How might districts identify local barriers to participation for children with cerebral palsy?

OBJECTIVES: To explore how data about participation and the local environment might be used to identify barriers to participation for children with cerebral palsy. METHODS: Participation is measured at 5 years of age using the six domains of the lifestyle assessment questionnaire. Individual child score profiles are compared with expected patterns from similar children and with the use of relevant services. Average scores for districts are compared with selected local authority performance indicators. Self-completion questionnaires are used to solicit information on barriers from families of children. RESULTS: Individual children who have poorer-than-expected participation scores and those who make less-than-expected use of relevant services can be identified. Children who live in districts with more facilities for the disabled appear to have better social participation scores. Common local barriers as well as specific access problems for individuals are revealed by simple questionnaires to families. Attitudes of strangers and staff in public places, lack of suitable transport and the presence of stairs are important barriers in many settings. CONCLUSIONS: Environmental barriers for children with cerebral palsy can be identified and related to their participation.

A qualitative study, using focused interviews, of the information needs of families whose children's names are on a cerebral palsy register.

BACKGROUND: We were concerned that information held on a register of children with cerebral palsy was not being made available to contributing families, and that the existence and purpose of the register was not sufficiently understood. METHODS: Focused interviews with 13 families selected by purposive sampling. RESULTS: Many parents would like more information about the data on registers which include their child. They would also like to be more involved in the design of the register and its reports, but there are practical difficulties in making time available for such activity. Although parents were surprised that they had not already been given more information about the existence and purpose of the register, none were resentful that their child's name was on the register once they were reassured about confidentiality and database security. Parents also had general concerns unrelated to the register which were about their need to be treated as equals by professionals, to have more information about equipment and to have earlier and clearer diagnoses. CONCLUSIONS: Registers of children with uncommon conditions have well-established roles in epidemiology, planning and research. By fully involving parents in ways suggested by this study, registers can also empower parents. The study should also provide reassurance to those who hold such registers without explicit consent that the requirement to now obtain consent should not create resentment or jeopardize completeness.