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.

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.

Mothers' perceptions of child weight status and the subsequent weight gain of their children: a population-based longitudinal study.

BACKGROUND: There is a plethora of cross-sectional work on maternal perceptions of child weight status showing that mothers typically do not classify their overweight child as being overweight according to commonly used clinical criteria. Awareness of overweight in their child is regarded as an important prerequisite for mothers to initiate appropriate action. The gap in the literature is determining whether, if mothers do classify their overweight child's weight status correctly, this is associated with a positive outcome for the child's body mass index (BMI) at a later stage. OBJECTIVE: To explore longitudinal perceptions of child weight status from mothers of a contemporary population-based birth cohort (Gateshead Millennium Study) and relationships of these perceptions with future child weight gain. METHODS: Data collected in the same cohort at 7, 12 and 15 years of age: mothers' responses to two items concerning their child's body size; child's and mother's BMI; pubertal maturation; demographic information. RESULTS: Mothers' perceptions of whether their child was overweight did not change markedly over time. Child BMI was the only significant predictor of mothers' classification of overweight status, and it was only at the extreme end of the overweight range and in the obese range that mothers reliably described their child as overweight. Even when mothers did appropriately classify their child as overweight at an earlier stage, this was not related to relatively lower child BMI a few years later. CONCLUSIONS: Mothers tend to classify their child as overweight in only more extreme cases. It is an important finding that no beneficial impact was shown on later child BMI in overweight children whose mothers classified their child's weight status as overweight at an earlier stage.

Incidence, characteristics and course of narrow phenotype paediatric bipolar I disorder in the British Isles.

OBJECTIVE: To estimate the surveillance incidence of first-time diagnosis of narrow phenotype bipolar I disorder (NPBDI) in young people under 16 years by consultants in child and adolescent psychiatry (CCAP) in the British Isles and describe symptoms, comorbidity, associated factors, management strategies and clinical outcomes at 1-year follow-up. METHOD: Active prospective surveillance epidemiology was utilised to ask 730 CCAP to report cases of NPBDI using the child and adolescent psychiatry surveillance system. RESULTS: Of the 151 cases of NPBDI reported, 33 (age range 10-15.11 years) met the DSM-IV analytical case definition with 60% having had previously undiagnosed mood episodes. The minimum 12-month incidence of NPBDI in the British Isles was 0.59/100 000 (95% CI 0.41-0.84). Irritability was reported in 72% cases and comorbid conditions in 51.5% cases with 48.5% cases requiring admission to hospital. Relapses occurred in 56.67% cases during the 1-year follow-up. CONCLUSIONS: These rates suggest that the first-time diagnosis of NPBDI in young people <16 years of age by CCAP in the British Isles is infrequent; however, the rates of relapse and admission to hospital warrant close monitoring.

Emotional impact of genetic trials in progressive paediatric disorders: a dose-ranging exon-skipping trial in Duchenne muscular dystrophy.

BACKGROUND: Gene-modifying trials offer hope for improvement in chronic paediatric disorders, but they may also lead to disappointment and have an adverse emotional effect on families. This study aimed to examine emotional impact on participants in a paediatric exon-skipping trial. METHODS: Nineteen male children with Duchenne muscular dystrophy (DMD), and their parents, taking part in a dose-ranging study of an i.v. administered morpholino splice-switching oligomer (which can restore the reading frame in DMD and induce dystrophin expression) underwent a psychosocial/psychiatric examination at trial entry. Emotional impact was assessed at trial completion using questionnaires. RESULTS: The mean child age was 8.9 years (SD 2.1); 13(68%) were attending mainstream school. Most families were well adjusted psychosocially at trial entry. Post-trial median child emotional impact scores were 5/10 (n= 18), but impact was rated as positive by 6/14 (42%), neutral/mixed by 5 (35%) and negative by 3 (21%). Median post-trial psychosocial/psychiatric change scores in children and parents were minimal. Actual post-trial negative impact was statistically significantly associated with higher expected impact at trial entry, at which time the families of the three children displaying actual negative impact reported higher family stress levels in combination with a variety of other psychosocial risks factors. CONCLUSIONS: In carefully selected families with low levels of psychosocial stress/distress at trial entry, and with good support from paediatric research units (including psychiatric input when required), genetic trials in progressive disorders such as DMD can have a predominantly positive or neutral emotional impact. Nevertheless, negative impact is reported by a minority of families and possible psychosocial predictors deserving further scrutiny have been identified.

Models of transitional care for young people with complex health needs: a scoping review.

BACKGROUND: Young people with complex healthcare needs (CHNs) face the challenge of transferring from child to adult health services. This study sought to identify successful models of transitional care for young people with CHNs. Three conditions were used as exemplars: cerebral palsy, autism spectrum disorders and diabetes. METHODS: Scoping review: using search terms concerning transitional care, four databases were systematically searched for papers published in English between 1980 and April 2010. Additional informal search methods included recommendations from colleagues working with young people with each of the three conditions and making contact with clinical and research teams with expertise in transitional care. Inclusion and exclusion criteria were applied to define the papers selected for review. A separate review of policy documents, adolescent health and transition literature was also undertaken; 10 common summary categories for the components of high-quality services were identified. All papers were coded using a framework analysis which evaluated the data in two ways using the 10 transition categories and four elements of Normalization Process Theory that are important for successful implementation and integration of healthcare interventions. RESULTS: Nineteen papers were selected for review. A very limited literature of models of service provision was identified for young people with cerebral palsy and diabetes. No models were identified for young people with autism spectrum disorders. Furthermore most publications were either descriptions of new service provision or time-limited pilot studies with little service evaluation or consideration of key elements of effective implementation. CONCLUSIONS: Despite agreement about the importance of effective transitional care, there is a paucity of evidence to inform best practice about both the process of and what constitutes effective transitional care. There is therefore an urgent need for research to evaluate current transitional care practices for young people with CHNs.

FAB: First UK feasibility trial of a future randomised controlled trial of Family focused treatment for Adolescents with Bipolar disorder.

BACKGROUND: This first mixed-methods UK trial examined the feasibility and acceptability of a future definitive randomised controlled trial (RCT) to evaluate whether Family Focussed Treatment for Adolescents with Bipolar Disorder (FFT-A) UK version can improve family functioning and well-being as part of the management of Paediatric Bipolar Disorder (PBD). METHOD: The trial used a randomised, parallel group, non-blinded design where participants received FFT-A UK (16 sessions over 6 months) immediately or after 12 months (delayed arm). Measures of family functioning, well-being and quality of life of the young person and the main carer (most commonly a parent) were completed at baseline, 6 and 12-months in both arms. Primary outcome measures included rates of eligibility, consent and retention along with estimates of variability in the measures and assessment of the intervention delivery. Qualitative interviews allowed assessment of participants' views about FFT-A and the trial processes. RESULTS: Twenty-seven of 36 young persons with PBD and their families consented; of these, 14 families were randomised to the immediate and 13 to the delayed arm. Two families from the immediate arm withdrew consent and discontinued participation. Quantitative measures were completed by 22 families (88%) at 6-months and 21 families (84%) at 12-months. Qualitative interviews were conducted with 30 participants (9 young people, 15 parents and 6 other family members). Nine families attended 3 post-trial focus groups. CONCLUSION: It was feasible to recruit and retain to this trial. The results highlighted that trial design and measures were acceptable to participants. A benefit in family relationships was reported by participants which they attributed to the intervention in qualitative interviews. Families recommended that future modifications include definitive trial(s) recruiting participants in the age range 15-25 years as it felt this was the age range with maximum need. Trial registration ISRCTN, ISRCTN59769322. Registered 20 January 2014, http://www.isrctn.com/ISRCTN59769322.

Autism Diagnostic Interview-Revised: a revised version of a diagnostic interview for caregivers of individuals with possible pervasive developmental disorders.

Describes the Autism Diagnostic Interview-Revised (ADI-R), a revision of the Autism Diagnostic Interview, a semistructured, investigator-based interview for caregivers of children and adults for whom autism or pervasive developmental disorders is a possible diagnosis. The revised interview has been reorganized, shortened, modified to be appropriate for children with mental ages from about 18 months into adulthood and linked to ICD-10 and DSM-IV criteria. Psychometric data are presented for a sample of preschool children.

Infantile autism and urinary excretion of peptides and protein-associated peptide complexes.

Sixty-nine early morning urine samples obtained from three groups of young adult males (autistic, mentally handicapped, and a group of men of normal intelligence) were analyzed using the methods described by Trygstad et al. (1980). No consistent patterns of urinary chromatographic profiles were identified. A number of possible contributing factors are discussed in relation to this failure to replicate the results of previous studies.

Autism: the phenotype in relatives.

There is broad agreement that genetic influences are central in the development of idiopathic autism. Whether relatives manifest genetically related milder phenotypes, and if so how these relate to autism proper, has proved a more contentious issue. A review of the relevant studies indicates that relatives are sometimes affected by difficulties that appear conceptually related to autistic behaviors. These range in severity from pervasive developmental disorders to abnormalities in only one area of functioning, and possibly extend to related personality traits. Issues involved in clarifying the components of milder phenotypes and their relationship to autism are outlined.

Autism diagnostic interview: a standardized investigator-based instrument.

The development of a new standardized investigator-based interview for use in the differential diagnosis of pervasive developmental disorders is described, together with a diagnostic algorithm (using ICD-10 criteria) based on its use. Good interrater reliability for algorithm items was shown between four raters, two in Canada and two in the UK, who rated 32 videotaped interviews. The items also significantly discriminated between 16 autistic and 16 nonautistic mentally handicapped subjects. The algorithm based on ICD-10 identified all 16 autistic individuals and none of the 16 nonautistic subjects.

New interview and observation measures of the broader autism phenotype: impressions of interviewee measure.

A 20 item observational measure of social functioning, the Impression of Interviewee rating scale, is one of three measures devised to assess the broader autism phenotype. The sample studied included families containing at least two individuals with autism spectrum disorder; observations were undertaken by the researcher who interviewed the subject. An exploratory factor analysis suggested a single factor was most appropriate (Cronbach's α of 0.78). There was a modest but significant retest correlation of 0.42. Correlations between live ratings and blind consensus ratings of vignettes were high (0.93). Correlations with the interview measures were moderate but statistically significant. In conclusion, the observational scale provides a promising start but further work is required before general use can be recommended.

Autism spectrum disorder diagnostic assessments: improvements since publication of the National Autism Plan for Children.

OBJECTIVES: To assess in the context of a publically funded healthcare system, change in UK autism spectrum disorder (ASD) clinical diagnostic practice following the recommendations of the National Autism Plan for Children (NAP-C 2003). METHODS: In 2007, a questionnaire based on standards from the NAP-C was sent to UK child development teams (CDTs); results were compared with 2001 data from the National Initiative for Autism Screening and Assessment. MAIN FINDINGS: Responses were received from 149 of 243 UK CDTs (61%). Most teams used standardised autism diagnostic assessments. There was greater access to members of the multidisciplinary team than in 2001. Only one-third of teams had a defined timescale for completion of assessment; of those teams, about half met the recommended NAP-C target. CONCLUSIONS: Since 2001, there has been an improvement in diagnostic services for children with ASD, however, inequalities remain. Providers should continue to improve services in order to deliver timely and comprehensive assessments for children with ASD.

Spectrum of Feeding Problems and Gastrointestinal Symptoms in Children with Autism Spectrum Disorders: A Scoping Review

Introduction: Managing feeding problems and gastrointestinal (GI) symptoms in children with Autism Spectrum Disorders (ASD) is a challenging process for professionals and parents especially if they become persistent and longstanding. The aim of the study was to assess the evidence on feeding problems and GI symptoms among primary school children with ASD. Methods: A scoping review using electronic journal databases, published reports and other types of publication in the last 10 years was conducted. Key terms were defined in the searches and a scoping review framework was used to chart the evidence on feeding problems and GI symptoms in children with ASD. Eighty three articles met the inclusion criteria and 50 articles were used in the review. Results: There is emerging literature reporting consistently on a wide spectrum of feeding problems and GI symptoms among children with ASD. In addition, there is little published literature reporting or investigating the impact of these problems in children with ASD which include financial, social and stress impact on parents, children and their families. This review indicates that many school-aged children with ASD can experience several types of feeding problems and GI symptoms. Conclusion: It is important for professionals working in the community or public health, and educational settings to be able to identify these issues at an early stage, so that professionals could support parents with appropriate information and advice. A specific tool is needed to assess feeding problems and GI symptoms in children with ASD.

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.

Families in a state of flux: the experience of grandparents in autism spectrum disorder.

BACKGROUND: The experience of being a grandparent of a grandchild with autism spectrum disorder (ASD) is a previously under-researched area. This study sets out to examine the grandparents' own perspective in an exploratory way using a qualitative approach to answer the question, 'What is the experience like?' METHOD: A qualitative research project, using a purposive sampling technique and semi-structured interviews to examine the experiences of six grandparents of children with ASD diagnosed by a specialist team in a second opinion Tier 4 Child and Adolescent Mental Health Service. RESULTS: The experiences of the grandparents were characterized by three Key Themes which emerged from the interviews: (1) The Parental Bond (protective bonding towards grandchild and adult child); (2) Striving for Answers (searching for meaning); (3) Keeping Intact (holding the family together). CONCLUSION: The study suggests some interesting insights and confirms the need for more attention to this area. A key question raised by the study is how a child-focused multidisciplinary team can embrace its role in delivering a family-centred service. The clinical implications of this project have led to a change of practice in the specialist team. Further research would be appropriate to investigate the therapeutic effectiveness and cost effectiveness of involving grandparents as part of the assessment process.

Autism: current understanding and management.

Autism is now generally considered to be an organic neurodevelopmental disorder. This review considers the current understanding of genetic factors and associated medical conditions, together with a discussion of some aspects of management and treatment of affected children and adolescents.

Selective subcortical abnormalities in autism.

Nine physically healthy, adult autistic men, with normal or near normal intelligence, and 13 healthy male controls were examined in a CT brain scan study. CT scans were analysed with a fully automated computer-assisted program, and regional brain radiodensities were measured with careful attention to artefacts. Autistic patients revealed significantly larger third, but not lateral, ventricular size and significantly lower mean caudate, but equivalent mean frontal and thalamic, radiodensities compared to controls. The sizes of the Sylvian fissures and interhemispheric fissure were equivalent between groups. The findings are consistent with selective subcortical abnormalities in autism.