PPIE in a technical research study: Using public involvement to refine the concept and understanding and move towards a multidimensional concept of disability.

BACKGROUND: Disability is often an essentialised and oversimplified concept. We propose refining this while incorporating the multidimensional nature of disability by increasing the use of existing survey questions and their corresponding data to enrich, broaden and inform understandings of disability. METHODS: We combined patient and public involvement and engagement (PPIE) with focus groups and concept mapping to collaboratively map disability survey questions into conceptual models of disability with six members of the public with lived experiences of disability. RESULTS: Using reflexive thematic analysis, we identified three qualitative themes and eight subthemes through a series of four PPIE activities: (1) understanding concepts of disability based on individual experience, subthemes: 1.1-preference for the biopsychosocial model, 1.2-'Reviewing' instead of mapping survey questions and 1.3-comparing questions to real life; (2) consistency between understanding needs and implementing adjustments, subthemes: 2.1-connecting preparation and operation, 2.2-inclusivity and adjustments in activities and 2.3-feedback for improving activities and (3) real-world applications-targeted awareness raising, subthemes: 3.1-who, where, what and how to share activity findings and results, 3.2-sharing with human resource and equality, diversity and inclusion professionals. CONCLUSION: Members of the public who collaborated in these activities felt empowered, engaged and supported throughout this study. This approach offers a model for other researchers to cede power to the public over the research aspects typically reserved for researchers. PATIENT OR PUBLIC CONTRIBUTION: We involved members of the public with lived experience throughout this study-co-design, co-facilitation, collaboratively mapping the disability or disability-related survey questions into conceptual models of disability, evaluation of the activities, co-analysis and co-authorship.

The impact of formal care provision on informal care receipt for people over 75 in England.

In this study, we examine the relationship between formal care provision and informal care receipt from within the household for people over 75 years old using data from the British Household Panel Survey between 1991 and 2009. To address potential concerns about endogeneity of formal care we use a 'spatial lag' instrumental variable. We find a negative and statistically significant effect of formal care provision on informal care receipt from within the household, suggesting a substantial degree of substitutability between these two modes of care. These findings provide grounds for estimates of savings in the cost of informal care enabled by spending on formal care, which is important in light of the effects of the caregiving burden and associated responsibilities on carer's labour market participation.

Study protocol: evaluation of the 0-5 public health investment in England - a mixed-methods study integrating analyses of national linked administrative data with in-depth case studies.

INTRODUCTION: Health visiting is a long-established, nationally implemented programme that works with other services at a local level to improve the health and well-being of children and families. To maximise the impact and efficiency of the health visiting programme, policy-makers and commissioners need robust evidence on the costs and benefits of different levels and types of health visiting, for different families, in different local contexts. METHODS AND ANALYSIS: This mixed-methods study will analyse individual-level health visiting data for 2018/2019 and 2019/2020 linked with longitudinal data from children's social care, hospitals and schools to estimate the association of number and type of health visiting contacts with a range of children and maternal outcomes. We will also use aggregate local authority data to estimate the association between local models of health visiting and area-level outcomes. Outcomes will include hospitalisations, breast feeding, vaccination, childhood obesity and maternal mental health. Where possible, outcomes will be valued in monetary terms, and we will compare total costs to total benefits of different health visiting service delivery models. Qualitative case studies and extensive stakeholder input will help explain the quantitative analyses and interpret the results in the context of local policy, practice and circumstance. ETHICS AND DISSEMINATION: The University College London Research Ethics Committee approved this study (ref 20561/002). Results will be submitted for publication in a peer-reviewed journal and findings will be shared and debated with national policy-makers, commissioners and managers of health visiting services, health visitors and parents.

Job Quality and Job Separation of Direct Care Workers in England.

Background and Objectives: Most job leavers in the long-term care (LTC) sector in England do not leave the sector, but rather move to other LTC employers. Nevertheless, the high "churn" can have a negative impact on continuity and quality of care, care providers' recruitment and training costs, and the remaining staff workload and motivation. This study aimed to provide quantitative evidence on the drivers of direct care workers' job separation in England, with a focus on job quality. Research Design and Methods: We used yearly data (2016-19) from the Adult Social Care Workforce Data Set, the leading source of LTC workforce data in England, including information on both LTC workers and employers. The analysis considered panel data econometric methods that accounted for unobserved heterogeneity at worker and employer levels. Results: After controlling for observed individual, organizational, and local market characteristics as well as unobserved worker and employer heterogeneity, we found that everything else being equal, wages and employment conditions (i.e., full-time contracts and contracts with guaranteed working hours) significantly reduce job separation. For example, a 10% wage increase from the sample mean would reduce the job separation rate by about 3 percentage points. This wage effect was more than halved (i.e., downward biased) when not accounting for unobserved effects. Discussion and Implications: The persistent high staff turnover in LTC in England highlights the need for finding practical solutions faced by care providers and policy-makers. Our findings showed that improving pay and employment conditions can be the way forward while methodologically stressing the importance of accounting for unobserved variable bias.

Recruitment, retention and employment growth in the long-term care sector in England.

This paper studies the relationship between turnover, hiring and employment growth in the long-term care (LTC) sector in England and sheds light on how challenges in both recruitment and retention affect the sector's ability to meet growing demand for care services. Using the Adult Social Care Workforce Data Set (ASC-WDS), a large longitudinal dataset of LTC establishments in England, and fixed effects estimation methods we: (a) quantify the relationship between the in/outflow of care workers and the expansion/contraction of employment within establishments, (b) establish the role of staff retention policy for workforce expansion, and (c) identify the role of recruitment frictions and its impact on hiring and employment contraction. Our analysis indicates that care worker turnover and employment growth are negatively related. A one percentage point increase in employment contraction is associated with a 0.71 percentage point rise in turnover, while a one percentage point increase in employment expansion is associated with a 0.23 percentage point fall in turnover. In contrast, we find that hiring rates and employment growth are positively related. A one percentage point increase in employment expansion is associated with a 0.76 percentage point rise in hiring, while a one percentage point increase in employment contraction is associated with a 0.26 percentage point decrease in hiring. We argue that the negative turnover-employment growth relationship within expanding establishments provides evidence that better staff retention is associated with higher employment growth. Using information on establishments' annual change in vacancies, and controlling for changes in new labor demand, we also find rising year-on-year vacancies amongst establishments with declining employment. This provides evidence that recruitment frictions drive the declining rate of replacement hiring amongst contracting establishments. Across sectors, we find that the employment growth-turnover and the employment decline-hiring relationships are relatively stronger in the private and voluntary sectors compared to the public sector, suggesting that the impact of staff retention and recruitment frictions on employment is more acute in these sectors.

Valuing informal carers' quality of life using best-worst scaling-Finnish preference weights for the Adult Social Care Outcomes Toolkit for carers (ASCOT-Carer).

This study developed Finnish preference weights for the seven-attribute Adult Social Care Outcomes Toolkit for carers (ASCOT-Carer) and investigated survey fatigue and learning in best-worst scaling (BWS) experiments. An online survey that included a BWS experiment using the ASCOT-Carer was completed by a sample from the general population in Finland. A block of eight BWS profiles describing different states from the ASCOT-Carer were randomly assigned to each respondent, who consecutively made four choices (best, worst, second best and second worst) per profile. The analysis panel data had 32,160 choices made by 1005 respondents. A scale multinomial logit (S-MNL) model was used to estimate preference weights for 28 ASCOT-Carer attribute levels. Fatigue and learning effects were examined as scale heterogeneity. Several specifications of the generalised MNL model were employed to ensure the stability of the preference estimates. The most and least-valued states were the top and bottom levels of the control over daily life attribute. The preference weights were not on a cardinal scale. We observed the position effect of the attributes on preferences associated with the best or second-best choices. A learning effect was found. The established preference weights can be used in evaluations of the effects of long-term care services and interventions on the quality of life of service users and caregivers. The learning effect implies a need to develop study designs that ensure equal consideration to all profiles (choice tasks) in a sequential choice experiment.

Do You Prefer Safety to Social Participation? Finnish Population-Based Preference Weights for the Adult Social Care Outcomes Toolkit (ASCOT) for Service Users.

Introduction. The Adult Social Care Outcomes Toolkit (ASCOT) was developed in England to measure people's social care-related quality of life (SCRQoL). Objectives. The aim of this article is to estimate preference weights for the Finnish ASCOT for service users (ASCOT). In addition, we tested for learning and fatigue effects in the choice experiment used to elicit the preference weights. Methods. The analysis data (n = 1000 individuals) were obtained from an online survey sample of the Finnish adult general population using gender, age, and region as quotas. The questionnaire included a best-worst scaling (BWS) experiment using ASCOT. Each respondent sequentially selected four alternatives (best, worst; second-best, second-worst) for eight BWS tasks (n = 32,000 choice observations). A scale multinomial logit model was used to estimate the preference parameters and to test for fatigue and learning. Results. The most and least preferred attribute-levels were "I have as much control over my daily life as I want" and "I have no control over my daily life." The preference weights were not on a cardinal scale. The ordering effect was related to the second-best choices. Learning effect was in the last four tasks. Conclusions. This study has developed a set of preference weights for the ASCOT instrument in Finland, which can be used for investigating outcomes of social care interventions on adult populations. The learning effect calls for the development of study designs that reduce possible bias relating to preference uncertainty at the beginning of sequential BWS tasks. It also supports the adaptation of a modelling strategy in which the sequence of tasks is explicitly modelled as a scale factor.

What's important when caring for a loved one? Population-based preference weights for the Adult Social Care Outcomes Toolkit for informal carers (ASCOT-Carer) for Austria.

PURPOSE: The Adult Social Care Outcomes Toolkit for informal carers (ASCOT-Carer) can be used to assess long-term care-related quality of life (LTC-QoL) of adult informal carers of persons using LTC services. The ASCOT-Carer instrument has been translated into several languages, but preference weights reflecting the relative importance of different outcome states are only available for England so far. In this paper, we estimated preference weights for the German version of the ASCOT-Carer for Austria and investigated the value people place on different QoL-outcome states. METHODS: We used data from a best-worst scaling (BWS) experiment and estimated a scale-adjusted multinomial logit (S-MNL) model to elicit preference weights for the ASCOT-Carer domain-levels. Data were collected using an online survey of the Austrian general population (n = 1001). RESULTS: Top levels in the domains of 'Space and time to be yourself', 'Occupation' and 'Control over daily life' were perceived as providing the highest utility, and states with high needs in the same domains seen as particularly undesirable. 'Personal safety' was the only domain where levels were roughly equidistant. In all other domains, the difference between the top two levels ('ideal state' and 'no needs') was very small. CONCLUSION: The paper provides preference weights for the German version of ASCOT-Carer to be used in Austrian populations. Furthermore, the results give insight into which LTC-QoL-outcomes are seen as particularly (un)desirable, and may therefore help to better tailor services directed at informal carers and the persons they care for.

Population-based preference weights for the Adult Social Care Outcomes Toolkit (ASCOT) for service users for Austria: Findings from a best-worst experiment.

BACKGROUND: The Adult Social Care Outcomes Toolkit (ASCOT) measures quality-of-life (QoL) outcomes of long-term care (LTC) service provision. Country-specific preference weights are required to calculate ASCOT scores. ASCOT has been translated into German, but lacks preference weights for German-speaking countries. OBJECTIVES: This paper aims to establish Austrian preference weights for the German version of the ASCOT service user measure, using best-worst scaling (BWS). METHODS: Data were collected using an online BWS-experiment from a general population sample (n=1,000) of Austrian adults. We use a scale-adjusted multinomial logit model (S-MNL) accounting for positioning effects to estimate preference weights. RESULTS: Austrians value the top attribute-levels in the ASCOT domains 'being meaningfully occupied during the day' and 'having control over daily life' most highly, whereas high needs were the least preferred in the domains 'dignity' and 'social participation'. From a methods perspective, we found significant positioning effects only for 'best' choices, with statements at the top of a list being picked more often than those further down in the list. Factors related to survey completion (self-assessed understanding of the tasks and survey completion time) were shown to have the greatest effect on individual choice consistency. DISCUSSION: The paper provides Austrian preference weights for the German version of ASCOT for service users. The weights also provide insight into how Austrians value different LTC-QoL states. Future research may investigate how values for different LTC-QoL states differ between socioeconomic groups.

Muscle strengthening intervention for boys with haemophilia: Developing and evaluating a best-practice exercise programme with boys, families and health-care professionals.

BACKGROUND: Muscle strengthening exercises have the potential to improve outcomes for boys with haemophilia, but it is unclear what types of exercise might be of benefit. We elicited the views of health-care professionals, boys and their families to create and assess a home-based muscle strengthening programme. OBJECTIVE: To design and develop a muscle strengthening programme with health-care professionals aimed at improving musculoskeletal health, and refine the intervention by engaging boys with haemophilia and their families (Study 1). Following delivery, qualitatively evaluate the feasibility and acceptability of the exercise programme with the boys and the study's physiotherapists (Study 2). DESIGN: A person-based approach was used for planning and designing the exercise programme, and evaluating it post-delivery. The following methods were utilized: modified nominal group technique (NGT) with health-care professionals; focus group with families; exit interviews with boys; and interviews with the study's physiotherapists. RESULTS: Themes identified to design and develop the intervention included exercises to lower limb and foot, dosage, age accommodating, location, supervision and monitoring and incentivization. Programme refinements were carried out following engagement with the boys and families who commented on: dosage, location, supervision and incentivization. Following delivery, the boys and physiotherapists commented on progression and adaptation, physiotherapist contact, goal-setting, creating routines and identifying suitable timeframes, and a repeated theme of incentivization. CONCLUSIONS: An exercise intervention was designed and refined through engagement with boys and their families. Boys and physiotherapists involved in the intervention's delivery were consulted who found the exercises to be generally acceptable with some minor refinements necessary.

Development of Japanese utility weights for the Adult Social Care Outcomes Toolkit (ASCOT) SCT4.

PURPOSE: In developed countries, progressive rapid aging is increasing the need for social care. This study aimed to determine Japanese utility weights for the Adult Social Care Outcomes Toolkit (ASCOT) four-level self-completion questionnaire (SCT4). METHODS: We recruited 1050 Japanese respondents from the general population, stratified by sex and age, from five major cities. In the best-worst scaling (BWS) phase, respondents ranked various social care-related quality of life (SCRQoL) states as "best," "worst," "second-best," or "second-worst," as per the ASCOT. Then, respondents were asked to evaluate eight different SCRQOL states by composite time-trade off (cTTO). A mixed logit model was used to analyze BWS data. The association between cTTO and latent BWS scores was used to estimate a scoring formula that would convert BWS scores to SC-QALY (social care quality-adjusted life year) scores. RESULTS: Japanese BWS weightings for ASCOT-SCT4 were successfully estimated and found generally consistent with the UK utility weights. However, coefficients on level 3 of "Control over daily life" and "Occupation" domains differed markedly between Japan and the UK. The worst Japanese SCRQoL state was lower than that for the UK, as Japanese cTTO results showed more negative valuations. In general, Japanese SC-QALY score (for more than 90% of health states) was lower than that for the UK. CONCLUSIONS: We successfully obtained Japanese utility weights for ASCOT SCT4. This will contribute to the measurement and understanding of social care outcomes.

Carer Social Care-Related Quality of Life Outcomes: Estimating English Preference Weights for the Adult Social Care Outcomes Toolkit for Carers.

BACKGROUND: There is increasing interest in assessing the effects of interventions on older people, people with long-term conditions and their informal carers for use in economic evaluation. The Adult Social Care Outcomes Toolkit for Carers (ASCOT-Carer) is a measure that specifically assesses the impact of social care services on informal carers. To date, the ASCOT-Carer has not been preference-weighted. OBJECTIVES: To estimate preference-based index values for the English version of the ASCOT-Carer from the general population in England. METHODS: The ASCOT-Carer consists of 7 domains, each reflecting aspects of social care-related quality of life in informal carers. Preferences for the ASCOT-Carer social care-related quality of life states were estimated using a best-worst scaling exercise in an online survey. The survey was administered to a sample of the general adult population in England (n = 1000). Participants were asked to put themselves into the hypothetical state of being an informal carer and indicate which attribute they thought was the best (first and second) and worst (first and second) from a profile list of 7 attributes reflecting the 7 domains, each ranging at a different level (1-4). Multinomial logit regression was used to analyze the data and estimate preference weights for the ASCOT-Carer measure. RESULTS: The most valued aspect by English participants was the 'occupation' attribute at its highest level. Results further showed participants rated having no control over their daily life as the lowest attribute-level of all those presented. The position of the 7 attributes influenced participants' best and worst choices, and there was evidence of both scale and taste heterogeneity on preferences. CONCLUSION: This study has established a set of preference-based index values for the ASCOT-Carer in England derived from the best-worst scaling exercise that can be used for economic evaluation of interventions on older individuals and their informal carers.

Protocol for a feasibility randomised controlled trial of a musculoskeletal exercise intervention versus usual care for children with haemophilia.

INTRODUCTION: Haemophilia is a rare, inherited disorder in which blood does not clot normally, resulting in bleeding into joints and muscles. Long-term consequence is disabling joint pain, stiffness, muscle weakness, atrophy and reduced mobility. The purpose of this proposed feasibility of a randomised controlled trial (RCT) is to test the feasibility of an age-appropriate physiotherapy intervention designed to improve muscle strength, posture and the way boys use their joints during walking and everyday activities. METHODS AND ANALYSIS: A small-scale two-centre RCT of a 12-week muscle strengthening exercise intervention versus usual care for young children with haemophilia will be conducted. Primary outcomes will be safety and adherence to the exercise intervention. Secondary outcomes will include recruitment, retention and adverse event rates, clinical data, muscle strength, joint biomechanics and foot loading patterns during walking, 6 min timed walk, timed-up-and-down-stairs, EQ-5D-Y, participants' perceptions of the study, training requirements and relevant costs. Recruitment, follow-up, safety and adherence rates will be described as percentages. Participant diary and interview data will be analysed using a framework analysis. Demographic and disease variable distributions will be analysed for descriptive purposes and covariant analysis. Estimates of differences between treatment arms (adjusted for baseline) and 75% and 95% CIs will be calculated. ETHICS AND DISSEMINATION: The study has ethical approval from the London-Fulham Research Ethics Committee (17/LO/2043) as well as Health Research Authority approval. As well as informing the design of the definitive trial, results of this study will be presented at local, national and international physiotherapy and haemophilia meetings as well as manuscripts submitted to peer-reviewed journals. We will also share the main findings of the study to all participants and the Haemophilia Society.

Safety and efficacy of co-careldopa as an add-on therapy to occupational and physical therapy in patients after stroke (DARS): a randomised, double-blind, placebo-controlled trial.

BACKGROUND: Dopamine is a key modulator of striatal function and learning and might improve motor recovery after stroke. Previous small trials of dopamine agonists after stroke provide equivocal evidence of effectiveness on improving motor recovery. We aimed to assess the safety and efficacy of co-careldopa plus routine occupational and physical therapy during early rehabilitation after stroke. METHODS: This double-blind, multicentre, randomised controlled trial of co-careldopa versus placebo in addition to routine NHS occupational and physical therapy was done at 51 UK NHS acute inpatient stroke rehabilitation services. We recruited patients with new or recurrent clinically diagnosed ischaemic or haemorrhagic (excluding subarachnoid haemorrhage) stroke 5-42 days before randomisation, who were unable to walk 10 m or more, had a score of less than 7 points on the Rivermead Mobility Index, were expected to need rehabilitation, and were able to access rehabilitation after discharge from hospital. Participants were assigned (1:1) using stratified random blocks to receive 6 weeks of oral co-careldopa or matched placebo in addition to routine NHS physiotherapy and occupational therapy. The initial two doses of co-careldopa were 62·5 mg (50 mg of levodopa and 12·5 mg of carbidopa) and the remaining doses were 125 mg (100 mg of levodopa and 25 mg of carbidopa). Participants were required to take a single oral tablet 45-60 min before physiotherapy or occupational therapy session. The primary outcome was ability to walk independently, defined as a Rivermead Mobility Index score of 7 or more, at 8 weeks. Primary and safety analyses were done in the intention-to-treat population. The trial is registered on the ISRCTN registry, number ISRCTN99643613. FINDINGS: Between May 30, 2011, and March 28, 2014, of 1574 patients found eligible, 593 (mean age 68·5 years) were randomly assigned to either the co-careldopa group (n=308) or to the placebo group (n=285), on an average 18 days after stroke onset. Primary outcome data were available for all 593 patients. We found no evidence that the ability to walk independently improved with co-careldopa (125 [41%] of 308 patients) compared with placebo (127 [45%] of 285 patients; odds ratio 0·78 [95% CI 0·53-1·15]) at 8 weeks. Mortality at 12 months did not differ between the two groups (22 [7%] vs 17 [6%]). Serious adverse events were largely similar between groups. Vomiting during therapy sessions, after taking the study drug, was the most frequent adverse event and was more frequent in the co-careldopa group than the placebo group (19 [6·2%] vs 9 [3·2%]). INTERPRETATION: Co-careldopa in addition to routine occupational and physical therapy does not seem to improve walking after stroke. Further research might identify subgroups of patients with stroke who could benefit from dopaminergic therapy at different doses or times after stroke with more intensive motor therapy. FUNDING: Medical Research Council.

Comparing internet and face-to-face surveys as methods for eliciting preferences for social care-related quality of life: evidence from England using the ASCOT service user measure.

PURPOSE: Traditionally, researchers have relied on eliciting preferences through face-to-face interviews. Recently, there has been a shift towards using internet-based methods. Different methods of data collection may be a source of variation in the results. In this study, we compare the preferences for the Adult Social Care Outcomes Toolkit (ASCOT) service user measure elicited using best-worst scaling (BWS) via a face-to-face interview and an online survey. METHODS: Data were collected from a representative sample of the general population in England. The respondents (face-to-face: n = 500; online: n = 1001) completed a survey, which included the BWS experiment involving the ASCOT measure. Each respondent received eight best-worst scenarios and made four choices (best, second best, worst, second worst) in each scenario. Multinomial logit regressions were undertaken to analyse the data taking into account differences in the characteristics of the two samples and the repeated nature of the data. RESULTS: We initially found a number of small significant differences in preferences between the two methods across all ASCOT domains. These differences were substantially reduced-from 15 to 5 out of 30 coefficients being different at the 5% level-and remained small in value after controlling for differences in observable and unobservable characteristics of the two samples. CONCLUSIONS: This comparison demonstrates that face-to-face and internet surveys may lead to fairly similar preferences for social care-related quality of life when differences in sample characteristics are controlled for. With or without a constant sampling frame, studies should carefully design the BWS exercise and provide similar levels of clarification to participants in each survey to minimise the amount of error variance in the choice process.

Correction to: Parental Health Spillover in Cost­Effectiveness Analysis: Evidence from Self­Harming Adolescents in England.

The Open Access license, which previously read.

The impact of long-term care on primary care doctor consultations for people over 75 years.

Many countries are adopting policies to create greater coordination and integration between acute and long-term care services. This policy is predicated on the assumption that these service areas have interdependent outcomes for patients. In this paper, we study the interdependencies between the long-term (home care) services and consultations with a primary care doctor, as used by people over 75 years. Starting with a model of individual's demand for doctor consultations, given supply, we formalize the hypothesis that exogenous increases to home care supply will reduce the number of consultations where these services are technical substitutes. Furthermore, greater coordination of public service planning and use of pooled budgets could lead to better outcomes because planners can account for these externalities. We test our main hypothesis using data from the British Household Panel Study for 1991-2009. To address potential concerns about endogeneity, we use a set of instrumental variables for home care motivated by institutional features of the social care system. We find that there is a statistically significant substitution effect between home care and doctor visits, which is robust across a range of specifications. This result has implications for policies that consider increased coordination between health care and social care systems.

Parental Health Spillover in Cost-Effectiveness Analysis: Evidence from Self-Harming Adolescents in England.

OBJECTIVE: This article presents alternative parental health spillover quantification methods in the context of a randomised controlled trial comparing family therapy with treatment as usual as an intervention for self-harming adolescents, and discusses the practical limitations of those methods. METHODS: The trial followed a sample of 754 participants aged 11-17 years. Health utilities are measured using answers to the EuroQoL 5 Dimensions 3 Levels (EQ-5D-3L) for the adolescent and the Health Utility Index (HUI2) for one parent at baseline, 6 and 12 months. We use regression analyses to evaluate the association between the parent's and adolescent's health utilities as part of an explanatory regression model including health-related and demographic characteristics of both the adolescent and the parent. We then measure cost-effectiveness over a 12-month period as mean incremental cost-effectiveness ratios using various spillover quantification methods. We propose an original quantification based on the use of a household welfare function along with an equivalence scale to generate a health gain within the family to be added to the adolescent's quality-adjusted life-year gain. RESULTS: We find that the parent's health utility increased over the duration of the trial and is significantly and positively associated with adolescent's health utility at 6 and 12 months but not at baseline. When considering the adolescent's health gain only, the incremental cost-effectiveness ratio is £40,453 per quality-adjusted life-year. When including the health spillover to one parent, the incremental cost-effectiveness ratio estimates range from £27,167 per quality-adjusted life-year to £40,838 per quality-adjusted life-year and can be a dominated option depending on the quantification method used. CONCLUSION: According to the health spillover quantification method considered, the incremental cost-effectiveness ratios vary from within the National Institute for Health and Care Excellence (NICE) cost-effectiveness threshold range to not being cost-effective.

A pragmatic randomised controlled trial and economic evaluation of family therapy versus treatment as usual for young people seen after second or subsequent episodes of self-harm: the Self-Harm Intervention - Family Therapy (SHIFT) trial.

BACKGROUND: Self-harm in adolescents is common and repetition rates high. There is limited evidence of the effectiveness of interventions to reduce self-harm. OBJECTIVES: To assess the clinical effectiveness and cost-effectiveness of family therapy (FT) compared with treatment as usual (TAU). DESIGN: A pragmatic, multicentre, individually randomised controlled trial of FT compared with TAU. Participants and therapists were aware of treatment allocation; researchers were blind to allocation. SETTING: Child and Adolescent Mental Health Services (CAMHS) across three English regions. PARTICIPANTS: Young people aged 11-17 years who had self-harmed at least twice presenting to CAMHS following self-harm. INTERVENTIONS: Eight hundred and thirty-two participants were randomised to manualised FT delivered by trained and supervised family therapists (n = 415) or to usual care offered by local CAMHS following self-harm (n = 417). MAIN OUTCOME MEASURES: Rates of repetition of self-harm leading to hospital attendance 18 months after randomisation. RESULTS: Out of 832 young people, 212 (26.6%) experienced a primary outcome event: 118 out of 415 (28.4%) randomised to FT and 103 out of 417 (24.7%) randomised to TAU. There was no evidence of a statistically significant difference in repetition rates between groups (the hazard ratio for FT compared with TAU was 1.14, 95% confidence interval 0.87 to 1.49; p = 0.3349). FT was not found to be cost-effective when compared with TAU in the base case and most sensitivity analyses. FT was dominated (less effective and more expensive) in the complete case. However, when young people's and caregivers' quality-adjusted life-year gains were combined, FT incurred higher costs and resulted in better health outcomes than TAU within the National Institute for Health and Care Excellence cost-effectiveness range. Significant interactions with treatment, indicating moderation, were detected for the unemotional subscale on the young person-reported Inventory of Callous-Unemotional Traits (p = 0.0104) and the affective involvement subscale on the caregiver-reported McMaster Family Assessment Device (p = 0.0338). Caregivers and young people in the FT arm reported a range of significantly better outcomes on the Strengths and Difficulties Questionnaire. Self-reported suicidal ideation was significantly lower in the FT arm at 12 months but the same in both groups at 18 months. No significant unexpected adverse events or side effects were reported, with similar rates of expected adverse events across trial arms. CONCLUSIONS: For adolescents referred to CAMHS after self-harm, who have self-harmed at least once before, FT confers no benefits over TAU in reducing self-harm repetition rates. There is some evidence to support the effectiveness of FT in reducing self-harm when caregivers reported poor family functioning. When the young person themselves reported difficulty expressing emotion, FT did not seem as effective as TAU. There was no evidence that FT is cost-effective when only the health benefits to participants were considered but there was a suggestion that FT may be cost-effective if health benefits to caregivers are taken into account. FT had a significant, positive impact on general emotional and behavioural problems at 12 and 18 months. LIMITATIONS: There was significant loss to follow-up for secondary outcomes and health economic analyses; the primary outcome misses those who do not attend hospital following self-harm; and the numbers receiving formal FT in the TAU arm were higher than expected. FUTURE WORK: Evaluation of interventions targeted at subgroups of those who self-harm, longer-term follow-up and methods for evaluating health benefits for family groups rather than for individuals. TRIAL REGISTRATION: Current Controlled Trials ISRCTN59793150. FUNDING: This project was funded by the NIHR Health Technology Assessment programme and will be published in full in Health Technology Assessment; Vol. 22, No. 12. See the NIHR Journals Library website for further project information.