Obtaining preference scores for an abbreviated self-completion version of the Teen-Addiction Severity Index (ASC T-ASI) to value therapy outcomes of systemic family interventions: a discrete choice experiment.

BACKGROUND: Systemic family interventions for adolescents with problems of substance use and/or delinquency are increasingly focused subject of economic evaluations. Treatment effects go beyond improvements in commonly measured health-related quality of life (HRQOL). The Teen-Addiction Severity Index (T-ASI) was identified as capable of capturing these broad outcomes. However, it lacks preference-based scores. An abbreviated self-completion version (ASC T-ASI) was created and validated, covering the T-ASI domains substance use, school, work, family, social relationships, justice, and mental health. This study aimed to obtain societal preference scores for the ASC T-ASI. METHODS: Preferences were elicited in a sample of the Dutch general adult population (n = 1500), using a web-based Discrete Choice Experiment. Choice tasks included two unlabeled alternatives with attributes and levels corresponding to the domains and levels of the ASC T-ASI. A pilot study (n = 106) informed priors, optimal presentation, and number of choice tasks applied in the main study. Data were analyzed using a mixed multinomial logit model. RESULTS: Preference scores were logically ordered, with lower scores for worse ASC T-ASI states. Scores were most influenced by reductions in problems concerning the domains substance use, mental health, justice, and family. Tariffs were calculated for each ASC T-ASI state, ranging from 0 (worst situation) to 1 (best situation). CONCLUSIONS: The tariffs enable preference-based assessments of the broad effects of systemic family interventions for adolescents with problems of substance use and/or delinquency. The outcome reflects addiction-related rather than health-related utility and can be used next to generic HRQOL instruments in relevant economic evaluations. Given the source used for the preferences, interpretations and valuation of scores require attention.

Clinicians' Views on Therapeutic Outcomes of Systemic Interventions and on the Ability of the EQ-5D to Capture these Outcomes.

BACKGROUND: Systemic interventions focus on improvements of interactions between clients and their environments, and are increasingly used to treat adolescents with problems of substance use and delinquency. Clients' progress may include broad and non-medical effects. When performing economic evaluations of these interventions, the common outcome of costs per quality adjusted life year (cost/QALY) may not capture all of these effects. AIMS OF THE STUDY: The current study is an explorative study. It aims to investigate which outcomes clinicians consider relevant to the therapeutic success of systemic interventions and whether these, according to them, are sufficiently captured by the EQ-5D instrument. METHODS: Semi-structured interviews were performed with seven clinicians at two mental health institutions in the Netherlands. Clinicians were asked to list the most relevant outcomes of systemic interventions. They were asked whether they considered the EQ-5D dimensions to sufficiently capture these outcomes or if they missed aspects or outcome domains. RESULTS: The clinicians mentioned several broad effects relevant for the evaluation of systemic interventions. These were aspects of family functioning, parental functioning, social competencies, school attendance, etc. They considered several EQ-5D dimensions relevant (i.e. in particular 'usual activities' and 'anxiety/depression'), yet they indicated that the instrument lacked systemic dimensions (i.e. family relations and relations with others) and addiction-related aspects. DISCUSSION: The interviewed clinicians considered several dimensions of the EQ-5D useful in evaluating effects of systemic interventions, yet they expressed the need to add additional dimensions particularly relevant to systemic aspects to the instrument when performing economic evaluations of systemic interventions. The explorative analysis was limited by the small number of interviewed clinicians. Furthermore, a relatively high proportion of clinicians were specialized in Multidimensional Family Therapy, a type of systemic intervention particularly used to treat adolescents with substance use disorders and related problems. Hence the importance of addiction-related improvements may have been over-emphasized in this group of respondents. IMPLICATIONS FOR HEALTH CARE PROVISION AND USE: Practical implications of the current study may be the need for enhancements of the current health economic methodology for evaluating systemic interventions as to capture additional aspects specifically relevant to these interventions. This may lead to different choices in the use of instruments for the evaluation of treatment progress and success in clinical practice. IMPLICATIONS FOR HEALTH POLICIES: By improving the health economic toolkit to evaluate systemic interventions one may provide policy recommendations in line with the therapeutic goals of the interventions. IMPLICATIONS FOR FURTHER RESEARCH: Further research could be directed at investigating the suitability of other available instruments than the EQ-5D for economic evaluations of systemic interventions.

The cost-effectiveness of family/family-based therapy for treatment of externalizing disorders, substance use disorders and delinquency: a systematic review.

BACKGROUND: Family therapy and family-based treatment has been commonly applied in children and adolescents in mental health care and has been proven to be effective. There is an increased interest in economic evaluations of these, often expensive, interventions. The aim of this systematic review is to summarize and evaluate the evidence on cost-effectiveness of family/family-based therapy for externalizing disorders, substance use disorders and delinquency. METHODS: A systematic literature search was performed in PubMed, Education Resource information Centre (ERIC), Psycinfo and Cochrane reviews including studies conducted after 1990 and before the first of August of 2013. Full economic evaluations investigating family/family-based interventions for adolescents between 10 and 20 years treated for substance use disorders, delinquency or externalizing disorders were included. RESULTS: Seven hundred thirty-one articles met the search criteria and 51 studies were initially selected. The final selection resulted in the inclusion of 11 studies. The quality of these studies was assessed. Within the identified studies, there was great variation in the specific type of family/family-based interventions and disorders. According to the outcomes of the checklists, the overall quality of the economic evaluations was low. Results varied by study. Due to the variations in setting, design and outcome it was not feasible to pool results using a meta-analysis. CONCLUSIONS: The quality of the identified economic evaluations of family/family-based therapy for treatment of externalizing disorders, adolescent substance use disorders and delinquency was insufficient to determine the cost-effectiveness. Although commonly applied, family/family-based therapy is costly and more research of higher quality is needed.

Value of Information Analysis Applied to the Economic Evaluation of Interventions Aimed at Reducing Juvenile Delinquency: An Illustration.

OBJECTIVES: To investigate whether a value of information analysis, commonly applied in health care evaluations, is feasible and meaningful in the field of crime prevention. METHODS: Interventions aimed at reducing juvenile delinquency are increasingly being evaluated according to their cost-effectiveness. Results of cost-effectiveness models are subject to uncertainty in their cost and effect estimates. Further research can reduce that parameter uncertainty. The value of such further research can be estimated using a value of information analysis, as illustrated in the current study. We built upon an earlier published cost-effectiveness model that demonstrated the comparison of two interventions aimed at reducing juvenile delinquency. Outcomes were presented as costs per criminal activity free year. RESULTS: At a societal willingness-to-pay of €71,700 per criminal activity free year, further research to eliminate parameter uncertainty was valued at €176 million. Therefore, in this illustrative analysis, the value of information analysis determined that society should be willing to spend a maximum of €176 million in reducing decision uncertainty in the cost-effectiveness of the two interventions. Moreover, the results suggest that reducing uncertainty in some specific model parameters might be more valuable than in others. CONCLUSIONS: Using a value of information framework to assess the value of conducting further research in the field of crime prevention proved to be feasible. The results were meaningful and can be interpreted according to health care evaluation studies. This analysis can be helpful in justifying additional research funds to further inform the reimbursement decision in regard to interventions for juvenile delinquents.

Association between societal costs and treatment response in children and adolescents with ADHD and their parents. A cross-sectional study in the Netherlands.

Attention Deficit Hyperactivity Disorder (ADHD) is associated with considerable burden of illness at a patient, family and societal level. Although pharmacological treatment is recommended by authoritative guidelines, evidence on its influence on the broader burden of illness is limited. As treatment induces costs, proper healthcare decision making requires evidence on the associated societal costs or benefits and particularly the difference that response to treatment can make. Data on ADHD related resource use of patients 8-18 years and parents were collected by means of a cross-sectional, online survey amongst members of the Dutch parent association. Children were stratified to responders and non-responders to treatment according to pre-defined expert definitions. Analyses were performed on 618 questionnaires (428 responders; 190 non-responders to treatment). Children were 11.8 years on average and mainly boys (82 %). Total monthly costs for children were €578 and €839 for responders and non-responders, respectively (p = 0.021), with a breakdown to direct medical costs (€322 vs. €512; p = 0.068), direct non-medical costs (€222 vs. €296; p = 0.090), and indirect non-medical costs (€34 vs. €57; p < 0.001). For parents, total costs were €246 vs. €399 for the responding and non-responding children, respectively (p = 0.006), with a breakdown to direct medical costs (€130 vs. €211; p = 0.010) and indirect non-medical costs (€116 vs. €181; p = 0.092). Total monthly costs of children and their parents together were €824 and €1228 for responders and non-responders to treatment, respectively (p = 0.002). These results stress the importance of a focus on response to treatment, not only beneficial for patients and their family, but also resulting in considerable societal benefits.

Probabilistic Markov Model Estimating Cost Effectiveness of Methylphenidate Osmotic-Release Oral System Versus Immediate-Release Methylphenidate in Children and Adolescents: Which Information is Needed?

BACKGROUND: Incidence of attention deficit hyperactivity disorder (ADHD) in children and adolescents has been increasing. The disorder results in high societal costs. Policymakers increasingly use health economic evaluations to inform decisions on competing treatments of ADHD. Yet, health economic evaluations of first-choice medication of ADHD in children and adolescents are scarce and generally do not include broader societal effects. OBJECTIVES: This study presents a probabilistic model and analysis of methylphenidate osmotic-release oral system (OROS) versus methylphenidate immediate-release (IR). We investigate and include relevant societal aspects in the analysis so as to provide cost-effectiveness estimates based on a broad societal perspective. METHODS: We enhanced an existing Markov model and determined the cost effectiveness of OROS versus IR for children and adolescents responding suboptimally to treatment with IR. Enhancements included screening of a broad literature base, updated utility values, inclusion of costs and effects on caregivers and a change of the model type from deterministic to probabilistic. RESULTS: The base case scenario resulted in lower incremental costs (€-5815) of OROS compared with IR and higher incremental quality-adjusted life-year (QALY) gains (0.22). Scenario analyses were performed to determine sensitivity to changes in transition rates, utility of caregivers, medical costs of caregivers and daily medication dose. CONCLUSIONS: The results indicate that, for children responding suboptimally to treatment with IR, the beneficial effect of OROS on compliance may be worth the additional costs of medication. The presented model adds to the health economic information available for policymakers and to considerations on a broader perspective in cost-effectiveness analyses.

Association between quality of life and treatment response in children with attention Deficit Hyperactivity Disorder and their parents.

BACKGROUND: It has been shown that Attention Deficit Hyperactivity Disorder (ADHD) lowers the Quality of Life (QoL) of patients and their families. Medication as part of the treatment has a favourable effect on symptoms as well as functioning. Evidence on the impact of pharmacological treatment on symptoms of ADHD and the QoL of the patient and their family is still limited. There is a need for further research on QoL in ADHD as well as the relationship between ADHD and the impact on families rather than solely on patients. AIMS OF THE STUDY: Measure QoL of children with ADHD and their parents and explore the association of QoL with treatment response. METHODS: A cross-sectional survey was performed using an online questionnaire to collect QoL data of children with ADHD (based on proxy reporting of parents) and their parents in a sample of members of an ADHD parent association. QoL was measured by EQ-5D and KIDSCREEN-10. Treatment response was based on descriptions by experts, based on compliance and functioning. RESULTS: Analyses were based on 618 questionnaires (treatment responder n=428, treatment non-responder n=190). Mean age of the children was 11.8 years (82.4% male). QoL according to EQ-5D utility was 0.83 and 0.74 for responders and non-responders, respectively (p<0.001). For KIDSCREEN-10 the index was 42.24 and 40.33 for responders and non-responders, respectively (p<0.001). EQ-5D utility scores of the parents were 0.83 on average; no association with their child's treatment response could be established. A significant positive correlation between EQ-5D utility of the children and EQ-5D utility of the parents (R2=0.207, p<0.001) was found. The association between treatment response and children's QoL was significantly influenced by age category, having a sibling with ADHD, and presence of comorbidity. DISCUSSION: Strengths of this study are its sample size and the inclusion of QoL of parents, which has not been reported simultaneously before. The facts that data were derived from members of the ADHD parent association, the data for children were based on parents' report, and lack of possibility for confirmation of the clinical diagnosis are the main study limitations. IMPLICATIONS FOR HEALTH CARE PROVISION AND USE: QoL of children with ADHD was shown to be significantly positively associated with response to treatment and negatively affected by comorbidity. In determining the treatment effects in ADHD, QoL and family overall well-being should be a standard consideration as well as an integrated part of health policy discussions on ADHD. IMPLICATIONS FOR HEALTH POLICIES: Policymakers in the field of ADHD should focus on QoL of the patient, but also on the broader effects of effective treatment on the well-being of the parents. IMPLICATIONS FOR FURTHER RESEARCH: Suggestions for further research include the repetition of this study including a control group and obtaining children's self-report on QoL and clinicians report on diagnosis.

Validity and responsiveness of the EQ-5D and the KIDSCREEN-10 in children with ADHD.

BACKGROUND: The aim of our study is to compare the validity of a generic preference-based Quality of Life (QoL) instrument for adults to that of a generic child-specific QoL instrument in children and adolescents with attention deficit hyperactivity disorder (ADHD). METHODS: EQ-5D and KIDSCREEN-10 data were collected using a questionnaire survey performed among parents with a child or adolescent diagnosed with ADHD. The measurements were compared to assess (dis)similarities of the instruments' constructs and responsiveness to different health states. Principal component analysis (PCA) with varimax rotation was used to identify factors underlying the constructs of both instruments. Instruments' index scores of respondents with different treatment and comorbidity profiles were compared using Student's t tests. Cohen's effect sizes were calculated for an indirect comparison of the instruments' responsiveness and discriminating ability. Separate analyses were performed in children aged 8-12 and 13-18 years. RESULTS: A strong relation was found between the EQ-5D and KIDSCREEN-10 index scores. However correlations between EQ-5D and KIDSCREEN-10 items were moderate or low. The PCA identified five separate factors of quality of life. A physical and a mental factor included a combination of three EQ-5D dimensions and six KIDSCREEN-10 items; the remaining EQ-5D and KIDSCREEN-10 items constituted complementary factors without any overlap between the separate instruments. Scores of both instruments differed significantly according to respondents' response to treatment and comorbidity profile. Cohen's effect sizes indicated comparable results of the instruments' responsiveness and discriminative ability. CONCLUSIONS: The results highlight that the instruments measure different constructs of QoL in children with ADHD. Despite this, the analyses showed comparable responsiveness and discriminative ability of the instruments. These results suggest that for economic evaluations, the EQ-5D is an appropriate and valid instrument for measuring QoL in children.

Framework for modelling the cost-effectiveness of systemic interventions aimed to reduce youth delinquency.

BACKGROUND: Many interventions initiated within and financed from the health care sector are not necessarily primarily aimed at improving health. This poses important questions regarding the operationalisation of economic evaluations in such contexts. AIMS OF THE STUDY: We investigated whether assessing cost-effectiveness using state-of-the-art methods commonly applied in health care evaluations is feasible and meaningful when evaluating interventions aimed at reducing youth delinquency. METHODS: A probabilistic Markov model was constructed to create a framework for the assessment of the cost-effectiveness of systemic interventions in delinquent youth. For illustrative purposes, Functional Family Therapy (FFT), a systemic intervention aimed at improving family functioning and, primarily, reducing delinquent activity in youths, was compared to Treatment as Usual (TAU). "Criminal activity free years" (CAFYs) were introduced as central outcome measure. Criminal activity may e.g. be based on police contacts or committed crimes. In absence of extensive data and for illustrative purposes the current study based criminal activity on available literature on recidivism. Furthermore, a literature search was performed to deduce the model's structure and parameters. RESULTS: Common cost-effectiveness methodology could be applied to interventions for youth delinquency. Model characteristics and parameters were derived from literature and ongoing trial data. The model resulted in an estimate of incremental costs/CAFY and included long-term effects. Illustrative model results point towards dominance of FFT compared to TAU. DISCUSSION: Using a probabilistic model and the CAFY outcome measure to assess cost-effectiveness of systemic interventions aimed to reduce delinquency is feasible. However, the model structure is limited to three states and the CAFY measure was defined rather crude. Moreover, as the model parameters are retrieved from literature the model results are illustrative in the absence of empirical data. IMPLICATIONS FOR HEALTH CARE PROVISION AND USE: The current model provides a framework to assess the cost-effectiveness of systemic interventions, while taking into account parameter uncertainty and long-term effectiveness. IMPLICATIONS FOR HEALTH POLICIES: The framework of the model could be used to assess the cost-effectiveness of systemic interventions alongside (clinical) trial data. Consequently, it is suitable to inform reimbursement decisions, since the value for money of systemic interventions can be demonstrated using a decision analytic model. IMPLICATIONS FOR FURTHER RESEARCH: Future research could be focussed on testing the current model based on extensive empirical data, improving the outcome measure and finding appropriate values for that outcome.