Economic evaluation is a tool used to inform decision makers on the efficiency of comparative healthcare interventions and inform resource allocation decisions. There is a growing need for the use of economic evaluations to assess the value of interventions for children with neurodevelopmental disorders (NDDs), a population that has increasing demands for healthcare services. Unfortunately, few evaluations have been conducted to date, perhaps stemming from challenges in applying existing economic evaluation methodologies in this heterogeneous population. Opportunities exist to innovate methods to address key challenges in conducting economic evaluations of interventions for children with NDDs. In this paper, we discuss important considerations and highlight areas for future work. This includes the paucity of appropriate instruments for measuring outcomes meaningful to children with NDDs and their families, difficulties in the measurement of costs due to service utilization in a wide variety of sectors, complexities in the measurement of caregiver and family effects and considerations in estimating long-term productivity costs. Innovation and application of evaluation approaches in these areas will help inform decisions around whether the resources currently spent on interventions for children with NDDs represent good value for money, or whether greater benefits for children could be generated by spending money in other ways.

BackgroundEarly detection of neurodevelopmental disorders (NDDs) enables access to early interventions for children. We assess the Ages and Stages Questionnaire (ASQ)’s ability to identify children with a NDD in population data.MethodChildren 4 to 5 years old in the National Longitudinal Survey of Children and Youth (NLSCY) from cycles 5 to 8 were included. The sensitivity, specificity, positive and negative predictive values were calculated for the ASQ at 24, 27, 30, 33, 36 and 42 months. Fixed effects regression analyses assessed longitudinal associations between domain scores and child age.ResultsSpecificity for the ASQ was high with 1SD or 2SD cutoffs, indicating good accuracy in detecting children who will not develop a NDD, however the sensitivity varied over time points and cut-offs. Sensitivity for the 1 SD cutoff at 24 months was above the recommended value of 70% for screening. Differences in ASQ domains scores between children with and without NDD increases with age.ConclusionsThe high specificity and negative predictive values of the ASQ support its use in identifying children who are not at the risk of developing a NDD. The capacity of the ASQ to identify children with a NDD in the general population is limited except for the ASQ-24 months with 1SD and can be used to identify children at risk of NDD.Electronic supplementary materialThe online version of this article (10.1186/s12887-018-1105-z) contains supplementary material, which is available to authorized users.

The employment rate among persons with autism spectrum disorder has been noted as unacceptably low. Employment-support services are increasingly linked to the potential for favorable job outcomes, yet little is known about employment-support practices and the outcome of these interventions. This mixed-methods study examined employment-support resources for persons with autism spectrum disorder. An online survey was completed by 137 senior clinicians or administrators in employment-support programs in Canada. Additionally, 122 follow-up interviews were conducted with individuals with autism spectrum disorder (n = 71) and their parents/caregivers (n = 51). Findings indicate that the quality and beneficial impact of employment-support services for adults with autism spectrum disorder may be more favorably perceived by employment-support personnel than by individuals with autism spectrum disorder and their families. Furthermore, employment-support personnel were more disparaging about autism spectrum disorder vocational support capacity within their community, compared to their own programs. Individuals with autism spectrum disorder and their families seek services that support both accessing and retaining employment. Capacity-building in employment support for youth and adults with autism spectrum disorder is recommended, based on a reported insufficiency of, and a lack of evidence guiding, existing services. Program recommendations and an emerging model for integrated vocational support in autism spectrum disorder are offered.

AIM To describe how generic preference-based health-related quality of life (HRQoL) instruments have been used in research involving children with neurodevelopmental disorders (NDD).METHOD A systematic search of nine databases identified studies that used generic preference-based HRQoL instruments in children with NDD. Data extracted following the Preferred Reporting Items for Systematic Review and Meta-Analyses extension for Scoping Review guidelines included type of NDD, instrument used, respondent type, justification, and critical appraisal for these selections.RESULTS Thirty-six studies were identified: four cost-utility analyses; 15 HRQoL assessments; five economic burden studies; three intervention studies; and nine 'other'. The Health Utilities Index (Mark 2 and Mark 3) and EuroQoL 5D (EQ-5D; three-level EQ-5D, five-level EQ-5D, and the youth version of the EQ-5D) instruments were most frequently used (44% and 31% respectively). The relatively low use of these instruments overall may be due to a lack of psychometric evidence, inconsistency in justification for and lack of clarity on appropriate respondent type and age, and geographical challenges in applying preference weights.INTERPRETATION This study highlights the dearth of studies using generic preference-based HRQoL instruments in children with NDD. The use of cost-utility analysis in this field is limited and validation of these instruments for children with NDD is needed. The quality of data should be considered before guiding policy and care decisions.

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