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PurposeWhole-exome (WES) and whole-genome sequencing (WGS) increase the diagnostic yield in autism spectrum disorder (ASD) compared to chromosomal microarray (CMA), but there have been no comprehensive cost analyses. The objective was to perform such an assessment of CMA, WES, and WGS and compare the incremental cost per additional positive finding in hypothetical testing scenarios.MethodsFive-year patient and program costs were estimated from an institutional perspective. WES and WGS estimates were based on HiSeq 2500 with an additional WGS estimate for HiSeq X platforms. Parameter uncertainty was assessed with probabilistic and deterministic sensitivity analysis.ResultsThe cost per ASD sample was CAD$1,655 (95% CI: 1,611; 1,699) for WES, CAD$2,851 (95% CI: 2,750; 2,956) for WGS on HiSeq X, and CAD$5,519 (95% CI: 5,244; 5,785) on HiSeq 2500, compared to CAD$744 (95% CI 714, 773) for CMA. The incremental cost was over CAD$25,000 per additional positive finding if CMA was replaced by newer technology.ConclusionWhile costs for WES and WGS remain high, future reductions in material and equipment costs, and increased understanding of newly discovered variants and variants of unknown significance will lead to improved value.

Objective It is key for women to return to screening at regular intervals to ensure the effectiveness of early detection and protective effects of the Pap test. A correspondence program was initiated in Ontario in 2013 to implement a recall and reminder letter sent to women 2.9 years from their prior normal Pap test. This study sought to evaluate the impact of the recall letter and reminder on retention rates for cervical screening. Methods A cohort study with a historical control was carried out with an exposed group, defined as women receiving a letter for recall in the week of November 21–27, 2013, and a non-exposed, control arm of women who did not receive the letter but who would have been eligible for correspondence in the same time period in 2012. Results The study population comprised 5182 women in the exposed group and 4223 women in the non-exposed group. Women receiving the letter were more likely to return to screening, with an aOR of 1.82 (95% CI 1.66–1.99). Other significant factors included being registered with a Patient Enrolment Model family physician and having a series of prior Pap tests in the past screening history. Conclusion A correspondence program benefits the retention of women in organized cervical screening programs.

Background: A common feature of most reviews or catalogues of health utilities has been their focus on adult health states or derivation of values from adult populations. More generally, utility measurement in or on behalf of children has been constrained by several methodological concerns. The objective of this study was to conduct the first comprehensive systematic review and meta-analysis of primary utility data for childhood conditions and descriptors, and to determine the effects of methodological factors on childhood utilities.Methods: The review followed PRISMA guidelines. PubMed, Embase, Web of Science, PsycINFO, EconLit, CINAHL and Cochrane Library were searched for primary studies reporting health utilities for childhood conditions or descriptors using direct or indirect valuation methods. The Paediatric Economic Database Evaluation (PEDE) Porject was also searched for cost-utility analyses with primary utility values. Mean or median utilities for each of the main samples were catalogued, and weighted averages of utilities for each health condition were estimated, by valuation method. Mixed-effects meta-regression using hierarchical linear modeling was conducted for the most common valuation methods to estimate the utility decrement for each health condition category relative to general childhood population health, as well as the independent effects of methodological factors.Results: The literature searches resulted in 272 eligible studies. These yielded 3,414 utilities when all sub-groups were considered, covering all ICD-10 chapters relevant to childhood health, 19 valuation methods, 12 respondent types, 8 modes of administration, and data from 36 countries. A total of 1,191 utility values were obtained when only main study samples were considered, and these were catalogued by health condition or descriptor, and methodological characteristics. 1,073 mean utilities for main samples were used for fixed-effects meta-analysis by health condition and valuation method. Mixed-effects meta-regressions estimated that 53 of 76 ICD-10 delineated health conditions, valued using the HUI3, were associated with statistically significant utility decrements relative to general population health, whereas 38 of 57 valued using a visual analog scale (VAS) were associated with statistically significant VAS decrements. For both methods, parental proxy assessment was associated with overestimation of values, whereas adolescents reported lower values than children under 12 y. VAS responses were more heavily influenced by mode of administration than the HUI3.Conclusion: Utilities and their associated distributions, as well as the independent contributions of methodological factors, revealed by this systematic review and meta-analysis can inform future economic evaluations within the childhood context.

This study measured resource utilization and costs for pre-school autism spectrum disorder (ASD)-related services in community-based sectors from multiple payer perspectives in two Canadian provinces, Nova Scotia (NS) and New Brunswick (NB), during the 12 months prior to and following the start of early intensive behavioural intervention (EIBI). The results indicate significant differences between NB and NS in utilization of services and costs to families, public sector and society. Differences can be attributed to variation in EIBI delivery models and may also be influenced by differences in diagnostic assessment practices. The study results provide resource utilization rates and costs which could be used in future economic evaluations and to inform policy making to improve outcomes for children with ASD.

Economic evaluations conducted in children have unique features compared to adults. Important developments in pediatric economic evaluation in recent years include new options for valuing health states for cost-utility analysis (CUA) and shifting child health priorities. The Pediatric Economic Database Evaluation (PEDE) project includes a comprehensive database of pediatric health economic evaluations published since 1980. The objective of this scoping review was to identify trends over time in the use of CUA and other analytic techniques, and the therapeutic areas chosen for study. Areas covered: Medical and grey literature were searched, key characteristics were extracted, frequencies were tabulated and cross-tabulations were performed. Differences between early (1980 and 1999) and late (2000 and 2014) periods were assessed using a chi-squared statistic. Of the 2,776 pediatric economic evaluations published between 1980 and 2014, substantially more were cost-effectiveness analyses (CEAs) and CUAs than cost benefit analyses and cost minimization analyses (63.9 and 24.9% versus 7.6 and 3.6%, respectively). This pattern was consistent regardless of the type of intervention, disease or age group studied. A trend toward higher proportions of CUAs and CEAs was evident in the later period (X

The purpose of the study is to estimate the EQ-5D-derived health utilities associated with selected chronic conditions (hypertension, heart disease, arthritis, asthma or COPD, cancer, diabetes, chronic back pain, and anxiety or depression) and to estimate minimally important differences (MID) based on the Commonwealth Fund Survey of Sicker Adults in Canada. We used a cross-sectional survey of 3765 sick adults in Canada conducted in 2011 by the Commonwealth Fund. Health utilities were calculated for the entire sample and for each of the eight chronic health conditions. An ordinary least squares regression was used to estimate the utility decrement associated with these conditions with and without adjustment for socio-demographic factors. The MIDs were estimated using the anchor- and distribution-based methods. The adjusted utility decrement varied from 0.028 (95 % confidence interval (CI) −0.049, −0.008) for diabetes to 0.124 (95 % CI −0.142, −0.105) for anxiety or depression. The anchor-based MID for the entire group was 0.044 (95 % CI 0.025, 0.062), and the distribution-based MID for the entire group was 0.091. The condition-specific MIDs using the distribution-based method ranged from 0.089 for cancer to 0.108 for asthma or COPD. The MID estimated by the distribution-based method was larger than the MID estimated by the anchor-based method, indicating that the choice of method matters. The impact of arthritis, anxiety or depression, and chronic back pain on health utility was substantial, all exceeding or approximating the MID estimated using either anchor- or distribution-based methods.

Childhood illness can impose significant costs and health strains on family members, but these are not routinely captured by pediatric economic evaluations. This review investigated how family “spillover effects” related to costs and health outcomes are considered in pediatric cost-utility analyses (CUAs). We reviewed pediatric CUAs published between 2000 and 2015 using the Tufts Medical Center Cost-effectiveness Analysis (CEA) Registry and the Pediatric Economic Database Evaluation (PEDE) Registry. We selected studies conducted from the societal perspective and included in both registries. We investigated how frequently family spillover was incorporated into analyses, and how the inclusion of spillover health effects and costs changed CUA results. We found 142 pediatric CUAs meeting inclusion criteria. Of those, 105 (72%) considered either family spillover costs (n = 98 time costs, n = 33 out-of-pocket costs, n = 2 caregiver healthcare costs) or health outcomes (n = 15). Twenty-four studies included 43 pairs of incremental cost-effectiveness ratios (ICERs) with and without spillover. In 19 pairs of ICERs, adding spillover changed the ICER enough to cross a common cost-effectiveness threshold (i.e., $50,000/QALY, $100,000/QALY, $150,000/QALY; values are in 2016 US$). Incorporating spillover generally made interventions more cost-effective (n = 18; 42%), or did not change CUA results enough to cross a threshold (n = 24; 56%). Including family spillover reduced ICERs by 31% ($40,000/QALY) on average. Most pediatric CUAs conducted from a societal perspective include family costs but fewer include family health effects. Inclusion of family spillover effects tends to make CUA results more favorable. Future pediatric CUAs should aim to more fully incorporate the family burden of illness.

The clinical use of whole-genome sequencing (WGS) is expected to alter pediatric medical management. The study aimed to describe the type and cost of healthcare activities following pediatric WGS compared to chromosome microarray (CMA). Healthcare activities prompted by WGS and CMA were ascertained for 101 children with developmental delay over 1 year. Activities following receipt of non-diagnostic CMA were compared to WGS diagnostic and non-diagnostic results. Activities were costed in 2016 Canadian dollars (CDN). Ongoing care accounted for 88.6% of post-test activities. The mean number of lab tests was greater following CMA than WGS (0.55 vs. 0.09; p = 0.007). The mean number of specialist visits was greater following WGS than CMA (0.41 vs. 0; p = 0.016). WGS results (diagnostic vs. non-diagnostic) modified the effect of test type on mean number of activities (p