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Supplementary Appendix A: Table S1. The intervention package within the Salut Programme targeting parents and their children from foetal life to 1.5 years of age. Table S2. Unit costs used in costing analysis (in 2020 $US) - average estimates from Cost per patient database. Table S3. Mean costs per mother per child related to resource use in the Salut and the non-Salut areas, before matching, for the total sample (in 2020 $US). Table S4. Mean costs per mother per child related to resource use in the Salut and the non-Salut areas, before matching, for the longitudinal sample (in 2020 $US).

Purpose: To explore and develop methods to use TTO and DCE data collected in self-administered online surveys to elicit a CALY-SWE value set. Methods: Building on existing methodological knowledge around DCE and TTO studies, we optimized the web survey in an integrated approach that consisted of a qualitative face validity study, iterative web survey development including a three-stage roll-out, a customized experimental design, a sample size simulation. Based on the inconsistencies of TTO answers per participant, we assessed TTO data quality by calculating a score, and examined the effect of excluding TTO data according to this score on the value set modelling. Results: Participants in the quality study informed improvements in the survey’s visual design and phrasing. Based on the sample size simulation, we judged a sample size of 1500 with a balance of six DCE and five TTO questions to be appropriate for the valuation study. Change made for the second stage, for example the introduction of a learning state and of color-coding, improved TTO data quality. Excluding TTO answers per-participant based on the score lead to an improved TTO data foundation for the model by several metrics such as no inconsistent coefficients and reduced standard errors relative to the coefficient magnitude. Conclusion: Developing value sets was feasible with online administered DCE and TTO questions if the web survey is sufficiently optimized and coherent with the experimental design. The severity of inconsistencies could be used to identify and exclude poor quality TTO data to strengthen the value set modelling.

Importance: The availability of behavior therapy for individuals with Tourette syndrome (TS) and chronic tic disorder (CTD) is limited. Objective: To determine the efficacy and cost-effectiveness of internet-delivered exposure and response prevention (ERP) for children and adolescents with TS or CTD. Design, Setting, and Participants: This single-masked, parallel group, superiority randomized clinical trial with nationwide recruitment was conducted at a research clinic in Stockholm, Sweden. Out of 615 individuals assessed for eligibility, 221 participants meeting diagnostic criteria for TS or CTD and aged 9 to 17 years were included in the study. Enrollment began in April 2019 and ended in April 2021. Data were analyzed between October 2021 and March 2022. Interventions: Participants were randomized to 10 weeks of therapist-supported internet-delivered ERP for tics (111 participants) or to therapist-supported internet-delivered education for tics (comparator group, 110 participants). Main Outcomes and Measures: The primary outcome was change in tic severity from baseline to the 3-month follow-up as measured by the Total Tic Severity Score of the Yale Global Tic Severity Scale (YGTSS-TTSS). YGTSS-TTSS assessors were masked to treatment allocation. Treatment response was operationalized as a score of 1 ("Very much improved") or 2 ("Much improved") on the Clinical Global Impression-Improvement scale. Results: Data loss was minimal, with 216 of 221 participants (97.7%) providing primary outcome data. Among randomized participants (152 [68.8%] boys; mean [SD] age, 12.1 [2.3] years), tic severity improved significantly, with a mean reduction of 6.08 points on the YGTSS-TTSS in the ERP group (mean [SD] at baseline, 22.25 [5.60]; at 3-month follow-up, 16.17 [6.82]) and 5.29 in the comparator (mean [SD] at baseline, 23.01 [5.92]; at 3-month follow-up, 17.72 [7.11]). Intention-to-treat analyses showed that the 2 groups improved similarly over time (interaction effect, -0.53; 95% CI, -1.28 to 0.22; P =.17). Significantly more participants were classified as treatment responders in the ERP group (51 of 108 [47.2%]) than in the comparator group (31 of 108 [28.7%]) at the 3-month follow-up (odds ratio, 2.22; 95% CI, 1.27 to 3.90). ERP resulted in more treatment responders at little additional cost compared with structured education. The incremental cost per quality-adjusted life-year gained was below the Swedish willingness-to-pay threshold, at which ERP had a 66% to 76% probability of being cost-effective. Conclusions and Relevance: Both interventions were associated with clinically meaningful improvements in tic severity, but ERP led to higher response rates at little additional cost. Trial Registration: ClinicalTrials.gov identifier: NCT03916055.

Physical conditions in children and adolescents are often under reported during main 13 stream school years and may underlie mental health disorders. Additionally, comparisons between 14 younger and older schoolchildren may shed light on developmental differences in the way in which 15 physical conditions translate into conduct problems. The aim of the current study was to examine 16 the incidence of psychosomatic complaints (PSC) in young and older adolescent boys and girls who 17 also report conduct problems. 3,132 Swedish adolescents (age range 15-18 years, 47% boys) com 18 pleted the Uppsala Life and Health Cross-Sectional Survey (LHS) at school. LHS question scores 19 were categorised by two researchers who independently identified questions that aligned with 20 DSM-5 conduct disorder (CD) criteria and PSC. MANOVA assessed the effects of PSC, age, and 21 gender on scores that aligned with DSM criteria for CD. Main effects of gender; age; and PSC on 22 scores of conduct problem scores were observed. Adolescents with higher PSC scores had higher 23 conduct problem scores. Boys had higher serious violation of rules scores than girls, particularly 24 older boys with higher PSC scores. Psychosomatic complaints could be a useful objective identifier 25 for children and adolescents at risk of developing conduct disorders. This may be especially rele 26 vant when reliance on a child’s self-reporting of behavior may not help to prevent long-term dis 27 turbance to quality of life.

IMPORTANCE: The availability of behavior therapy for individuals with Tourette syndrome (TS) and chronic tic disorder (CTD) is limited. OBJECTIVE: To determine the efficacy and cost-effectiveness of internet-delivered exposure and response prevention (ERP) for children and adolescents with TS or CTD. DESIGN, SETTING, AND PARTICIPANTS: This single-masked, parallel group, superiority randomized clinical trial with nationwide recruitment was conducted at a research clinic in Stockholm, Sweden. Out of 615 individuals assessed for eligibility, 221 participants meeting diagnostic criteria for TS or CTD and aged 9 to 17 years were included in the study. Enrollment began in April 2019 and ended in April 2021. Data were analyzed between October 2021 and March 2022. INTERVENTIONS: Participants were randomized to 10 weeks of therapist-supported internet-delivered ERP for tics (111 participants) or to therapist-supported internet-delivered education for tics (comparator group, 110 participants). MAIN OUTCOMES AND MEASURES: The primary outcome was change in tic severity from baseline to the 3-month follow-up as measured by the Total Tic Severity Score of the Yale Global Tic Severity Scale (YGTSS-TTSS). YGTSS-TTSS assessors were masked to treatment allocation. Treatment response was operationalized as a score of 1 ("Very much improved") or 2 ("Much improved") on the Clinical Global Impression-Improvement scale. RESULTS: Data loss was minimal, with 216 of 221 participants (97.7%) providing primary outcome data. Among randomized participants (152 [68.8%] boys; mean [SD] age, 12.1 [2.3] years), tic severity improved significantly, with a mean reduction of 6.08 points on the YGTSS-TTSS in the ERP group (mean [SD] at baseline, 22.25 [5.60]; at 3-month follow-up, 16.17 [6.82]) and 5.29 in the comparator (mean [SD] at baseline, 23.01 [5.92]; at 3-month follow-up, 17.72 [7.11]). Intention-to-treat analyses showed that the 2 groups improved similarly over time (interaction effect, -0.53; 95% CI, -1.28 to 0.22; P = .17). Significantly more participants were classified as treatment responders in the ERP group (51 of 108 [47.2%]) than in the comparator group (31 of 108 [28.7%]) at the 3-month follow-up (odds ratio, 2.22; 95% CI. 1.27 to 3.90). ERP resulted in more treatment responders at little additional cost compared with structured education. The incremental cost per quality-adjusted life-year gained was below the Swedish willingness-to-pay threshold, at which ERP had a 66% to 76% probability of being cost-effective. CONCLUSIONS AND RELEVANCE: Both interventions were associated with clinically meaningful improvements in tic severity, but ERP led to higher response rates at little additional cost.