Genomics and precision medicine are being increasingly integrated into clinical practice. These technologies may lead to better diagnostic accuracy, earlier referral to appropriate treatment interventions and a better treatment response based on targeted use of drugs and other interventions. However, new generations of sequencing technologies are costly and can produce additional findings that result in further downstream healthcare costs. It is not yet clear whether such technologies are cost-effective or where they should be positioned in a diagnostic pathway to maximize value for money. TASK force members are working to generate evidence regarding economic value, including social and ethical considerations to inform implementation.

Table of contents

1. Cost and cost-effectiveness of whole genome sequencing in paediatric cardiac conditions
2. Cost and cost-effectiveness of alternative genome-based diagnostic strategies
3. The clinical and personal utility of genomic testing
4. Cascade genetic testing
5. Genome sequencing policy research
6. A cost-effectiveness analysis of maternal genotyping to guide treatment for postpartum pain and avert infant adverse events
7. Cost-effectiveness analysis of thiopurine methyltransferase genotype testing
8. Genetic counselling
9. Other

Cost and cost-effectiveness of whole genome sequencing in paediatric cardiac conditions

Prospective study led by Drs. Wendy Ungar and Robin Hayeems on clinical utility and cost-effectiveness of whole genome sequencing for children with cardiomyopathy. In progress.

• Jegathisawaran J, Tsiplova K, Hayeems R, Marshall CR, Stavropoulos DJ, Pereira SL, Thiruvahindrapuram B, Liston E, Reuter MS, Manshaei R, Cohn I, Jobling R, Kim RH, Mital S, Ungar WJ. Trio genome sequencing for developmental delay and pediatric heart conditions: A comparative microcost analysis. Genetics in Medicine. 2022; 24(5): 1027-1036.
• Jegathisawaran J, Tsiplova K, Hayeems R, Ungar WJ. Microcosting of whole genome sequencing (WGS) of trios in a heterogeneous pediatric cardiac population. Task Report No. 2020-01. 2020. Highlights, Executive Summary, Full Report.

Cost and cost-effectiveness of alternative genome-based diagnostic strategies in pediatric neurodevelopment disorders and rare diseases

• Hayeems RZ, Gillespie MK, Szuto A, Marshall C, Ungar WJ, Xiao B, Wu V, Li S, Stavropoulos DJ, Chisholm C, Costain G, Huang L, Beaulieu Bergeron M, Sawyer S, Mendoza-Londono R, Venkataramanan V, Lau L, Smith B, Higginbotham T, Hartley T, Somerville MJ, Boycott KM. Genome-wide Sequencing Ontario: A pilot implementation for rare disease diagnostics. Final report, May 31, 2023.
• Ungar WJ, Hayeems RZ, Marshall CR, Gillespie MK, Szuto A, Chisholm C, Stavropoulos DJ, Huang L, Jarinova O, Wu V, Tsiplova K, Lau L, Lee W, Venkataramanan V, Sawyer S, Mendoza-Londono R, Somerville MJ, Boycott KM and the Genome Sequencing Ontario Secondary Findings Study Team.   Protocol for a prospective observational cost-effectiveness analysis of returning secondary findings of genome sequencing for unexplained suspected genetic conditions. Clinical Therapeutics. 2023; 45(8):702-709.
• Hayeems RZ, Marshall CR, Gillespie MK, Szuto A, Chisholm C, Stavropoulos DJ, Price EM, Khan R, Lee W, Huang L, Jarinova O, Ungar WJ, Mendoza R, Somerville MJ, Boycott KM. Genome-wide Sequencing Ontario (GSO): An implementation pilot protocol to improve rare disease diagnostics. Canadian Medical Association Journal. 2022; 10(2): E460-465.
• Jegathisawaran J, Tsiplova K, Hayeems RZ, Marshall CR, Stavropoulos DJ, Pereira SL, Thiruvahindrapuram B, Liston E, Reuter MS, Manshaei R, Cohn I, Jobling R, Kim RH, Mital S, Ungar WJ. Trio genome sequencing for developmental delay and pediatric heart conditions: A comparative microcost analysis. Genetics in Medicine. 2022; 24(5): 1027-1036.
• Hayeems RZ, Michaels-Igbokwe C, Venkataramanan V, Hartley T, Acker M, Gillespie M, Ungar WJ, Mendoza-Londana R, Bernier F, Boycott KM, Marshall DA. The complexity of diagnosing rare disease: An organising framework for outcomes research and health economics based on real world evidence. Genetics in Medicine. 2022; 24(3): 694-702.
• Li C, Vandersluis S, Holubowich C, Ungar WJ, Goh ES, Boycott KM, Sikich N, Dhalla I, Ng V. Correspondence on “Cost or price of sequencing? Implications for economic evaluations in genomic medicine” by Grosse and Gudgeon. Genetics in Medicine. 2022; 24(1): 251-252.
• Li C, Vandersluis S, Holubowich C, Ungar WJ, Goh ES, Boycott KM, Sikich N, Dhalla I, Ng V. Cost-effectiveness of genome-wide sequencing for unexplained developmental disabilities and multiple congenital anomalies. Genetics in Medicine. 2021; 23(1): 451-460.
• Jegathisawaran J, Tsiplova K, Hayeems R, Ungar WJ. Determining precise costs for genomic sequencing technologies – A necessary prerequisite. Journal of Community Genetics, 2020; 11(2): 235-238.
• Yuen T, Penner M, Carter MT, Szatmari P, Ungar WJ. Cost-effectiveness of genome and exome sequencing in children diagnosed with autism spectrum disorder. Applied Health Economics & Health Policy, 2018; 16(4): 481-493.
• Jegathisawaran J, Tsiplova K, Ungar WJ. A microcosting and cost-consequence analysis of genomic testing strategies (including trios) in autism spectrum disorder. TASK Report No. 2018-01. 2018. Highlights, Executive Summary, Full Report, Appendices, Supplement, Fact Sheet
• Yuen T. Generating Evidence to Streamline the Clinical Pathway in Autism Spectrum Disorder Using Simulation Models: Cost-effectiveness Comparisons of Screening and Genetic Testing Strategies. Doctoral Dissertation. June 2017.
• Hayeems RZ, Bhawra J, Tsiplova K, Meyn MS, Monfared N, Bowdin S, Stavropoulos DJ, Marshall CR, Basran R, Shuman C, Ito S, Cohn I, Hum C, Girdea M, Brudno M, Cohn RD, Scherer SW, Ungar WJ. Care and cost consequences of pediatric whole genome sequencing compared to chromosome microarray. European Journal of Human Genetics. 2017; 25(12): 1303-1312.
• Oei K, Hayeems RZ, Ungar WJ, Cohn RD, Cohen E. Genetic testing among children in a complex care program. Children. 2017; 4(5): 42.
• Tsiplova K, Zur RM, Marshall CR, Stavropoulos DJ, Pereira SL, Merico D, Young EJ, Sung WWL, Scherer SW, Ungar WJ. A microcosting and cost-consequence analysis of genomic testing strategies in autism spectrum disorder. Genetics in Medicine. 2017; 19(11): 1268-1275.
• Tsiplova K. Zur RM, Ungar WJ. A microcosting and cost-consequence analysis of genomic testing strategies in developmental delay and autism spectrum disorder. TASK Report No. 2016-02.2. 2016. Highlights, Executive Summary, Full Report, Appendices, Supplement.

The clinical and personal utility of genomic testing

Prospective study led by Drs. Robin Hayeems and Wendy Ungar to develop and validate psychometric tools to quantify the clinical and personal utility of genetic tests. In Progress.

• Lee W, Hirjikaka D, Grewal S, Shaw A, Luca S, Clausen M, Bombard Y, Hayeems R, Navigator Study Team. Genetics providers’ perspectives on the use of digital tools in clinical practice. Genetics in Medicine. 2024; 26(6):101122.
• Marshall DA, Hua N, Buchanan J, Christensen KD, Frederix GWJ, Goranitis I, IJzerman MJ, Jansen JP, Lavelle TA, Regier DA, Smith HS, Ungar WJ, Weymann D, Wordsworth S, Phillips KA. Paving the path for implementation of clinical genomic sequencing globally – Are we ready? Health Affairs Scholar. 2024; 2(5):qxae053.
• Hayeems RZ, Luca S, Chad L, Quercia N, Xiao B, Hossain A, Meyn S, Pullenayegum E, Ungar WJ. Assessing the performance of the Clinician-reported Genetic testing Utility InDEx (C-GUIDE): Further evidence of inter-rater reliability. Clinical Therapeutics. 2023; 45(8):729-735.
• Lee W, Shickh S, Assamad D, Luca S, Clausen M, Somerville C, Tafler A, Shaw A, Hayeems RZ, Bombard Y, Genetics Navigator Study Team. Patient-facing digital tools for delivering genetic services: A systematic review. Journal of Medical Genetics. 2023; 60(1):1-10.
• Hayeems RZ, Luca S, Ungar WJ, Venkataramanan V, Tsiplova K, Bashir NS, Costain G, Inglese C, McNiven V, Quercia N, Shugar A, Yoon G, Cytrynbaum C, Dupuis L, Shao Z, Hewson S, Shuman C, Aul R, Liston E, Babul-Hirji R, Bushby A, Pullenayegum E, Chad L, Meyn MS. The Clinician-reported Genetic testing Utility InDEx (C-GUIDE): Preliminary evidence of validity and reliability. Genetics in Medicine. 2022; 24(2):430-438.
• Hayeems RZ, Luca S, Hurst ACE, Cochran M, Owens C, Hossain A, Chad L, Meyn MS, Pullenayegum E, Ungar WJ, Bick D.  Applying the Clinician-reported Genetic testing Utility InDEx (C-Guide) to genome sequencing: Further evidence of validity. European Journal of Human Genetics. 2022; 30(12):1423-1431.
• Hayeems RZ, Luca S, Assamad DA, Bhatt A, Ungar WJ. Utility of genetic testing from the perspective of parents/caregivers: A scoping review. Children, 2021; 8(4): 259.
• Hayeems RZ, Luca S, Ungar WJ, Bhatt A, Chad L, Pullenayegum E, Meyn MS. The development of the Clinician-reported Genetic testing Utility Index (C-GUIDE): A novel strategy for measuring the clinical utility of genetic testing. Genetics in Medicine. 2020; 22: 95-101.
• Hayeems RZ, Ungar WJ, Luca S, Pullenayegum E, Meyn MS. Genome diagnostics: Novel strategies for measuring value. Managed Care and Specialty Pharmacy. 2019; 25(10): 1096-1101.

Cascade genetic testing

• Cernat A, Hayeems RZ, Ungar WJ. Cascade health service use in family members following genetic testing in children: A scoping literature review. European Journal of Human Genetics. 2021; 29(1): 1601-1610.
• Cernat A, Hayeems RZ, Prosser LA, Ungar WJ. Incorporating cascade effects of genetic testing in economic evaluation: A scoping review of methodological challenges. Children. 2021; 8(5): 346.

Genome sequencing policy research

• Marshall DA, Hua N, Buchanan J, Christensen KD, Frederix GWJ, Goranitis I, IJzerman MJ, Jansen JP, Lavelle TA, Regier DA, Smith HS, Ungar WJ, Weymann D, Wordsworth S, Phillips KA. Paving the path for implementation of clinical genomic sequencing globally – Are we ready? Health Affairs Scholar. 2024; 2(5):.
• Cernat A, Bashir NS, Ungar WJ. Considerations for developing regulations for direct-to-consumer genetic testing: a scoping review using the 3-I Framework. Journal of Community Genetics, 2022; 13(2): 155-170.
• Bashir N, Ungar WJ. The 3-I Framework: A framework for developing public policies regarding pharmacogenomics (PGx) testing in Canada. Genome. 2015; 58(12): 527-540.
• Caulfield T, Evans J, McGuire A, McCabe C, Bubela T, Cook-Deegan R, Fishman J, Hogarth S, Miller FA, Raviskty V, Biesecker B, Borry P, Cho M, Carroll JC, Etchegary H, Joly Y, Kato K, Soo-Jin Lee S, Rothenberg K, Sankar P, Szego MJ, Ossorio P, Pullman D, Rousseau F, Ungar WJ, Wilson B. Reflections on the cost of “low-cost” whole genome sequencing: Framing the health policy debate. PLoS Biology. 2013; 11(11): e1001699.

A cost-effectiveness analysis of maternal genotyping to guide treatment for postpartum pain and avert infant adverse events

• Moretti ME, Lato DF, Berger H, Koren G, Ito S, Ungar WJ. A cost-effectiveness analysis of maternal genotyping to guide treatment for postpartum pain and avert infant adverse events. The Pharmacogenomics Journal. 2018; 18(3): 391-397.
• Moretti ME, Ungar WJ, Berger H, Koren G, Ito S. A cost-effectiveness analysis of maternal genotyping to guide treatment for postpartum pain and avert infant adverse events. TASK Report No. 2015-01. Highlights, Executive Summary, Full Report
• Moretti ME. A Cost-effectiveness Analysis of Maternal Genotyping to Guide Treatment for Postpartum Pain and Avert Infant Adverse Events. November 2014.

Cost-effectiveness analysis of thiopurine methyltransferase genotype testing

• Zur RM, Roy L, Ito S, Beyene J, Carew C, Ungar WJ. Thiopurine s-methyltransferase testing for averting drug toxicity: A meta-analysis of diagnostic test accuracy The Pharmacogenomics Journal. 2016; 16(4): 305-311.
• Zur RM, Roy LM, Ungar WJ. Thiopurine s-methyltransferase testing for averting drug toxicity in patients receiving thiopurines: A meta-analysis of diagnostic test accuracy. Toronto: The Hospital for Sick Children, October 2015. TASK Report No. 2015-03. Highlights, Executive Summary, Full Report, Appendices.
• Roy L, Zur RM, Ungar WJ. Thiopurine s-methyltransferase testing for averting drug toxicity in patients receiving thiopurines: A systematic review and quality appraisal. TASK Report No. 2015-02. Highlights, Executive Summary, Full Report, Appendices.
• Burnett HF, Tanoshima R, Chandranipapongse W, Madadi P, Ito S, Ungar WJ. Testing for thiopurine methyltransferase status for safe and effective administration of thiopurines: A systematic review of clinical guidance documents. The Pharmacogenomics Journal. 2014; 14(6):493-502.
• Burnett H, Chandranipapongse W, Tanoshima R, Ito S, Ungar WJ.  Thiopurine dosing using thiopurine methyltransferase status: A systematic review of clinical guidance. TASK Report No. 2013-02. Highlights, Executive Summary, Full Report, Appendices.
• Donnan JR, Ungar WJ, Mathews M, Rahman P. Systematic review of thiopurine methyltransferase genotype and enzymatic testing strategies. Therapeutic Drug Monitoring. 2011; 33:192–199.
• Donnan JR, Ungar WJ, Mathews M, Hancock-Howard RL, Rahman P. A cost effectiveness analysis of thiopurine methyltransferase testing for guiding 6-mercaptopurine dosing in children with acute lymphoblastic leukemia.Pediatric Blood & Cancer. 2011; 57:231-239.
• Donnan J, Ungar WJ, Mathews M, Hancock Howard R. Health technology assessment of thiopurine methyltransferase genetic testing for guiding 6-mercaptopurine doses in pediatric patients with acute lymphoblastic leukemia, TASK Report No. 2010-02. 2010. Highlights, Executive Summary, Full Report, Appendices.

Genetic counselling

• Luca S, Clausen M, Shaw A, Lee W, Krishnapillai S, Adi-Wauran E, Faghfoury H, Costain G, Jobling R, Aronson M, Liston E, Silver J, Shuman C, Chad L, Hayeems RZ Bombard, Y, Genetics Navigator Study Team. Finding the Sweet Spot: Exploring patients’ acceptability of chatbots in genetic service delivery. Human Genetics. 2023; 142(3): 321-330.
• Goh E S-Y, Miller FA, Marshall DA, Ungar WJ. Genetic counsellors’ preferences for coverage of preimplantation genetic diagnosis: A discrete choice experiment. Clinical Genetics. 2019; 95(6): 684-692.
• Goh, E.S. Genetic Counsellors’ Preferences for Preimplantation Genetic Diagnosis: A Discrete Choice Experiment. Master of Science Dissertation. June 2017.

Other

• Xie X, Wang M, Goh ES-Y, Ungar WJ, Little J, Carroll JC, Okun N, Huang T, Rousseau F, Dougan S, Tu HA, Higgins C, Holubowich C, Sikich N, Dhalla IA, Ng V. Noninvasive prenatal testing for trisomies 21, 18, and 13, sex chromosome aneuploidies, and microdeletions in average-risk pregnancies: A cost-effectiveness analysis. Journal of Obstetrics and Gynaecology Canada. 2020; 42(6): 740-749.

Children with neurodevelopment disorders (NDDs) and their families face life-long challenges and require extensive ongoing educational, health and social service support. The severity and high prevalence of NDDs place a high economic burden on parents and public service systems. It is crucial to understand which programs, treatments and services provide the best value for money. The TASK team is involved in a number of projects in the area of paediatric NDDs, collecting cost data and evaluating cost-effectiveness of existing and novel interventions.

Table of contents

1. Health economics evaluation platform within CHILD BRIGHT
2. PACE (Parent And Child Early)coaching project
3. Cost-effectiveness and value of alternative screening, assessment and treatment approaches in autism spectrum disorder
4. The pathways in autism spectrum disorder (ASD) cohort study
5. The pre-school autism treatment impact (PATI) study: A population-based comparative effectiveness, cost and policy study of intervention models for preschoolers with autism spectrum disorders

Health economics evaluation platform within CHILD BRIGHT

Dr. Ungar is leading economic evaluations of interventions being studied by researchers within the CHILD-BRIGHT Network, a pan-Canadian research network examining interventions to optimize the health of Canadian children born with brain-based disorders. In progress.

• Berrigan, P, Hicks GG, Ungar WJ, Zwicker JD. Budget impact analysis of an epigenetic test used for diagnosing fetal alcohol spectrum disorder from the perspective of a laboratory budget holder in Manitoba, Canada. Pharmacoeconomics Open. 2022; 6(2): 253-263.
• Orkin J, Major N, Esser K, Parmar A, Couture E, Daboval T, Kieran E, Ly L, O’Brien K, Patel H, Synnes A, Robson K, Barreira L, Smith WL, Rizakos S, Willan A, Yaskina M, Moretti ME, Ungar WJ, Ballantyne M, Church P, Cohen E. Coached, Coordinated, Enhanced Neonatal Transition (CCENT): Protocol for a multicentre pragmatic randomized controlled trial for parents of high-risk infants. BMJ Open. 2021; 11(7): e046706.
• Gorter JW, Amaria K, Kovacs A, Rozenblum R, Thabane L, Galuppi B, Nguyen L, Strohm S, Mahlberg N, Via-Dufresne Ley A, Marelli A, CHILD-BRIGHT READYorNotTM Brain-Based Disabilities Trial Study Group. CHILD-BRIGHT READYorNot Brain-Based Disabilities Trial: protocol of a randomised controlled trial (RCT) investigating the effectiveness of a patient-facing e-health intervention designed to enhance healthcare transition readiness in youth. BMJ Open. 2021; 11(3):e048756.
• Berrigan P, Hodge J, Kirton A, Moretti ME, Ungar WJ, Zwicker J. Protocol for a cost utility analysis of neurostimulation and intensive camp-based therapy for children with perinatal stroke and hemiparesis based on a multicenter clinical trial. BMJ Open. 2021; 11(1): e041444.

PACE (Parent And Child Early) coaching project

As part of a collaborative multi-disciplinary PACE Coaching Project, TASK leads a cost-effectiveness analysis comparing parent coaching to services ordinarily available to toddlers at risk for autism spectrum disorder in British Columbia.

• Tsiplova K, Jegathisawaran J, Mirenda P, Kalynchuk K, Colozzo P, Smith V, Ungar WJ. Parent coaching intervention for children with suspected autism spectrum disorder: Cost analysis. Research in Autism Spectrum Disorders. 2022; 93:101949.
• Mirenda P, Colozzo P, Smith V, Kroc E, Kalynchuk K, Rogers SJ, Ungar WJ. PACE coaching: A randomized, community-based feasibility trial of modified ESDM for toddlers with suspected autism. Journal of Autism and Developmental Disorders. 2022; 52(12): 5322-5341.
• Mirenda P, Smith V, Colozzo P, Vismara LA, Ungar WJ, Kalynchuk K. Training coaches in community agencies to support parents of children with suspected autism: Outcomes, facilitators, and barriers. Journal of Autism and Developmental Disorders. 2022; 52:4931-4948.

Cost-effectiveness and value of alternative screening, assessment and treatment approaches in autism spectrum disorder

• Tsiplova K, Ungar WJ. Why it is so challenging to perform economic evaluations of interventions in autism and what to do about it. Autism Research, 1-10, 2023.
• Ungar WJ, Tsiplova K. Economic evaluations of early intensive behavioral interventions for autism. Chapter 36, pp. 679-699 in Matson JL, Sturmey P (Eds), Handbook of Autism and Pervasive Developmental Disorders. Autism and Child Psychopathology Series. Springer Nature Switzerland AG: Basel, 2022.
• Penner M, Anagnostou E, Ungar WJ. Environmental scan of Canadian and UK policies for autism spectrum disorder diagnostic assessment. Paediatrics & Child Health. 2019; 24(3): e125-e134.
• Yuen T, Penner M, Carter MT, Szatmari P, Ungar WJ. Assessing the accuracy of the Modified Checklist for Autism in Toddlers: A systematic review and meta-analysis. Developmental Medicine and Child Neurology. 2018; 60(11): 1093-1100.
• Penner M, Anagnostou E, Ungar WJ. Practice patterns and determinants of wait time for autism spectrum disorder diagnosis in Canada. Molecular Autism, 2018; 9:16.
• Penner M, Anagnostou E, Ungar WJ. Systematic review of clinical guidance documents for autism spectrum disorder diagnostic assessment. Autism, 2018; 22(5): 517-527.
• Yuen T. Generating Evidence to Streamline the Clinical Pathway in Autism Spectrum Disorder Using Simulation Models: Cost-effectiveness Comparisons of Screening and Genetic Testing Strategies. Doctoral Dissertation, June 2017.
• Penner M. Policy Analysis and Evaluation of National Clinician-reported Practices for the Diagnosis of Autism Spectrum Disorder. Masters Dissertation, June 2016.
• Ungar WJ. Next generation sequencing and health technology assessment in autism spectrum disorder. Canadian Journal of Child and Adolescent Psychiatry. 2015; 24(2): 123-127.
• Motiwala S, Gupta S, Lilly M, Ungar WJ, Coyte PC. The cost-effectiveness of expanding Intensive Behavioural Intervention (IBI) to all autistic children in Ontario. Healthcare Policy. 2006; 1(2):135-151.

The pathways in autism spectrum disorder (ASD) cohort study

Using data collected by the Pathways in ASD cohort study, TASK is examining how child and youth health, education and community services are integrated to support optimal trajectories of developmental health.

• Chen Y-J, Duku E, Zaidman-Zait A, Szatmari P, Smith IM, Ungar WJ, Zwaigenbaum L, Kerns C, Vaillancourt T, Bennett T, Elsabbagh M, Thompson A, Georgiades S. Variable patterns of daily activity participation across settings in autistic youth: A latent profile transition analysis. Autism. 2023; 27(8):2241-2255.
• Tsiplova K, Ungar WJ, Szatmari P, Cost K, Pullenayegum E, Duku E, Volden J, Smith IM, Waddell C, Zwaigenbaum L, Bennett T, Elsabbagh M, Georgiades S, Zaidman-Zaid A. Measuring the association between behavioural services and outcomes in young children with autism spectrum disorder. Research in Developmental Disabilities. 2023; 132:104392.
• Courchesne V, Bedford R, Pickles A, Duku E, Kerns C, Mirenda P, Bennett T, Georgiades S, Smith IM, Ungar WJ, Vaillancourt T, Zaidman-Zait A, Zwaigenbaum L, Szatmari P, Elsabbagh M. Non-verbal IQ and change in restricted and repetitive behaviors throughout childhood in autism: A longitudinal study using the Autism Diagnostic Interview-Revised. Molecular Autism. 2021; 12(57).
• Ameis SH, Haltigan JD, Lyon RE, Sawyer A, Mirenda P, Kerns C, Smith IM, Vaillancourt T, Volden J, Waddell C, Zwaigenbaum L, Bennett T, Duku E, Elsabbagh M, Georgiades S, Ungar WJ, Zaidman-Zait A, Lai M-C, Szatmari P. Middle-childhood executive functioning mediates associations between preschool autism symptoms and adolescent mental health, academic and functional outcomes in autistic children. Journal of Child Psychology and Psychiatry. 2022; 63(5): 553-562.
• Georgiades S, Tait PA, McNicholas PD, Duku E, Zwaigenbaum L, Smith IM, Bennett T, Elsabbagh M, Kerns CM, Mirenda P, Ungar WJ, Vaillancourt T, Volden J, Waddell C, Zaidman-Zait A, Gentles S, Szatmari P. Trajectories of symptom severity in children with autism: Variability and turning points through the transition to school. Journal of Autism and Developmental Disorders. 2022; 52(1): 392-401.
• Szatmari P, Cost KT, Duku E, Bennett T, Elsabbagh M, Georgiades S, Kerns C, Mirenda P, Smith IM, Ungar WJ, Vaillancourt T, Waddell C, Zaidman-Zait A, Zwaigenbaum L. Association of child and family attributes with outcomes in children with autism. JAMA Network Open. 2021; 4(3): e212530.
• Cost KT, Zaidman-Zait A, Mirenda P, Duku E, Zwaigenbaum L, Smith IM, Ungar WJ, Kerns C, Bennett T, Szatmari P, Georgiades S, Waddell C, Elssabagh M, Vaillancourt T. “Best things”: Parents describe their children with autism spectrum disorder over time. Journal of Autism and Developmental Disorders. 2021; 51(1): 4560–4574.
• Baribeau DA, Vigod S, Pullenayegum E, Kerns CM, Mirenda P, Smith IM, Vaillancourt T, Volden J, Waddell C, Zwaigenbaum L, Bennett T, Duku E, Elsabbagh M, Georgiades S, Ungar WJ, Anat Zaidman-Zait, Szatmari P. Co-occurring trajectories of anxiety and insistence on sameness behavior in autism spectrum disorder. British Journal of Psychiatry. 2020; 218(1): 20-27.
• Zaidman-Zait A, Mirenda P, Szatmari P, Duku E, Smith IM, Zwaigenbaum L, Vaillancourt T, Kerns C, Volden J, Waddell C, Bennett T, Georgiades S, Ungar WJ, Elsabbagh M. Profiles and predictors of academic and social school functioning among children with autism spectrum disorder. Journal of Clinical Child and Adolescent Psychology. 2021; 50(5): 656-668.
• Lee V, Duku E, Zwaigenbaum L, Bennett T, Szatmari P, Elsabbagh M, Kerns C, Mirenda P, Smith IM, Ungar WJ, Vaillancourt T, Volden J, Waddell C, Zaidman-Zait-A, Thompson A, Georgiades S. Temperament influences the relationship between symptom severity and adaptive functioning in children with autism spectrum disorder. Autism. 2020; 24(8): 2057-2070.
• Hunsche MC, Saqui S, Mirenda P, Zaidman-Zait A, Bennett T, Duku E, Elsabbagh M, Georgiades S, Smith IM, Szatmari P, Ungar WJ, Vaillancourt T, Waddell C, Zwaigenbaum L, Kerns CM. Parent-reported rates and clinical correlates of suicidality in children with autism spectrum disorder: A longitudinal study. Journal of Autism and Developmental Disorders. 2020; 50(1): 3496-3509.
• Zaidman Zait A, Zwaigenbaum L, Duku E, Bennett T, Szatmari P, Mirenda P, Smith IM, Vaillancourt T, Volden J, Waddell C, Kerns C, Elsabbagh M, Georgiades S, Ungar WJ, Bryson S, Fombonne E, Roberts W. Factor analysis of the children’s sleep habits questionnaire among preschool children with autism spectrum disorder. Research in Developmental Disabilities. 2020; 97(1): 103548.
• Bal VH, Fok M, Lord C, Smith IM, Mirenda P, Szatmari P, Vaillancourt T, Volden J, Waddell C, Zwaigenbaum L, Bennett T, Duku E, Elsabbagh M, Georgiades S, Ungar WJ, Zaidman-Zait-A. Predictors of longer-term development of expressive language in two independent longitudinal cohorts of language-delayed preschoolers with ASD. Journal of Child Psychology and Psychiatry. 2020; 61(7), 826-835.
• Baribeau DA, Vigod S, Pullenayegum E, Kerns CM, Mirenda P, Smith IM, Vaillancourt T, Volden J, Waddell C, Zwaigenbaum L, Bennett T, Duku E, Elsabbagh M, Georgiades S, Ungar WJ, Anat Zaidman-Zait, Szatmari P. Repetitive behavior severity as an early indicator of risk for elevated anxiety symptoms in autism spectrum disorder. Journal of the American Academy of Child and Adolescent Psychiatry. 2020; 59(7): 890-899.
• Dempsey EE, Smith IM, Flanagan HE, Duku E, Lawrence MA, Szatmari P, Zwaigenbaum L, Vaillancourt T, Volden J, Mirenda P, Waddell C, Georgiades S, Elsabbagh M, Ungar WJ, Bennett T. Psychometric properties of the Merrill-Palmer-Revised Scales of Development in preschool children with autism spectrum disorder. Assessment. 2020; 27(8): 1796-1809.
• Di Rezze B, Duku E, Szatmari P, Volden J, Georgiades S, Zwaigenbaum L, Smith IM, Vaillancourt T, Bennett TA, Elsabbagh M, Thompson A, Ungar WJ, Waddell C, and Pathways in ASD Study Team. Examining trajectories of daily living skills over the preschool years for children with autism spectrum disorder. Journal of Autism and Developmental Disorders. 2019; 49(11): 4390-4399.
• Bal VH, Fok M, Lord C, Smith IM, Mirenda P, Szatmari P, Vaillancourt T, Volden J, Waddell C, Zwaigenbaum L, Bennett T, Duku E, Elsabbagh M, Georgiades S, Ungar WJ, Zaidman-Zait-A. Predictors of longer-term development of expressive language in two independent longitudinal cohorts of language-delayed preschoolers with ASD. Journal of Child Psychology and Psychiatry. 2020; 61(7): 826-835.
• Baribeau DA, Vigod S, Pullenayegum E, Kerns CM, Mirenda P, Smith IM, Vaillancourt T, Volden J, Waddell C, Zwaigenbaum L, Bennett T, Duku E, Elsabbagh M, Georgiades S, Ungar WJ, Anat Zaidman-Zait, Szatmari P. Repetitive behavior severity as an early indicator of risk for elevated anxiety symptoms in autism spectrum disorder. Journal of the American Academy of Child and Adolescent Psychiatry. 2020; 59(7): 890-899.
• Peverill S, Smith I, Duku E, Szatmari P, Mirenda P, Vaillancourt T, Volden J, Zwaigenbaum L, Bennett T, Elsabbagh M, Georgiades S, Ungar WJ. Developmental trajectories of feeding problems in children with autism spectrum disorder. Journal of Pediatric Psychology. 2019; 44(8): 988-998.
• Conlon O, Volden J, Smith IM, Duku E, Zwaigenbaum L, Waddell C, Szatmari P, Mirenda P, Vaillancourt T, Bennett T. Georgiades S, Elsabbagh M, Ungar WJ. Gender differences in pragmatic communication in school-aged children with autism spectrum disorder (ASD). Journal of Autism and Developmental Disorders. 2019; 49(5): 1937-1948.
• Dempsey EE, Smith IM, Flanagan HE, Duku E, Lawrence MA, Szatmari P, Zwaigenbaum L, Vaillancourt T, Volden J, Mirenda P, Waddell C, Georgiades S, Elsabbagh M, Ungar WJ, Bennett T. Psychometric properties of the Merrill-Palmer-Revised Scales of Development in preschool children with autism spectrum disorder. Assessment. 2018; 27(8).
• Zaidman Zait A, Mirenda P, Szatmari P, Duku E, Smith IM, Vaillancourt T, Volden J, Waddell C, Bennett T, Zwaigenbaum L, Elsabaggh M, Georgiades S, Ungar WJ. Profiles of social and coping resources in families of children with autism spectrum disorder: Relations to parent and child outcomes. Journal of Autism and Developmental Disorders. 2018; 48(6): 2064-2076.

The pre-school autism treatment impact (PATI) study: A population-based comparative effectiveness, cost and policy study of intervention models for preschoolers with autism spectrum disorders

A multi-disciplinary and multi-site prospective study, where TASK is leading a cost analysis of the New Brunswick and Nova Scotia early intensive behavioural programs for preschoolers with autism spectrum disorder.

• PATI Team Study. Information Brief #1, Preschoolers with Autism: Comparing Early Interventions in Nova Scotia and New Brunswick. 2020.
• PATI Team Study. Information Brief #2, Preschoolers with Autism: Comparing Service Use and Costs in Nova Scotia and New Brunswick. 2020.
• D’Entremont B, Flanagan HE, Ungar WJ, Waddell C, Garon N, den Otter J, Leger N, Vezina F, Smith IM. Comparing the impact of differing preschool autism interventions on parents in two Canadian provinces. Journal of Autism and Developmental Disorders. 2022; 52:5018-5032.
• Smith IM, Waddell C, Ungar WJ, den Otter J, Murray P, Vezina F, D’Entremont B, Flanagan H, Garon N. Preschool Autism Services: A Tale of Two Canadian Provinces and the Implications for Policy. Paediatrics and Child Health. 2021; 26(3): 145-148
• Tsiplova K, Ungar WJ, Flanagan HE, den Otter J, Waddell C, Murray P, D’Entremont B, Garon N, Léger N, Bryson SE, Smith IM. Types of services and costs of programs for preschoolers with autism spectrum disorder across sectors: A comparison of two Canadian provinces. Journal of Autism and Developmental Disorders. 2019; 49(6): 2492-2508.
• Smith IM, Flanagan HE, Ungar WJ, D’Entremont B, Garon N, den Otter J, Waddell C, Bryson SE, Tsiplova K, Léger, N, Vezina F, Murray P. Comparing the one-year impact of preschool autism intervention programs in two Canadian provinces. Autism Research. 2019; 12: 667-681.
• Ungar WJ, Tsiplova K, Millar N, Smith IM. Development of the Resource Use Questionnaire (RUQ-P) for families with preschool children with neurodevelopmental disorders: Validation in children with autism spectrum disorder. Clinical Practice in Pediatric Psychology. 2018; 6(2): 164-178.

Health technology assessment (HTA) provides evidence for informed decision making in health policy. As part of its HTA activities, TASK informs policies in child health. TASK also conducts research specifically in the area of public health and pharmaceutical policy. For example, the TASK team has examined gaps in prescription drug coverage for paediatric drugs and is examining the value of pharmacogenomics testing.

Table of contents

1. Public health and policy decision-making
2. Prescription medicines

Public health and policy decision-making

• Gauvreau CL, Schreyer L, Gibson P, Koo A, Ungar WJ, Regier D, Chan K, Hayeems R, Gibson J, Palmer A, Peacock S, Denburg AE. Development of a value assessment framework for pediatric health technologies using multi-criteria decision analysis: expanding the value lens for funding decision-making. Value Health. 2024; 27(7): 879-888.
• Bashir NS,  Hughes A, Ungar WJ. Infliximab pricing in international economic evaluations in inflammatory bowel disease to inform biologic and biosimilar access policies. Medical Decision Making Policy & Practice. 2023; 8(1):1-22.
• Denburg AE, Giacomini M, Ungar WJ, Abelson J. The moral foundations of child health and social policies: A critical interpretive synthesis. Children. 2021; 8(1): 43.
• Denburg AE, Giacomini M, Ungar WJ, Abelson J. Ethical and social values for paediatric health technology assessment and drug policy. International Journal of Health Policy and Management. 2022: 11(3): 374-382.
• Xie X, Gajic-Veljanoski O, Falk L, Schaink AK, Lambrinos A, Wang M, Ng V, Ungar WJ, Sikich S. Challenges in health technology assessments of genetic tests.  Journal of Hospital Management and Health Policy. 2020; 4:27.
• Denburg AE, Ungar WJ, Chen S, Hurley J Abelson J. Does moral reasoning influence public values for health care priority setting? A population-based randomized stated preference survey. Health Policy. 2020; 124(6): 647-658.
• Ungar WJ, Denburg AE. An international perspective on ethical considerations in health economic evaluation: A primer for pediatric psychologists. Clinical Practice in Pediatric Psychology, 2018; 6(2); 190-200.
• Bashir N, Ungar WJ. The 3-I Framework: A framework for developing public policies regarding pharmacogenomics (PGx) testing in Canada. Genome. 2015; 58(12): 527-540.
• Caulfield T, Evans J, McGuire A, McCabe C, Bubela T, Cook-Deegan R, Fishman J, Hogarth S, Miller FA, Raviskty V, Biesecker B, Borry P, Cho M, Carroll JC, Etchegary H, Joly Y, Kato K, Soo-Jin Lee S, Rothenberg K, Sankar P, Szego MJ, Ossorio P, Pullman D, Rousseau F, Ungar WJ, Wilson B. Reflections on the cost of “low-cost” whole genome sequencing: Framing the health policy debate. PLoS Biology. 2013; 11(11): e1001699.
• Gauvreau C, Ungar WJ, Köhler JC, Zlotkin S. The use of cost-effectiveness analysis for pediatric immunization in developing countries. The Milbank Quarterly. 2012; 90(4):762-790.
• Sharieff W, Zlotkin S, Ungar WJ, Feldman BM, Krahn M, Tomlinson G. Economics of preventing premature mortality and impaired cognitive development in children through home-fortification: A health policy perspective. International Journal of Technology Assessment and Health Care. 2008; 24(3):303-311.
• Ungar WJ, Kozyrskyj A, Paterson M, Ahmad F. Effect of cost-sharing on asthma medication use in children. Archives of Pediatrics & Adolescent Medicine. 2008; 162(2):104-110.
• Han RK, Ungar WJ, Macarthur C. Cost effectiveness analysis of a proposed public health legislative/educational strategy to reduce tap water scald injuries in children. Injury Prevention. 2007; 13(4): 248-253.
• Piwko C, Ungar WJ, Einarson TR, Wolping J, Koren G. The weekly cost of nausea and vomiting of pregnancy for women calling the Toronto Motherisk program. Current Medical Research & Opinion. 2007; 23(4):833-840.
• Kopach R, Sadat S, Galloway I, Coyte PC, Ungar WJ. A cost-effectiveness analysis of medical documentation alternatives. International Journal of Technology Assessment in Health Care. 2005; 21(1):126-131.
• To T, Ungar W. Outpatient Health Services Use By Children in Ontario. Institute for Clinical Evaluative Sciences (ICES) Atlas Report. Uses of Health Services: Report No. 5. Institute for Clinical Evaluative Sciences, Toronto, ISBN 0-9686318-6-X, March 2001.

Prescription medicines

• Denburg AE, Giacomini M, Ungar WJ, Abelson J. The problem is small enough, the problem is big enough’: a qualitative study of health technology assessment and public policy on drug funding decisions for children. International Journal for Equity in Health. 2020; 19(1): 1-16
• Ungar WJ. Section 5.4: Publically-Funded Drug Coverage. In Guttmann A, Gandhi S, Hanvey Li P, Barwick M, Cohen E, Glazer S, Reisman J, Brownell M. Primary Health Care Services for Children and Youth in Canada: Access, Quality and Structure. In The Health of Canada’s Children and Youth: A CICH Profile. 2017.
• Denburg AE, Ungar WJ, Greenberg M. Public drug policy for children in Canada, Canadian Medical Association Journal. 2017; 189(30): E990-E994.
• Council of Canadian Academies. Improving Medicines for Children in Canada. Ottawa (ON): The Expert Panel on Therapeutic Products for Infants, Children, and Youth, Council of Canadian Academies. 2014.
• Ungar WJ, Paterson M, Cope S, Kozyrskyj A. Improving drug benefits for children with asthma: Building a research agenda. Healthcare Policy. 2008; 3(4):66-76.
• Ungar WJ, Kozyrskyj A, Paterson M, Ahmad F. Effect of cost-sharing on asthma medication use in children. Archives of Pediatrics & Adolescent Medicine. 2008; 162(2):104-110.
• Ungar WJ, Witkos M. Public drug plan coverage for children across Canada: A portrait of too many colours. Healthcare Policy. 2005; 1(1):100-122.
• Ungar WJ, Ariely R. Health insurance, access to prescription medicines and health outcomes in children. Expert Review of Pharmacoeconomics & Outcomes Research. 2005; 5(2):215-225.
• Ungar WJ, Daniels C, McNeill T, Seyed M. Children in need of Pharmacare: Medication funding requests at the Toronto Hospital for Sick Children. Canadian Journal of Public Health. 2003; 94(2):121-126.

TASK’s projects in health economic methods include patient preferences research; development and assessment of instruments to measure resource use, costs or quality of life; reviews of economic evaluation methods; and the use and applicability of the PEDE database and other economic evaluation databases for decision-making.

Table of contents

1. Caregiver and spillover effects
2. Health economic methods
3. Instrument and tool development
4. Quality of life and utility research
5. Preferences and preference elicitation techniques
6. Systematic reviews
7. Trends in evidence

Caregiver and spillover effects

• Lamsal R, Yeh A, Pullenayegum E, Ungar W. A systematic review of methods and practice for integrating maternal, fetal and child health outcomes, and family spillover effects into cost-utility analyses. PharmacoEconomics. 2024; 42:843-863.
• Lamsal R, Yeh A, Pullenayegum E, Ungar WJ. A sytematic review of methods used by pediatric cost-utility analyses to include family health spillover effects. PharmacoEconomics. 2024; 42(2):199-217.
• Henry E, Al-Janabi H, Brouwer W, Cullinan J, Engel L, Griffin S, Hulme C, Kingkaew P, Lloyd A, Payakachat N, Pennington B, Peña-Longobardo LM, Prosser LA, Shah K, Ungar WJ, Wilkinson T, Wittenberg E. Spillovers in Health Economic Evaluation and Research (SHEER) Task Force: Emerging good practice and recommendations for a future research agenda. PharmacoEconomics, 2024; 42(3):343-362.
• Belza C, Avitzur Y, Ungar WJ, Stremler R, Fehlings D, Wales PW.  Stress, anxiety and health-related quality of life in caregivers of children with intestinal failure on parenteral nutrition: A cross-sectional survey study. Journal of Parenteral and Enteral Nutrition. 2023; 47(3):342-353.
• Lin E, Durbin J, Ungar W, Lero D, Volpe T, Selick A. Measuring the family/friend caregiver experience in Ontario: Pilot study results. 2021.
• Lamsal R, Ungar WJ. Impact of growing up with a sibling with a neurodevelopmental disorder on the quality of life of an unaffected sibling: A scoping review. Disability and Rehabilitation, 2021; 43(4): 586-594.
• Lavelle TA, D’Cruz B, Mohit B, Ungar WJ, Prosser LA, Neumann PJ, Tsiplova K, Vera-Llonch M, Lin P-J. Family spillover effects in pediatric cost-utility analyses. Applied Health Economics and Health Policy. 2019; 17(2): 163-174.
• Tsimicalis A, Stevens B, Ungar WJ, Castro A, Greenberg M, Barr B. Shifting priorities for the survival of my child: Managing expenses, increasing debt and tapping into available resources to maintain the financial stability of the family. Cancer Nursing. 2019; 43(2): 147-157.
• Lin E, Durbin J, Guerriere D, Volpe T, Selick A, Kennedy J, Ungar WJ, Lero DS. Assessing care-giving demands, resources and costs of family/friend caregivers for persons with mental health disorders: A scoping review. Health & Social Care in the Community. 2018; 26: 613– 634.
• Ungar WJ. Section 5.4: Publically-Funded Drug Coverage. In Guttmann A, Gandhi S, Hanvey Li P, Barwick M, Cohen E, Glazer S, Reisman J, Brownell M. Primary Health Care Services for Children and Youth in Canada: Access, Quality and Structure. In The Health of Canada’s Children and Youth: A CICH Profile. 2017.
• Lin E, Durbin J, Lero DS, Selick A, Volpe T, Ungar WJ, Vu N. Measuring the Family/Friend Caregiver Experience in Ontario: Pilot Study Results Technical Report. Health System Performance Research Network (HSPRN), University of Toronto, November 15, 2016.
• Tsimicalis A, Stevens B, Ungar WJ, Greenberg M, McKeever P, Agha M, Guerriere D, Barr R, Naqvi A, Moineddin R. Determining the costs of families’ support networks following a child’s cancer diagnosis. Cancer Nursing: An International Journal for Cancer Care. 2013; 36(2):E8-E19.
• Tsimicalis A, Stevens B, Ungar WJ, McKeever P, Greenberg M, Agha M, Guerriere D, Naqvi A, Barr R. A mixed method approach to describe the out-of-pocket expenses incurred by families of children with cancer. Pediatric Blood and Cancer. 2013; 60(3):438-445.
• Guerriere DN, Tranmer JE, Ungar WJ, Manoharan V, Coyte PC. Valuing care recipient and family caregiver time: A comparison of methods. International Journal of Technology Assessment in Health Care. 2008; 24(1):52-59.
• Tranmer J, Guerriere D, Ungar WJ, Coyte PC. Valuing patient and caregiver time: A review of the literature. Pharmacoeconomics. 2005; 23(5):449-459.

Health economic methods

• Tsiplova K, Ungar WJ. Why it is so challenging to perform economic evaluations of interventions in autism and what to do about it. Autism Research, 1-10, 2023.
• Xie X, Schaink AK, Gao C, Gajic-Veljanoski O, Ungar WJ, Volodin A. Evaluating the correlations of cost and utility parameters from the summary statistics for probabilistic analysis in economic evaluations. Expert Review of Pharmacoeconomics & Outcomes Research, 23(8):901-909, 2023.
• Subasri M, Cressman C, Arje D, Schreyer L, Cooper E, Patel K, Ungar WJ, Barwick M, Denburg A, Hayeems RZ. Translating precision health for pediatrics: A scoping review. Children. 2023; 10(5):897.
• Xie X, Gajic-Veljanoski O, Ungar WJ, Hussain S, Tu HA. Modeling methods and the degree of parameter uncertainty in probabilistic analyses of economic evaluations. Network Modeling Analysis in Health Informatics and Bioinformatics. 2023; 12(1):9.
• Xie X, Tiggelaar S, Wang M, Vandersluis S, Ungar WJ. Developing economic models for assessing the cost-effectiveness of multiple diagnostic tests: Methods and Applications. Medical Decision Making. 2022; 42(7): 861-871.
• Ungar WJ. Assessing value in child health. Children. 2021; 8(11): 972.
• Bashir NS, Walters TD, Griffiths AM, Ito S, Ungar WJ. Imputing missing patient-level data and propensity score matching in cost-effectiveness analysis. Expert Review of Pharmacoeconomics & Outcomes Research. 2022; 22(3): 442-454.
• Cernat A, Hayeems RZ, Prosser LA, Ungar WJ. Incorporating cascade effects of genetic testing in economic evaluation: A scoping review of methodological challenges. Children. 2021, 8(5): 346.
• Ungar WJ, Zur RM. Costs and consequences: An introduction to economic evaluation in radiology. In Doria AS, Tomlinson G, Beyene J and Moineddin R (Eds) Research Methods in Radiology. New York: Theme Medical Publishers Inc., 2017.
• Ungar WJ. Technology Assessment at Sick Kids (TASK): A Health Technology Assessment Research Unit devoted to Child Health. In Sampietro-Colom L, Martin J (Eds), Hospital-based Health Technology Assessment. New York: Springer Press, 2017.
• Ungar WJ. A further examination of the problem of double-counting in incremental cost-utility analyses. Expert Review of Pharmacoeconomics and Outcomes Research. 2016; 16(3): 333-335.
• Zlotnik Shaul R, Ungar WJ. Maximizing the benefit and mitigating the risks of moral hazard. American Journal of Bioethics. 2016; 16(7):44-6.
• Ungar WJ, Zur RM. Health economic evaluation for improving child health in low- and middle-income countries. In MacLeod S, Hill S, Rane A, Koren G (Eds), Challenges in Optimizing Drug Treatment for Children in the Developing World. New York: Springer Press, 2015.
• Ungar WJ, Prosser LA, Burnett H. Values and evidence colliding: Health technology assessment in child health. Expert Review of Pharmacoeconomics and Outcomes Research. 2013; 13(4):417-419.
• Ungar WJ (Editor). Economic Evaluation in Child Health. Oxford: Oxford University Press, 2010.
• Ungar WJ. Preface, in Ungar WJ (ed) Economic Evaluation in Child Health. Oxford: Oxford University Press, 2010.
• Ungar WJ, Gerber A. The uniqueness of child health and challenges to measuring costs and consequences, in Ungar WJ (ed) Economic Evaluation in Child Health. Oxford: Oxford University Press, 2010.
• Costa V, Ungar WJ, Health technology assessment in child health, in Ungar WJ (ed) Economic Evaluation in Child Health. Oxford: Oxford University Press, 2010.
• Ungar WJ. Paediatric health economic evaluations: A world view. Healthcare Quarterly -World Health & Population. 2006; 8(4): 89-101.
• Ungar WJ. Gesundheitsökonomische Analysen in der Pädiatrie: Historischer Überblick und Untersuchung nach Art der Interventionen. In: Gerber A & Lauterbach KW (Eds). Gesundheitsökonomie und Pädiatrie. Stuttgart: Schattauer Publishers. September 2006.
• Ungar WJ, Santos MT. Quality appraisal of pediatric health economic evaluations. International Journal of Technology Assessment and Health Care. 2005; 21(2):203-210.
• Ungar W. Bias – it’s everywhere! A commentary on the impact of bias and the assessment of agreement in the measurement of medication use in epidemiology research. Pharmacoepidemiology and Drug Safety. 1998; 7:425-427.
• Ungar W. The choice of pharmacoeconomic study design during drug development, Part 1. Pharmacoepidemiology and Drug Safety. 1997; 6: 391-397.
• Ungar W. The choice of pharmacoeconomic study design during drug development, Part 2. Pharmacoepidemiology and Drug Safety. 1997; 6(6): 399-407.
• Ungar W. Good Clinical Practices: Principles and application of the European Community note for guidance. Drug Information Journal. 1994; 28:339-348.

Instrument and tool development

• Gauvreau CL, Schreyer L, Gibson P, Koo A, Ungar WJ, Regier D, Chan K, Hayeems R, Gibson J, Palmer A, Peacock S, Denburg AE. Development of a value assessment framework for pediatric health technologies using multi-criteria decision analysis: expanding the value lens for funding decision-making. Value in Health. 2024; 27(7): 879-888.
• Butcher, NJ. Monsour A, Mew EJ, Chan A-W, Moher D, Mayo-Wilson E, Terwee CB, Chee-A-Tow A, Baba A, Gavin F, Grimshaw J, Kelly LE, Saeed L, Thabane L, Askie L, Smith M, Farid-Kapadia M, Williamson P, Szatmari P, Tugwell P, Golub RM, Monga S, Vohra S, Marlin S, Ungar WJ, Offringa M. Guideline for reporting outcomes in trial protocols: The SPIRIT-Outcomes 2022 extension.  JAMA. 2022; 328(22): 2345-22356.
• Butcher NJ, Monsour A, Mew EJ, Chan A-W, Moher D, Mayo-Wilson E, Terwee CB, Chee-A-Tow A, Baba A, Gavin F, Grimshaw J, Kelly LE, Saeed L, Thabane L, Askie L, Smith M, Farid-Kapadia M, Williamson P, Szatmari P, Tugwell P, Golub RM, Monga S, Vohra S, Marlin S, Ungar WJ, Offringa M. Guidelines for reporting outcomes in trial reports: The CONSORT-Outcomes 2022 extension. JAMA. 2022; 328(22): 2252-2264.
• Butcher N, Monsour A, Mew EJ, Szatmari P, Pierro A, Kelly LE, Farid-Kapadia M, Chee-a-tow A, Saeed L, Monga S, Ungar W, Terwee CB, Vohra S, Fergusson D, Askie LM, Williamson PR, Chan A-W, Moher D, Offringa M. Improving outcome reporting in clinical trial reports and protocols: Study protocol for the Instrument for reporting Planned Endpoints in Clinical Trials (InsPECT). Trials, 20(1): 161, 2019.
• Ungar WJ, Tsiplova K, Millar N, Smith IM. Development of the Resource Use Questionnaire (RUQ-P) for families with preschool children with neurodevelopmental disorders: Validation in children with autism spectrum disorder. Clinical Practice in Pediatric Psychology. 2018; 6(2): 164-178.
• Guerriere DN, Ungar WJ, Corey M, Croxford R, Tranmer JE, Tullis E, Coyte PC. Evaluation of the Ambulatory and Home Care Record: Agreement between self-reports and administrative data. International Journal of Technology Assessment in Health Care. 2006; 22(2):203-10.
• Davidson-Grimwood SR, Snelgrove D, Ungar WJ. The development of an interview guide for use in dyad child-parent health related quality of life assessment. PRO Newsletter (peer-reviewed section). 2006; 37:12-14.
• Koren G, Piwko C, Ahn E, Boskovic R, Maltepe C, Navioz Y, Ungar WJ. Validation studies of the pregnancy unique-quantification of emesis (PUQE) scores. Journal of Obstetrics & Gynecology. 2005; 25(3): 241-244.
• Ungar WJ, Santos MT. The Pediatric Economic Database Evaluation (PEDE) project: Establishing a database to study trends in pediatric economic evaluation. Medical Care. 2003; 41(10):1142-1152.
• Ungar WJ, Santos MT. The Pediatric Quality Appraisal Questionnaire (PQAQ): An instrument for evaluation of the pediatric health economics literature. Value in Health. 2003; 6(5):584-593.
• Ungar W, Santos M. The Pediatric Economic Database Evaluation (PEDE) project. Ottawa: Canadian Coordinating Office for Health Technology Assessment (CCOHTA); 2002. Technology Report no 26.
• Ungar WJ, Josse R, Lee S, Ryan N, Adachi J, Hanley D, Breton M-C, Brown J. The Canadian SCORE questionnaire – optimizing the use of technology for low bone density assessment. Journal of Clinical Densitometry. 2000; 3(3):269-280.

Quality of life and utility research

• Ungar WJ, Herdman M. Meeting the challenges of preference-weighted health-related quality of life measurement in children. PharmacoEconomics. 2024; 42(Suppl 1):3-8.
• Xie X, Schaink AK, Gao C, Gajic-Veljanoski O, Ungar WJ, Volodin A. Evaluating the correlations of cost and utility parameters from the summary statistics for probabilistic analysis in economic evaluations. Expert Review of Pharmacoeconomics & Outcomes Research, 23(8):901-909, 2023.
• Bashir NS, Walters TD, Griffiths AM, Otley A, Critch J, Ungar WJ. A comparison of the Child Health Utility 9D and the Health Utilities Index for estimating health utilities in pediatric inflammatory bowel disease. Quality of Life Research, 32(9):2527-2539, 2023.
• Bashir NS, Walters TD, Griffiths AM, Otley A, Critch J, Ungar WJ. A comparison of preference-based, generic and disease-specific health-related quality of life in pediatric inflammatory bowel disease. Journal of the Canadian Association of Gastroenterology. 2023; 6(2):73-79.
• Belza C, Avitzur Y, Ungar WJ, Stremler R, Fehlings D, Wales PW.  Stress, anxiety and health-related quality of life in caregivers of children with intestinal failure on parenteral nutrition: A cross-sectional survey study. Journal of Parenteral and Enteral Nutrition. 2023; 47(3):342-353.
• Bashir NS, Walters TD, Griffiths AM, Ungar WJ. An assessment of the validity and reliability of the pediatric Child Health Utility 9D in children with inflammatory bowel disease. Children. 2021; 8(5): 343.
• Oliveira C, de Silva NT, Ungar WJ, Bayoumi AM, Avitzur Y, Hoch JS, Maxwell J, Wales PW. Health-related quality of life in neonates and infants: A conceptual framework. Quality of Life Research. 2020; 29(5): 1159-1168.
• Lamsal R, Ungar WJ. Impact of growing up with a sibling with a neurodevelopmental disorder on the quality of life of an unaffected sibling: A scoping review. Disability and Rehabilitation. 2021; 43(4): 586-594.
• Kwon J, Kim SW, Ungar WJ, Tsiplova K, Madan J, Petrou S. Patterns, trends and methodological associations in the measurement and valuation of childhood health utilities. Quality of Life Research. 2019; 28(7): 1705-1724.
• Kwon J, Kim SW, Ungar WJ, Tsiplova K, Madan J, Petrou S. A systematic review and meta-analysis of childhood health utilities. Medical Decision Making. 2018; 38(3): 277-305.
• Ungar WJ. A further examination of the problem of double-counting in incremental cost-utility analyses. Expert Review of Pharmacoeconomics and Outcomes Research. 2016; 16(3): 333-335.
• Kromm SK, Bethell J, Kraglund F, Edwards S, Laporte A, Coyte PC, Ungar WJ. Characteristics and quality of pediatric cost-utility analyses. Quality of Life Research. 2012; 21(8):1315-1325.
• Ungar WJ. Understanding the value of information from pediatric clinical research. Pediatric Drugs. 2012; 14(5):295-297.
• Ungar WJ, Boydell K, Dell S, Feldman BM, Marshall D, Willan A, Wright J. A parent-child dyad approach to the assessment of health status and health-related quality of life in children with asthma. PharmacoEconomics. 2012; 30(8):697-712.
• Sung L, Petrou S, Ungar WJ, Measurement of health utilities in children, in Ungar WJ (ed) Economic Evaluation in Child Health. Oxford: Oxford University Press, 2010.
• Ungar WJ, Mirabelli C, Cousins M, Boydell KM. A qualitative analysis of a dyad approach to quality of life measurement in children with asthma. Social Science & Medicine. 2006; 63(9):2354-66.
• Ungar WJ. Challenges in health state valuation in pediatric economic evaluation: Are QALYs contraindicated? PharmacoEconomics. 2011; 29(8):641-652.

Preferences and preference elicitation techniques

• Luca S, Clausen M, Shaw A, Lee W, Krishnapillai S, Adi-Wauran E, Faghfoury H, Costain G, Jobling R, Aronson M, Liston E, Silver J, Shuman C, Chad L, Hayeems RZ Bombard, Y, Genetics Navigator Study Team. Finding the Sweet Spot: Exploring patients’ acceptability of chatbots in genetic service delivery. Human Genetics. 2023; 142(3): 321-330.
• Denburg AE, Ungar WJ, Chen S, Hurley J Abelson J. Does moral reasoning influence public values for health care priority setting? A population-based randomized stated preference survey. Health Policy. 2020; 124(6): 647-658.
• Goh E S-Y, Miller FA, Marshall DA, Ungar WJ. Genetic counsellors’ preferences for coverage of preimplantation genetic diagnosis: A discrete choice experiment. Clinical Genetics. 2019; 95(6): 684-692.
• Goh, E.S. Genetic Counsellors’ Preferences for Preimplantation Genetic Diagnosis: A Discrete Choice Experiment. Master of Science Dissertation. June 2017.
• Ungar WJ, Hadioonzadeh A, Najafzadeh M, Tsao NW, Dell S, Lynd LD. Preferences for asthma control in parents and adolescents using a best-worst scaling experiment. Respiratory Medicine. 2014; 108(6):842-851.
• Ungar WJ, Hadioonzadeh A, Najafzadeh M, Tsao NW, Dell S, Lynd LD. Parents and adolescents preferences for asthma control: A best-worst scaling choice experiment using an orthogonal main effects design. BMC Pulmonary Medicine. 2005; 15:146.
• Azarpazhooh A, Dao T, Ungar WJ, Da Costa J, Figueiredo R, Krahn M, Friedman S. Patients’ values related to treatment options for teeth with apical periodontitis. Journal of Endodontics. 2016; 42(3): 365-70.
• Azarpazhooh A, Dao T, Ungar WJ, Chaudry F, Figueiredo R, Krahn M, Friedman S. Clinical decision-making for a tooth with apical periodontitis: the patients’ preferred level of participation. Journal of Endodontics. 2014; 40(6):784-789.
• Regier DA, Watson V, Burnett H, Ungar WJ. Task complexity and response certainty in discrete choice experiments: An application to drug treatments for juvenile idiopathic arthritis. Journal of Behavioral and Experimental Economics. 2014; 50:40-49.
• Burnett HF, Regier DA, Feldman BM, Miller FA, Ungar WJ. Parents’ preferences for drug treatments in juvenile idiopathic arthritis: A discrete choice experiment. Arthritis Care & Research. 2012; 64(9):1382-1391.
• Hancock-Howard RL, Ungar WJ, Marshall D, Einarson A, Koren G. Public preferences for counseling regarding antidepressant use during pregnancy: A discrete choice experiment. Birth Defects Research Part A: Clinical and Molecular Teratology. 2012; 94(7):532-539.

Systematic reviews

• Lamsal R, Yeh A, Pullenayegum E, Ungar W. A systematic review of methods and practice for integrating maternal, fetal and child health outcomes, and family spillover effects into cost-utility analyses. PharmacoEconomics. 2024; 42:843-863.
• Lamsal R, Yeh A, Pullenayegum E, Ungar WJ. A sytematic review of methods used by pediatric cost-utility analyses to include family health spillover effects. PharmacoEconomics. 2024; 42(2):199-217.
• Subasri M, Cressman C, Arje D, Schreyer L, Cooper E, Patel K, Ungar WJ, Barwick M, Denburg A, Hayeems RZ. Translating precision health for pediatrics: A scoping review. Children. 2023; 10(5):897.
• Lee W, Shickh S, Assamad D, Luca S, Clausen M, Somerville C, Tafler A, Shaw A, Hayeems RZ, Bombard Y, Genetics Navigator Study Team. Patient-facing digital tools for delivering genetic services: A systematic review. Journal of Medical Genetics. 2023; 60(1):1-10.
• Asare AO, Wong AMF, Maurer D, Kulandaivelu Y, Saunders N, Ungar WJ. Economic evaluations of vision screening to detect amblyopia and refractive errors in children: A systematic review. Canadian Journal of Public Health. 2022; 113(2): 297-311.
• Hayeems RZ, Luca S, Assamad DA, Bhatt A, Ungar WJ. Utility of genetic testing from the perspective of parents/caregivers: A scoping review. Children, 2021; 8(4): 259.
• Lamsal R, Ungar WJ. Impact of growing up with a sibling with a neurodevelopmental disorder on the quality of life of an unaffected sibling: A scoping review. Disability and Rehabilitation. 2021; 43(4): 586-594.
• Cernat A, Hayeems RZ, Ungar WJ. Cascade health service use in family members following genetic testing in children: A scoping literature review. European Journal of Human Genetics. 2021; 29(1): 1601-1610.
• Cernat A, Hayeems RZ, Prosser LA, Ungar WJ. Incorporating cascade effects of genetic testing in economic evaluation: A scoping review of methodological challenges. Children. 2021; 8(5): 346.
• Lin E, Durbin J, Guerriere D, Volpe T, Selick A, Kennedy J, Ungar WJ, Lero DS. Assessing care-giving demands, resources and costs of family/friend caregivers for persons with mental health disorders: A scoping review. Health & Social Care in the Community. 2018; 26: 613– 634.
• Kwon J, Kim SW, Ungar WJ, Tsiplova K, Madan J, Petrou S. A systematic review and meta-analysis of childhood health utilities. Medical Decision Making. 2018; 38(3): 277-305.
• Yuen T, Penner M, Carter MT, Szatmari P, Ungar WJ. Assessing the accuracy of the Modified Checklist for Autism in Toddlers: A systematic review and meta-analysis. Developmental Medicine and Child Neurology. 2018; 60(11): 1093-1100.
• Penner M, Anagnostou E, Ungar WJ. Systematic review of clinical guidance documents for autism spectrum disorder diagnostic assessment. Autism. 2018; 22(5): 517-527.
• Ting EEK, Sander B, Ungar WJ. Systematic review of the cost-effectiveness of influenza immunization programs. Vaccine. 2017. 35(15): 1828-1843.
• Zur RM, Roy L, Ito S, Beyene J, Carew C, Ungar WJ. Thiopurine s-methyltransferase testing for averting drug toxicity: A meta-analysis of diagnostic test accuracy. The Pharmacogenomics Journal. 2016; 16(4): 305-311.
• Zur RM, Roy LM, Ungar WJ. Thiopurine s-methyltransferase testing for averting drug toxicity in patients receiving thiopurines: A meta-analysis of diagnostic test accuracy. Toronto: The Hospital for Sick Children, October 2015. TASK Report No. 2015-03. Highlights, Executive Summary Full Report, Appendices.
• Roy L, Zur RM, Ungar WJ. Thiopurine s-methyltransferase testing for averting drug toxicity in patients receiving thiopurines: A systematic review and quality appraisal. TASK Report No. 2015-02. Highlights, Executive Summary, Full Report, Appendices.
• Ting EEK, Sander B, Ungar WJ. Systematic review of the cost-effectiveness of influenza immunization programs: A Canadian perspective. December 2015. TASK Report No. 2015-04. Highlights, Abstract, Full Report, Appendices.
• Burnett HF, Tanoshima R, Chandranipapongse W, Madadi P, Ito S, Ungar WJ. Testing for thiopurine methyltransferase status for safe and effective administration of thiopurines: A systematic review of clinical guidance documents. The Pharmacogenomics Journal. 2014; 14(6):493-502.
• Burnett H, Chandranipapongse W, Tanoshima R, Ito S, Ungar WJ.  Thiopurine dosing using thiopurine methyltransferase status: A systematic review of clinical guidance. TASK Report No. 2013-02. Highlights, Executive Summary, Full Report, Appendices.
• Donnan JR, Ungar WJ, Mathews M, Rahman P. Systematic review of thiopurine methyltransferase genotype and enzymatic testing strategies. Therapeutic Drug Monitoring. 2011; 33:192–199.
• Costa V, Ungar WJ. Systematic review on orchidopexy for the Canadian Paediatric Surgical Wait Times Project. TASK Report No. 2009-01. 2009. Full Report
• Hancock RL, Koren G, Einarson, A, Ungar WJ. The effectiveness of Teratology Information Services (TIS). Reproductive Toxicology. 2007; 23(2):125-32.
• Cooper M, Ungar W, Zlotkin S. An assessment of inter-rater agreement of the literature filtering process in the development of evidence-based dietary guidelines. Public Health Nutrition. 2006; 9(4):494-500.

Trends in evidence

• Kwon J, Kim SW, Ungar WJ, Tsiplova K, Madan J, Petrou S. Patterns, trends and methodological associations in the measurement and valuation of childhood health utilities. Quality of Life Research. 2019; 28(7): 1705-1724.
• Payakachat N, Tilford JM, Ungar WJ. National Database for Autism Research (NDAR): Big Data opportunities for health services research and health technology assessment. Pharmacoeconomics. 2016; 34(2): 127-38.
• Sullivan SM, Tsiplova K, Ungar WJ. A scoping review of pediatric economic evaluation 1980-2014: Do trends over time reflect changing priorities in evaluation methods and childhood disease? Expert Review of Pharmacoeconomics and Outcomes Research. 2016; 16(5): 599-607.
• Sullivan S, Ungar WJ. Will the growth spurt continue? Trends in child health economic evaluation: 1980 to 2013. TASK Report No. 2016-01. Highlights, Executive Summary, Full Report.
• Ungar WJ, Santos MT. Trends in paediatric health economic evaluation: 1980 to 1999. Archives of Disease in Childhood. 2004; 89:26-29.
• Lipstein EA, Brinkman WB, Fiks AG, Hendrix KS, Kryworuchko J, Miller VA, Prosser LA, Ungar WJ, Fox D. An emerging field of research:  Challenges in pediatric decision making. Medical Decision Making. 2015; 35:403-408.
• Kromm SK, Bethell J, Kraglund F, Edwards S, Laporte A, Coyte PC, Ungar WJ. Characteristics and quality of pediatric cost-utility analyses. Quality of Life Research. 2012; 21(8):1315-1325.
• Hancock RL, Ungar WJ, Einarson A, Koren G. International practices in the provision of teratology information: A survey of international teratogen information programs and comparisons with the North American model. Journal of Evaluation in Clinical Practice. 2010; 16(5):957-963.
• Hancock RL, Ungar WJ, Einarson A, Goodstadt M, Koren G. Providing information regarding exposures in pregnancy: a survey of North American Teratology Information Services. Reproductive Toxicology. 2008; 25(3): 381-387.
• Ungar WJ, Coyte PC. The Pharmacy Medication Monitoring Program Advisory Board. Health services utilization reporting in respiratory patients Health services utilization reporting in respiratory patients. Journal of Clinical Epidemiology. 1998; 51(12):1335-1342.

TASK performs systematic reviews, cost-effectiveness analyses and methods research in the area of respiratory conditions, including asthma and bronchiolitis.

• Syed N, Ryu MH, Dhillon S, Schaeffer MR, Ramsook AH, Leung JM, Ryerson CJ, Carlsten C, Guenette JA. Canadian Respiratory Research Network. Effects of traffic‐related air pollution on exercise endurance, dyspnea, and cardiorespiratory responses in health and COPD: A randomized, placebo‐controlled, crossover trial. Chest. 2022; 161(3): 662‐675.
• Najafzadeh M, Ungar WJ, Hadioonzadeh A, Tsao N, Lynd LD. Comparing different experimental designs for best-worst scaling choice experiments: The case of asthma control. International Journal of Health Preference Research. 2018: 1:3-16.
• Mendlowitz A, Widjaja E, Phan C, Sun Z, Moretti ME, Schuh S, Coyte PC, Hancock-Howard R, Freedman S, Ungar WJ. A cost analysis of pulse oximetry as a determinant in the decision to admit infants with mild to moderate bronchiolitis. Pediatric Emergency Care. 2018.
• Ungar WJ, Hadioonzadeh A, Najafzadeh M, Tsao NW, Dell S, Lynd LD. Parents and adolescents preferences for asthma control: A best-worst scaling choice experiment using an orthogonal main effects design. BMC Pulmonary Medicine. 2015; 15:146.
• Ungar WJ, Hadioonzadeh A, Najafzadeh M, Tsao NW, Dell S, Lynd LD. Quantifying preferences for asthma control in parents and adolescents using a best-worst scaling experiment. Respiratory Medicine. 2014; 108(6):842-851. 
• Schuh S, Freedman S, Coates A, Allen U, Parkin P, Stephens D, Ungar W, Da Silva Z, Willan A. Effect of oximetry on hospitalization in bronchiolitis – a randomized clinical trial. JAMA. 2014; 312(7):712-718.
•  Ungar WJ, Boydell K, Dell S, Feldman BM, Marshall D, Willan A, Wright J. A parent-child dyad approach to the assessment of health status and health-related quality of life in children with asthma. PharmacoEconomics. 2012; 30(8):697-712.
• Ungar WJ. Medication cost-sharing and health outcomes in children with asthma. JAMA. 2012; 307(12):1316-1318.
• Ungar WJ, Paterson JM, Gomes T, Bikangaga P, Gold M, To T, Kozyrskyj AL. Relationship of asthma management, socioeconomic status, and medication insurance characteristics to exacerbation frequency in children with asthma. Annals of Allergy, Asthma & Immunology. 2011; 106:17–23.
• To T, Wang C, Dell SD, Fleming-Carroll B, Parkin P, Scolnik D, Ungar WJ, the PAMG Team. Can an evidence-based guideline reminder card improve acute asthma management in emergency department? Respiratory Medicine. 2010; 104(9):1263-70.
• To T, Wang C, Dell S, Fleming-Carroll B, Parkin P, Scolnik D, Ungar W, the PAMG Team. Risk factors for repeat adverse asthma events in children after visiting an emergency department. Ambulatory Pediatrics. 2008; 8(5):281–287.
• Ungar WJ, Cope SF, Kozyrskyj A, Paterson JM. Socioeconomic factors and home allergen exposure in children with asthma. Journal of Pediatric Health Care. 2010; 24(2):108-115.
• Cope SF, Ungar WJ, Glazier RH. International differences in asthma guidelines for children. International Archives of Allergy and Immunology. 2009; 148(4):265-278.
• Cope SF, Ungar WJ, Glazier RH. Socioeconomic factors and asthma control in children. Pediatric Pulmonology. 2008; 43(8):745-752.
• Ungar WJ, Paterson M, Cope S, Kozyrskyj A. Improving drug benefits for children with asthma: Building a research agenda. Healthcare Policy. 2008; 3(4):66-76.
• Ungar WJ, Kozyrskyj A, Paterson M, Ahmad F. Effect of cost-sharing on asthma medication use in children. Archives of Pediatrics & Adolescent Medicine. 2008; 162(2):104-110.
• Yong JHE, Schuh S, Rashidi R, Vanderby S, Lau R, Laporte A, Nauenberg E, Ungar WJ. A cost-effectiveness analysis of omitting radiography in diagnosis of acute bronchiolitis. Pediatric Pulmonology. 2009; 44(2):122-127.
• Polisena J, Tam S, Lodha A, Laporte A, Coyte PC, Ungar WJ. An economic evaluation of asthma action plans for children with asthma. Journal of Asthma. 2007; 44 (7):501-508.
• Ungar WJ, Davidson-Grimwood SR, Cousins M. Parents were accurate proxy reporters of urgent pediatric asthma health services – a retrospective agreement analysis. Journal of Clinical Epidemiology. 2007; 60(11):1176-1183.
• Ungar WJ. Improving Drug Benefits for Children with Asthma: Building a Research Agenda, Post-Workshop Report. AllerGen NCE Inc. June 2007
• Ungar WJ, Mirabelli C, Cousins M, Boydell KM. A qualitative analysis of a dyad approach to quality of life measurement in children with asthma. Social Science & Medicine. 2006; 63(9):2354-66.
• Kozyrskyj AL, Dahl ME, Ungar WJ, Becker AB, Law BJ. Antibiotic treatment of wheezing in children with asthma: What is the practice? Pediatrics. 2006; 117(6):e1104-10.
• Ungar WJ, Macdonald T, Cousins M. Better breathing or better living? A qualitative analysis of the impact of asthma medication acquisition on standard living and quality of life in low income families of children with asthma. Journal of Pediatric Health Care. 2005; 19(6):354-62.
• Ungar WJ, Chapman KR, Santos MT. Assessment of a medication-based asthma index for population research. American Journal of Respiratory and Critical Care Medicine. 2002; 165:190-194.
• Ungar WJ, Coyte PC. The Pharmacy Medication Monitoring Program Advisory Board. Prospective study of the patient-level cost of asthma care in children. Pediatric Pulmonology. 2001; 32:101-108.
• Ungar WJ, Coyte PC. The Pharmacy Medication Monitoring Program Advisory Board. Measuring productivity loss days in asthma patients. Health Economics. 2000; 9(1): 37-46.
• Ungar WJ, Coyte PC. The Pharmacy Medication Monitoring Program Advisory Board. Health services utilization reporting in respiratory patients. Journal of Clinical Epidemiology. 1998; 51(12):1335-1342.
• Ungar WJ. An assessment of a prospective measure of health services utilization and the cost of asthma. Doctoral dissertation, University of Toronto, June 1997.
• Ungar W. The influence of cigarette smoke exposure on diazepam metabolism in the guinea pig maternal-fetal unit. M.Sc. dissertation, McGill University, August, 1984.

Juvenile idiopathic arthritis research conducted at TASK includes assessing patient preferences for drug treatment and cost effectiveness of biologic response modifiers.

• Luca NJ, Burnett HF, Ungar WJ, Moretti ME, Beukelman T, Feldman BM, Schwartz G, Bayoumi AM. Cost-effectiveness analysis of first line treatment with biologic agents in polyarticular juvenile idiopathic arthritis. Arthritis Care and Research. 2016; 68(12): 1803-1811.
• Burnett HF, Ungar WJ, Regier DA, Feldman BM, Miller FA. Parent willingness-to-pay for biologic treatments in juvenile idiopathic arthritis. Value in Health. 2014; 17(8):830-837.
• Regier DA, Watson V, Burnett H, Ungar WJ. Task complexity and response certainty in discrete choice experiments: An application to drug treatments for juvenile idiopathic arthritis. Journal of Behavioral and Experimental Economics. 2014; 50:40-49.
• Burnett HF, Regier DA, Feldman BM, Miller FA, Ungar WJ. Parents’ preferences for drug treatments in juvenile idiopathic arthritis: A discrete choice experiment. Arthritis Care & Research. 2012; 64(9):1382-1391.
• Ungar WJ, Costa V, Burnett H, Feldman BM, Laxer RM. The use of biologic response modifiers in polyarticular-course juvenile idiopathic arthritis: A systematic review. Seminars in Arthritis and Rheumatism. 2013; 42(6):597-618.
• Ungar WJ, Costa V, Hancock-Howard RL, Feldman B, Laxer RM. Cost-effectiveness of biologics in polyarticular-course juvenile idiopathic arthritis patients unresponsive to disease modifying anti-rheumatic drugs. Arthritis Care & Research. 2011; January; 63(1):111–119.
• Ungar WJ, Costa V, Burnett H. The use of biologic response modifiers in polyarticular course juvenile idiopathic arthritis – a systematic review update. TASK Report No. 2012-01. Report Update.
• Costa V, Ungar WJ, Hancock Howard R, Feldman B, Laxer R. The use of biologic response modifiers in polyarticular-course juvenile idiopathic arthritis, TASK Report No. 2010-01, January 2010. Highlights, Executive Summary, Full Report, Appendices.

TASK conducts cost-effectiveness analysis of paediatric health interventions and devices in the fields of infectious disease, appendicitis, fetal alcohol spectrum disorder, oncology and surgical interventions.

Table of contents

1. Infectious diseases
2. Appendicitis
3. Fetal disorders
4. Oncology
5. Gastrointestinal disease
6. Surgical interventions
7. Haemophilia
8. Ophthalmology

Infectious diseases

• Ting EEK, Sander B, Ungar WJ. Systematic review of the cost-effectiveness of influenza immunization programs. Vaccine. 2017. 35(15): 1828-1843.
• Ting EEK, Sander B, Ungar WJ. Systematic review of the cost-effectiveness of influenza immunization programs: A Canadian perspective. December 2015. TASK Report No. 2015-04. Highlights, Abstract, Full Report, Appendices.
• Gauvreau C, Ungar WJ, Köhler JC, Zlotkin S. The use of cost-effectiveness analysis for pediatric immunization in developing countries. The Milbank Quarterly. 2012. 90(4):762-790.
• Costa V, Ungar WJ. Caspofungin in the empiric treatment of febrile neutropenia in pediatric patients: A comparison with conventional and liposomal amphotericin B, TASK Report No. 2008-01, 10 September 2008. Highlights, Abstract, Executive Summary, Full Report, Appendices.
• Boutis K, Stephens D, Lam K, Ungar WJ, Schuh S. The impact of Severe Acute Respiratory Syndrome on a tertiary care pediatric emergency department. Canadian Medical Association Journal. 2004; 171(11): 1353-1358.

Appendicitis

• Hall NJ, Eaton S, Abbo O, Arnaud A, Beaudin M, Brindle M, Butter A, Davies D, Jancelewicz T, Johnson K, Keijzer R, Lapidus-Kroll E, Nasr A, Offringa M, Piche N, Rintala R, Skarsgard E, Svensson J, Ungar WJ, Wester T, Willan A, Zani A, St Peter SD, Pierro A. Appendectomy versus non-operative treatment for acute uncomplicated appendicitis in children: study protocol for a multicentre, open-label, non-inferiority, randomized controlled trial. BMJ Paediatrics Open. 2017; 1(1): e000028.
• Wan M, Doria A, Krahn M, Ungar WJ, Medina S, Sung L. Acute appendicitis in young children: cost-effectiveness of US versus CT in diagnosis–a Markov decision analytic model. Radiology. 2009; 250(2): 378-86.
• Doria AS, Amernic H, Dick P, Babyn P, Chait P, Langer J, Coyte PC, Ungar WJ. Cost-effectiveness analysis of weekday and weeknight or weekend shifts for assessment of appendicitis. Pediatric Radiology. 35(12):1186-95, 2005.

Fetal disorders

• Berrigan, P, Hicks GG, Ungar WJ, Zwicker JD. Budget impact analysis of an epigenetic test used for diagnosing fetal alcohol spectrum disorder from the perspective of a laboratory budget holder in Manitoba, Canada. Pharmacoeconomics Open, 2021.
• Xie X, Wang M, Goh ES-Y, Ungar WJ, Little J, Carroll JC, Okun N, Huang T, Rousseau F, Dougan S, Tu HA, Higgins C, Holubowich C, Sikich N, Dhalla IA, Ng V. Noninvasive prenatal testing for trisomies 21, 18, and 13, sex chromosome aneuploidies, and microdeletions in average-risk pregnancies: A cost-effectiveness analysis. Journal of Obstetrics and Gynaecology Canada. 2020; 42(6): 740-749.
• Hancock, RL. An Economic Evaluation of Teratology Information Services. Doctoral Dissertation, 2010.
• Goh YI, Rovet J, Ungar W, Koren G. Mega-dose Vitamin C and E in Preventing FASD:  The Decision to Terminate the Study Prematurely. Journal of Fetal Alcohol Syndrome International. 2007; 5:e3.
• Goh YI, Rovet J, Ungar WJ, Koren G. The antioxidant effect: Can we mitigate fetal alcohol spectrum disorder with antioxidants? Journal of Fetal Alcohol Syndrome International. 2005; 3:e10.
• Stade B, Ungar WJ, Stevens B, Beyene J, Koren G. Cost of fetal alcohol spectrum disorder in Canada. Canadian Family Physician. 2007; 53(8):1303-04.
• Stade B, Stevens B, Ungar W, Beyene J, Koren G. Health-related quality of life of Canadian children prenatally exposed to alcohol. Health & Quality of Life Outcomes. 2006; 13(4):81.
• Stade B, Ungar WJ, Stevens B, Beyene J, Koren G. The burden of prenatal exposure to alcohol: Measurement of cost. Journal of Fetal Alcohol Syndrome International. 2006; 4:e5.
• Wu R, Laporte A, Ungar WJ. Cost effectiveness of an electronic medication ordering and administration system in reducing adverse drug events. Journal of Evaluation in Clinical Practice. 2007; 13(3):440-448.

Oncology

• Tsimicalis A, Genest L, Stevens B, Ungar WJ, Barr B, Greenberg M, Guerriere D, Naqvi A, Agha M. The impact of a childhood cancer diagnosis on the children and siblings’ school attendance, performance and activities: A qualitative descriptive study. Journal of Pediatric Oncology Nursing. 2018; 35(2): 118-131.
• Tsimicalis A, Stevens B, Ungar WJ, Greenberg M, McKeever P, Agha M, Guerriere D, Barr R, Naqvi A, Moineddin R. Determining the costs of families’ support networks following a child’s cancer diagnosis. Cancer Nursing: An International Journal for Cancer Care. 2013; 36(2):E8-E19.
• Tsimicalis A, Stevens B, Ungar WJ, McKeever P, Greenberg M, Agha M, Guerriere D, Naqvi A, Barr R. A mixed method approach to describe the out-of-pocket expenses incurred by families of children with cancer. Pediatric Blood and Cancer. 2013; 60(3):438-445.
• Hancock-Howard RL, McMahon M, Menon-Nair A, Woo G, Aziza A, Laporte A, Nauenberg E, Connolly BL, Wales PW, Ungar WJ. Cost-effectiveness analysis of implantable venous access device insertion using interventional radiologic versus conventional operating room methods in pediatric patients with cancer. Journal of Vascular and Interventional Radiology. 2010; 21(5):677-84.

Gastrointestinal disease

TASK conducts research on cost-effectiveness and quality of life in pediatric inflammatory bowel disease including Crohn’s disease, and gastroenterology.

• Gattini D, Belza C, Kraus R, Avitzur Y, Ungar WJ, Wales P. Cost-utility analysis of teduglutide compared to standard care in weaning parenteral nutrition support in children with short bowel syndrome. Clinical Nutrition, 42(12):2363-2371, 2023.
• Bashir NS, Walters TD, Griffiths AM, Otley A, Critch J, Ungar WJ. A comparison of the Child Health Utility 9D and the Health Utilities Index for estimating health utilities in pediatric inflammatory bowel disease. Quality of Life Research, 32(9):2527-2539, 2023.
• Bashir NS, Walters TD, Griffiths AM, Otley A, Critch J, Ungar WJ. A comparison of preference-based, generic and disease-specific health-related quality of life in pediatric inflammatory bowel disease. Journal of the Canadian Association of Gastroenterology. 2023; 6(2):73-79.
• Bashir NS, Hughes A, Ungar WJ. Infliximab pricing in international economic evaluations in inflammatory bowel disease to inform biologic and biosimilar access policies. Medical Decision Making Policy & Practice. 2023; 8(1):1-22.
• Belza C, Avitzur Y, Ungar WJ, Stremler R, Fehlings D, Wales PW. Stress, anxiety and health-related quality of life in caregivers of children with intestinal failure on parenteral nutrition: A cross-sectional survey study. Journal of Parenteral and Enteral Nutrition. 2023; 47(3):342-353.
• Belza C, Ungar WJ, Avitzur Y, Stremler R, Fehlings D, Wales PW. Carrying the burden: Informal care requirements by caregivers of children with intestinal failure on home parenteral nutrition. Journal of Pediatrics. 2022; 250:75-82e3.
• Belza C, Patterson C, Ghent E, Avitzur Y, Ungar WJ, Fehlings D, Stremler R, Wales PW. “Line care governs our entire world”: Understanding the experience of caregivers of children with intestinal failure on long-term parenteral nutrition. Journal of Parenteral and Enteral Nutrition. 2022; 46(7):1602-1613.
• Bashir NS, Walters TD, Griffiths AM, Ungar WJ. An assessment of the validity and reliability of the pediatric Child Health Utility 9D in children with inflammatory bowel disease. Children. 2021, 208(5): 343.
• Moretti ME, Ungar WJ, Freedman SB, Schuh S. Cost-effectiveness of preferred fluids versus electrolytes in pediatric gastroenteritis. Canadian Journal of Emergency Medicine. 2021, 23(1): 646-654.
• Hughes A, Marshall J, Moretti M, Ungar WJ. A cost-utility analysis of biosimilar infliximab compared to reference infliximab in adult patients with Crohn’s Disease. Journal of the Canadian Association of Gastroenterology. 2020; 4(1): 48.
• Bashir NS, Walters TD, Griffiths AM, Ito S, Ungar WJ. Cost-effectiveness and clinical outcomes of early anti-tumor necrosis factor-α intervention in pediatric Crohn’s disease. Inflammatory Bowel Diseases. 2020; 26(8): 1239-1250.
• Hughes A, Marshall J, Moretti ME, Ungar WJ. A cost-utility analysis of biosimilar infliximab compared to reference infliximab in adult switch patients with Crohn’s disease: A Canadian analysis. TASK Technical Report No. 2019-02. 2019. Highlights, Executive Summary, Full Report.
• Bashir NS, Walters TD, Griffiths AM,  Ito S, Ungar WJ.  The economic evaluation of early intervention with Anti-Tumor Necrosis Factor-α treatments in pediatric Crohn’s disease. TASK Technical Report No. 2019-01. 2019. Highlights, Executive Summary, Full Report.
• Woo G, Tomlinson G, Yim C, Lilly L, Therapondos G, Wong DKH, Ungar WJ, Einarson TR, Sherman M, Heathcote EJ, Krahn M. Health state utilities and quality of life in patients with hepatitis B. Canadian Journal of Gastroenterology. 2012; 26(7):445-451.
• Anvari M, Allen C, Marshall J, Armstrong D, Goeree R, Ungar W, Goldsmith C. A randomized controlled trial of laparoscopic Nissen fundoplication (LNF) versus proton pump inhibitors for treatment of patients with chronic gastro-esophageal reflux disease (GERD): three year outcomes. Surgical Endoscopy. 2011; 25(8):2547-2554.
• Anvari M, Allen C, Marshall J, Armstrong D, Goeree R, Ungar W, Goldsmith C. A randomized controlled trial of Laparoscopic Nissen Fundoplication (LNF) versus proton pump inhibitors for treatment of patients with chronic Gastro-Esophageal Reflux Disease (GERD): One-year Follow-up. Surgical Innovation. 2006; 13(4):238-49.
• Urbach DR, Ungar WJ, Rabeneck L. Whither surgery in the treatment of gastroesophageal reflux disease (GERD)? Canadian Medical Association Journal. 2004; 170(2):219-221.

Surgical interventions

• Dong Z, Connolly BK, Ungar WJ, Coyte P. A cost analysis of peripherally inserted central catheter in paediatric patients. International Journal of Technology Assessment in Health Care. 2018; 34(1): 38-45.
• Burnett HF, Lambley R, West SK, Ungar WJ, Mireskandari, K. Cost-effectiveness analysis of clinic-based chloral hydrate sedation versus general anaesthesia for paediatric ophthalmological procedures. British Journal of Ophthalmology. 2015; 99:1565-1570.
• Burnett H, Lambley R, Ungar WJ, Mireskandari K. The cost-effectiveness of clinic-based chloral hydrate sedation versus general anaesthesia for paediatric ophthalmological procedures. TASK Report No. 2014-01, Highlights, Executive Summary, Full Report, Appendices
• Costa V, Ungar WJ. Systematic review on orchidopexy for the Canadian Paediatric Surgical Wait Times Project. TASK Report No. 2009-01. 2009. Full Report
• Piwko C, Lasry A, Alanezi K, Coyte PC, Ungar WJ. Economic evaluation of ondansetron versus dimenhydrinate for prevention of post-operative vomiting in children undergoing strabismus surgery. Pediatric Anesthesia. 2005; 15(9):755-761.
• Attard NJ, Wei X, Laporte A, Zarb GA, Ungar WJ. A cost minimization analysis of implant treatment in mandibular edentulous patients. International Journal of Prosthodontics. 2003; 16(3):271-276.

Haemophilia

• Khan U, Bogue C, Ungar WJ, Hilliard P, Blanchette V, Carcao, M, Doria AS. Cost-effectiveness analysis of different imaging strategies for diagnosis of hemophilic arthropathy. Haemophilia. 2010; 16(2):322-32.
• Carcao M, Ungar WJ, Feldman B. Cost-utility analysis in evaluating prophylaxis in haemophilia. Haemophilia. 2004; 10(Suppl 1):50-57.

Ophthalmology

• Asare A, Maurer D, Wong AMF, Ungar WJ, Saunders N. Cost-effectiveness of universal school and community-based vision testing strategies to detect amblyopia in children in Ontario, Canada. JAMA Network Open. 2023; 6(1): e2249384.
• Asare AO, Wong AMF, Maurer D, Kulandaivelu Y, Saunders N, Ungar WJ. Economic evaluations of vision screening to detect amblyopia and refractive errors in children: A systematic review. Canadian Journal of Public Health. 2022; 113(1): 297-311.
• Asare AO, Maurer D, Wong AMF, Ungar WJ, Saunders N. Socioeconomic status and vision care services in Ontario, Canada: A population-based cohort study. Journal of Pediatrics. 2022; 241:212-220.e2.
• Burnett HF, Lambley R, West SK, Ungar WJ, Mireskandari, K. Cost-effectiveness analysis of clinic-based chloral hydrate sedation versus general anaesthesia for paediatric ophthalmological procedures. British Journal of Ophthalmology. 2015; 99:1565-1570.
• Burnett H, Lambley R, Ungar WJ, Mireskandari K. The cost-effectiveness of clinic-based chloral hydrate sedation versus general anaesthesia for paediatric ophthalmological procedures. TASK Report No. 2014-01, Highlights, Executive Summary, Full Report, Appendices

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