Expectations and values about expanded newborn screening: a public engagement study
Robin Z. Hayeems PhD
Postdoctoral Fellow
Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada
Search for more papers by this authorCorresponding Author
Fiona A. Miller PhD
Associate Professor
Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada
Correspondence
Fiona Miller PhD
4th Floor
155 College Street
Toronto
ON
Canada M5G 3T6
E-mail: [email protected]
Search for more papers by this authorYvonne Bombard PhD
Postdoctoral Fellow, Research Fellow
Yale School of Public Health, Yale University, New Haven, CT, USA
The Center for Health Policy and Outcomes, Memorial Sloan-Kettering Cancer Center, New York, NY, USA
Search for more papers by this authorDenise Avard PhD
Associate Professor and Research Director
Department of Human Genetics, Centre of Genomics and Policy, McGill University, Montreéal, QC, Canada
Search for more papers by this authorJune Carroll MD
Associate Professor and Clinician Scientist
Department of Family and Community Medicine, Mount Sinai Hospital, University of Toronto, Toronto, ON, Canada
Search for more papers by this authorBrenda Wilson MBChB MSc MRCP (UK) FFPH
Professor
Department of Epidemiology and Community Medicine, University of Ottawa, Ottawa, ON, Canada
Search for more papers by this authorJulian Little PhD
Professor and Chair
Department of Epidemiology and Community Medicine, University of Ottawa, Ottawa, ON, Canada
Search for more papers by this authorPranesh Chakraborty MD
Director, Physician, Assistant Professor
Newborn Screening Ontario, Department of Pediatrics, Children's Hospital of Eastern Ontario, Ottawa, ON, Canada
Department of Pediatrics, University of Ottawa, Ottawa, ON, Canada
Search for more papers by this authorJessica Bytautas BA (Hons)
Research Officer
Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada
Search for more papers by this authorYves Giguere MD PhD
Associate Professor, Physician and Program Director
Department of Medical Biology, Medical Biochemistry and Pathology, Faculty of Medicine, Universiteé Laval, Québec City, QC, Canada
Quebec Newborn Blood Screening Program, CHU de Queébec, Québec City, QC, Canada
Search for more papers by this authorJudith Allanson MD
Physician, Professor
Department of Pediatrics, University of Ottawa, Ottawa, ON, Canada
Department of Genetics, Children's Hospital of Eastern Ontario, Ottawa, Canada
Search for more papers by this authorRenata Axler MBioethics
PhD Candidate
Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada
Search for more papers by this authorRobin Z. Hayeems PhD
Postdoctoral Fellow
Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada
Search for more papers by this authorCorresponding Author
Fiona A. Miller PhD
Associate Professor
Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada
Correspondence
Fiona Miller PhD
4th Floor
155 College Street
Toronto
ON
Canada M5G 3T6
E-mail: [email protected]
Search for more papers by this authorYvonne Bombard PhD
Postdoctoral Fellow, Research Fellow
Yale School of Public Health, Yale University, New Haven, CT, USA
The Center for Health Policy and Outcomes, Memorial Sloan-Kettering Cancer Center, New York, NY, USA
Search for more papers by this authorDenise Avard PhD
Associate Professor and Research Director
Department of Human Genetics, Centre of Genomics and Policy, McGill University, Montreéal, QC, Canada
Search for more papers by this authorJune Carroll MD
Associate Professor and Clinician Scientist
Department of Family and Community Medicine, Mount Sinai Hospital, University of Toronto, Toronto, ON, Canada
Search for more papers by this authorBrenda Wilson MBChB MSc MRCP (UK) FFPH
Professor
Department of Epidemiology and Community Medicine, University of Ottawa, Ottawa, ON, Canada
Search for more papers by this authorJulian Little PhD
Professor and Chair
Department of Epidemiology and Community Medicine, University of Ottawa, Ottawa, ON, Canada
Search for more papers by this authorPranesh Chakraborty MD
Director, Physician, Assistant Professor
Newborn Screening Ontario, Department of Pediatrics, Children's Hospital of Eastern Ontario, Ottawa, ON, Canada
Department of Pediatrics, University of Ottawa, Ottawa, ON, Canada
Search for more papers by this authorJessica Bytautas BA (Hons)
Research Officer
Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada
Search for more papers by this authorYves Giguere MD PhD
Associate Professor, Physician and Program Director
Department of Medical Biology, Medical Biochemistry and Pathology, Faculty of Medicine, Universiteé Laval, Québec City, QC, Canada
Quebec Newborn Blood Screening Program, CHU de Queébec, Québec City, QC, Canada
Search for more papers by this authorJudith Allanson MD
Physician, Professor
Department of Pediatrics, University of Ottawa, Ottawa, ON, Canada
Department of Genetics, Children's Hospital of Eastern Ontario, Ottawa, Canada
Search for more papers by this authorRenata Axler MBioethics
PhD Candidate
Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada
Search for more papers by this authorAbstract
Objectives
Newborn bloodspot screening (NBS) panels have expanded to include conditions for which treatment effects are less certain, creating debate about population-based screening criteria. We investigated Canadian public expectations and values regarding the types of conditions that should be included in NBS and whether parents should provide consent.
Methods
Eight focus groups (FG; n = 60) included education, deliberative discussion and pre-/post-questionnaires. Data were analysed quantitatively and qualitatively.
Results
Quantitatively, the majority supported NBS for serious disorders for which treatment is not available (95–98, 82%). A majority endorsed screening without explicit consent (77–88%) for treatable disorders, but 62% supported unpressured choice for screening for untreatable disorders. Qualitatively, participants valued treatment-related benefits for infants and informational benefits for families. Concern for anxiety, stigma and unwanted knowledge depended upon disease context and strength of countervailing benefits.
Conclusions
Anticipated benefits of expanded infant screening were prioritized over harms, with information provision perceived as a mechanism for mitigating harms and enabling choice. However, we urge caution around the potential for public enthusiasm to foster unlimited uptake of infant screening technologies.
Supporting Information
| Filename | Description |
|---|---|
| hex12047-sup-0001-AppendixS1.docWord document, 42.5 KB | Appendix S1. Discussion showcards and questions |
Please note: The publisher is not responsible for the content or functionality of any supporting information supplied by the authors. Any queries (other than missing content) should be directed to the corresponding author for the article.
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