Literature DB >> 21407262

Not so simple: a quasi-experimental study of how researchers adjudicate genetic research results.

Robin Zoe Hayeems1, Fiona Alice Miller, Li Li, Jessica Peace Bytautas.   

Abstract

Ethicists contend that researchers are obliged to report genetic research findings to individual study participants when they are clinically significant, that is, when they are clinically useful or personally meaningful to participants. Yet whether such standards are well understood and can be consistently applied remains unknown. We conducted an international, cross-sectional survey of cystic fibrosis (CF) and autism genetics researchers using a quasi-experimental design to explore factors influencing researchers' judgments. Eighty percent of researchers agreed, in principle, that clinically significant findings should be reported to individual participants. Yet judgments about when a specific finding was considered clinically significant or warranted reporting varied by scientific factors (replication, robustness, intentionality, and disease context), capacity of the research team to explain the results, and type of research ethics guidance. Further, judgments were influenced by the researchers' disease community (autism or CF), their primary role (clinical, molecular, statistical) and their beliefs regarding a general reporting obligation. In sum, judgments about the clinical significance of genetic research results, and about whether they should be reported, are influenced by scientific parameters as well as contextual factors related to the specific research project and the individual researcher. These findings call into question the assumption that the conditions under which an obligation to disclose arises are uniformly understood and actionable. Adjudicating the clinical readiness of provisional data may be a responsibility better suited to evaluative experts at arms' length of the provisional data in question, rather than a responsibility imposed upon researchers themselves.

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Year:  2011        PMID: 21407262      PMCID: PMC3137502          DOI: 10.1038/ejhg.2011.34

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  43 in total

1.  Community engagement in genetic research: results of the first public consultation for the Quebec CARTaGENE project.

Authors:  Béatrice Godard; Jennifer Marshall; Claude Laberge
Journal:  Community Genet       Date:  2007

2.  Health technology assessment in Canada: diversity and evolution.

Authors:  David M Hailey
Journal:  Med J Aust       Date:  2007-09-03       Impact factor: 7.738

3.  The search for clarity in communicating research results to study participants.

Authors:  D I Shalowitz; F G Miller
Journal:  J Med Ethics       Date:  2008-09       Impact factor: 2.903

4.  Informed consent for population-based research involving genetics.

Authors:  L M Beskow; W Burke; J F Merz; P A Barr; S Terry; V B Penchaszadeh; L O Gostin; M Gwinn; M J Khoury
Journal:  JAMA       Date:  2001-11-14       Impact factor: 56.272

5.  What is a meaningful result? Disclosing the results of genomic research in autism to research participants.

Authors:  Fiona Alice Miller; Robin Zoe Hayeems; Jessica Peace Bytautas
Journal:  Eur J Hum Genet       Date:  2010-03-17       Impact factor: 4.246

6.  Duty to disclose what? Querying the putative obligation to return research results to participants.

Authors:  F A Miller; R Christensen; M Giacomini; J S Robert
Journal:  J Med Ethics       Date:  2008-03       Impact factor: 2.903

7.  Public expectations for return of results from large-cohort genetic research.

Authors:  Juli Murphy; Joan Scott; David Kaufman; Gail Geller; Lisa LeRoy; Kathy Hudson
Journal:  Am J Bioeth       Date:  2008-11       Impact factor: 11.229

8.  Association between microdeletion and microduplication at 16p11.2 and autism.

Authors:  Lauren A Weiss; Yiping Shen; Joshua M Korn; Dan E Arking; David T Miller; Ragnheidur Fossdal; Evald Saemundsen; Hreinn Stefansson; Manuel A R Ferreira; Todd Green; Orah S Platt; Douglas M Ruderfer; Christopher A Walsh; David Altshuler; Aravinda Chakravarti; Rudolph E Tanzi; Kari Stefansson; Susan L Santangelo; James F Gusella; Pamela Sklar; Bai-Lin Wu; Mark J Daly
Journal:  N Engl J Med       Date:  2008-01-09       Impact factor: 91.245

9.  Mapping autism risk loci using genetic linkage and chromosomal rearrangements.

Authors:  Peter Szatmari; Andrew D Paterson; Lonnie Zwaigenbaum; Wendy Roberts; Jessica Brian; Xiao-Qing Liu; John B Vincent; Jennifer L Skaug; Ann P Thompson; Lili Senman; Lars Feuk; Cheng Qian; Susan E Bryson; Marshall B Jones; Christian R Marshall; Stephen W Scherer; Veronica J Vieland; Christopher Bartlett; La Vonne Mangin; Rhinda Goedken; Alberto Segre; Margaret A Pericak-Vance; Michael L Cuccaro; John R Gilbert; Harry H Wright; Ruth K Abramson; Catalina Betancur; Thomas Bourgeron; Christopher Gillberg; Marion Leboyer; Joseph D Buxbaum; Kenneth L Davis; Eric Hollander; Jeremy M Silverman; Joachim Hallmayer; Linda Lotspeich; James S Sutcliffe; Jonathan L Haines; Susan E Folstein; Joseph Piven; Thomas H Wassink; Val Sheffield; Daniel H Geschwind; Maja Bucan; W Ted Brown; Rita M Cantor; John N Constantino; T Conrad Gilliam; Martha Herbert; Clara Lajonchere; David H Ledbetter; Christa Lese-Martin; Janet Miller; Stan Nelson; Carol A Samango-Sprouse; Sarah Spence; Matthew State; Rudolph E Tanzi; Hilary Coon; Geraldine Dawson; Bernie Devlin; Annette Estes; Pamela Flodman; Lambertus Klei; William M McMahon; Nancy Minshew; Jeff Munson; Elena Korvatska; Patricia M Rodier; Gerard D Schellenberg; Moyra Smith; M Anne Spence; Chris Stodgell; Ping Guo Tepper; Ellen M Wijsman; Chang-En Yu; Bernadette Rogé; Carine Mantoulan; Kerstin Wittemeyer; Annemarie Poustka; Bärbel Felder; Sabine M Klauck; Claudia Schuster; Fritz Poustka; Sven Bölte; Sabine Feineis-Matthews; Evelyn Herbrecht; Gabi Schmötzer; John Tsiantis; Katerina Papanikolaou; Elena Maestrini; Elena Bacchelli; Francesca Blasi; Simona Carone; Claudio Toma; Herman Van Engeland; Maretha de Jonge; Chantal Kemner; Frederieke Koop; Frederike Koop; Marjolein Langemeijer; Marjolijn Langemeijer; Channa Hijmans; Channa Hijimans; Wouter G Staal; Gillian Baird; Patrick F Bolton; Michael L Rutter; Emma Weisblatt; Jonathan Green; Catherine Aldred; Julie-Anne Wilkinson; Andrew Pickles; Ann Le Couteur; Tom Berney; Helen McConachie; Anthony J Bailey; Kostas Francis; Gemma Honeyman; Aislinn Hutchinson; Jeremy R Parr; Simon Wallace; Anthony P Monaco; Gabrielle Barnby; Kazuhiro Kobayashi; Janine A Lamb; Ines Sousa; Nuala Sykes; Edwin H Cook; Stephen J Guter; Bennett L Leventhal; Jeff Salt; Catherine Lord; Christina Corsello; Vanessa Hus; Daniel E Weeks; Fred Volkmar; Maïté Tauber; Eric Fombonne; Andy Shih; Kacie J Meyer
Journal:  Nat Genet       Date:  2007-02-18       Impact factor: 38.330

10.  Communicating the results of clinical research to participants: attitudes, practices, and future directions.

Authors:  David I Shalowitz; Franklin G Miller
Journal:  PLoS Med       Date:  2008-05-13       Impact factor: 11.069

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  10 in total

1.  Parents' perspectives on participating in genetic research in autism.

Authors:  Magan Trottier; Wendy Roberts; Irene Drmic; Stephen W Scherer; Rosanna Weksberg; Cheryl Cytrynbaum; David Chitayat; Cheryl Shuman; Fiona A Miller
Journal:  J Autism Dev Disord       Date:  2013-03

2.  The art and science of biobanking.

Authors:  Bartha Maria Knoppers; Thomas J Hudson
Journal:  Hum Genet       Date:  2011-07-21       Impact factor: 4.132

3.  Paediatric research and the communication of not-so incidental findings.

Authors:  Bartha Maria Knoppers
Journal:  Paediatr Child Health       Date:  2012-04       Impact factor: 2.253

4.  Does a duty of disclosure foster special treatment of genetic research participants?

Authors:  Robin Z Hayeems; Fiona A Miller; Jessica P Bytautas; Li Li
Journal:  J Genet Couns       Date:  2013-05-17       Impact factor: 2.537

5.  One thing leads to another: the cascade of obligations when researchers report genetic research results to study participants.

Authors:  Fiona Alice Miller; Robin Zoe Hayeems; Li Li; Jessica Peace Bytautas
Journal:  Eur J Hum Genet       Date:  2012-02-15       Impact factor: 4.246

6.  Opinions and intentions of parents of an autistic child toward genetic research results: two typical profiles.

Authors:  Laurence Baret; Beatrice Godard
Journal:  Eur J Hum Genet       Date:  2011-06-15       Impact factor: 4.246

Review 7.  Risk factors for autism: translating genomic discoveries into diagnostics.

Authors:  Stephen W Scherer; Geraldine Dawson
Journal:  Hum Genet       Date:  2011-06-24       Impact factor: 4.132

8.  Potential research participants support the return of raw sequence data.

Authors:  Anna Middleton; Caroline F Wright; Katherine I Morley; Eugene Bragin; Helen V Firth; Matthew E Hurles; Michael Parker
Journal:  J Med Genet       Date:  2015-05-20       Impact factor: 6.318

9.  Health data research on sudden cardiac arrest: perspectives of survivors and their next-of-kin.

Authors:  Marieke A R Bak; Rens Veeken; Marieke T Blom; Hanno L Tan; Dick L Willems
Journal:  BMC Med Ethics       Date:  2021-01-28       Impact factor: 2.652

10.  Clinical genome sequencing and population preferences for information about 'incidental' findings-From medically actionable genes (MAGs) to patient actionable genes (PAGs).

Authors:  Thomas Ploug; Søren Holm
Journal:  PLoS One       Date:  2017-07-03       Impact factor: 3.240

  10 in total

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