Disclosure of research results: a randomized study on GENEPSO-PS cohort participants

Julien Mancini, Elodie Le Cozannet, Anne-Déborah Bouhnik, Noémie Resseguier, Christine Lasset, Emmanuelle Mouret-Fourme, Catherine Noguès, Claire Julian-Reynier, Julien Mancini, Elodie Le Cozannet, Anne-Déborah Bouhnik, Noémie Resseguier, Christine Lasset, Emmanuelle Mouret-Fourme, Catherine Noguès, Claire Julian-Reynier

Abstract

Background: There exist no recommendations as to how aggregate research results should best be disclosed to long-term cohort participants.

Objective: To study the impact of cohort results disclosure documents of various kinds on participants' satisfaction.

Design: Randomized study with a 2x2 factorial design.

Setting and participants: The GENEPSO-PS cohort is used to study the psychosocial characteristics and preventive behaviour of both BRCA1/2 carriers and non-carriers; 235 participants wishing to receive 'information about the survey results' answered a self-administered questionnaire.

Interventions: The impact of providing the following items in addition to a leaflet about aggregate psychosocial research results was investigated (i) an up-to-date medical information sheet about BRCA1/2 genetic topics, (ii) a photograph with the names of the researchers.

Main outcome measures: Satisfaction profiles drawn up using cluster analysis methods.

Results: Providing additional medical and/or research team information had no significant effect on satisfaction. The patients attributed to the 'poorly satisfied' group (n = 60, 25.5%) differed significantly from those in the 'highly satisfied' group (n = 51, 21.7%): they were younger [odds ratio (OR) = 0.96, 95% confidence interval (0.92-0.99), P = 0.028], less often had a daughter [OR = 4.87 (1.80-13.20), P = 0.002], had reached a higher educational level [OR = 2.94 (1.24-6.95), P = 0.014] and more frequently carried a BRCA1/2 mutation [OR = 2.73 (1.20-6.23), P = 0.017].

Conclusions: This original approach to disclosing research results to cohort participants was welcomed by most of the participants, but less by the more educated and by BRCA1/2 carriers. Although an easily understandable document is necessary, it might also be worth providing some participants with more in-depth information.

Keywords: BRCA1/2; cohort; disclosure; randomized controlled trials; research results; satisfaction.

© 2015 The Authors Health Expectations Published by John Wiley & Sons Ltd.

Figures

Figure 1
Figure 1
Participants’ flow diagram.

References

    1. Anastasova V, Mahalatchimy A, Rial‐Sebbag E et al Communication of results and disclosure of incidental findings in longitudinal paediatric research. Pediatric Allergy and Immunology: Official Publication of the European Society of Pediatric Allergy and Immunology, 2013; 24: 389–394.
    1. Christenhusz GM, Devriendt K, Dierickx K. To tell or not to tell? A systematic review of ethical reflections on incidental findings arising in genetics contexts. European Journal of Human Genetics: EJHG, 2013; 21: 248–255.
    1. Kaufman D, Murphy J, Scott J, Hudson K. Subjects matter: a survey of public opinions about a large genetic cohort study. Genetics in Medicine: Official Journal of the American College of Medical Genetics, 2008; 10: 831–839.
    1. Shalowitz DI, Miller FG. Communicating the results of clinical research to participants: attitudes, practices, and future directions. PLoS Medicine, 2008; 5: e91.
    1. Schulte PA. Ethical issues in the communication of results. Journal of Clinical Epidemiology, 1991; 44(Suppl 1): 57S–61S.
    1. Schulte PA, Singal M. Ethical issues in the interaction with subjects and disclosure of results In: Coughlin SS, Beauchamp TL. (eds) Ethics and Epidemiology. New York: Oxford University Press, 1996: 178–198.
    1. Calderwood L. Improving Between‐Wave Mailings on Longitudinal Surveys: A Randomised Experiment on the UK Millennium Cohort Study. Centre for Longitudinal Studies, Institute of Education, University of London, 2012: 37 Available at: , [Access checked on 15 July 2015].
    1. Julian‐Reynier C, Mancini J, Mouret‐Fourme E et al Cancer risk management strategies and perceptions of unaffected women 5 years after predictive genetic testing for BRCA1/2 mutations. European Journal of Human Genetics: EJHG, 2011; 19: 500–506.
    1. Bouhnik A‐D, Fabre R, Dorval M et al Development of a scale for assessing respondents’ perceptions of health research questionnaires (the REP‐HQ Scale). Journal of Clinical Epidemiology, 2012; 65: 1098–1106.
    1. Julian‐Reynier C, Fabre R, Coupier I et al BRCA1/2 carriers: their childbearing plans and theoretical intentions about having preimplantation genetic diagnosis and prenatal diagnosis. Genetics in Medicine: Official Journal of the American College of Medical Genetics, 2012; 14: 527–534.
    1. Eisinger F, Fabre R, Lasset C, Stoppa‐Lyonnet D, Julian‐Reynier C, Nogues C. Spontaneous disclosure of BRCA1/2 genetic test results to employers: a French prospective study. European Journal of Human Genetics: EJHG, 2012; 20: 981–983.
    1. Lapointe J, Dorval M, Noguès C, Fabre R, GENEPSO Cohort , Julian‐Reynier C. Is the psychological impact of genetic testing moderated by support and sharing of test results to family and friends? Familial Cancer, 2013; 12: 601–610.
    1. Julian‐Reynier C, Bouhnik A‐D, Mouret‐Fourme E et al Time to prophylactic surgery in BRCA1/2 carriers depends on psychological and other characteristics. Genetics in Medicine: Official Journal of the American College of Medical Genetics, 2010; 12: 801–807.
    1. Bureau E, Pellegrini I, Noguès C, Lasset C, Julian‐Reynier C. “Maybe they have found something new” participants’ views on returning cohort psychosocial survey results. Health Expectations: An International Journal of Public Participation in Health Care and Health Policy, In press.
    1. Machin D, Campbell MJ, Tan S‐B, Tan S‐H. Sample Size Tables for Clinical Studies. Hoboken, NJ, USA: John Wiley & Sons, 2011.
    1. Hall MA, Camacho F, Lawlor JS, Depuy V, Sugarman J, Weinfurt K. Measuring trust in medical researchers. Medical Care, 2006; 44: 1048–1053.
    1. Ware J Jr, Kosinski M, Keller SD. A 12‐Item Short‐Form Health Survey: construction of scales and preliminary tests of reliability and validity. Medical Care, 1996; 34: 220–233.
    1. Radloff LS. The CES‐D scale a self‐report depression scale for research in the general population. Applied Psychological Measurement, 1977; 1: 385–401.
    1. Fuhrer R, Antonucci TC, Gagnon M, Dartigues JF, Barberger‐Gateau P, Alperovitch A. Depressive symptomatology and cognitive functioning: an epidemiological survey in an elderly community sample in France. Psychological Medicine, 1992; 22: 159–172.
    1. Zwaenepoel L, Hoorens V, Peuskens J, Laekeman G, VZA‐Psychiatry Research Group . The “extent of information desired”‐scale in psychiatric in‐patients: a behavioural approach. Patient Education and Counseling, 2006; 62: 72–78.
    1. Williams RJ, Tse T, Harlan WR, Zarin DA. Registration of observational studies: is it time? CMAJ: Canadian Medical Association Journal = journal de l'Association medicale canadienne, 2010; 182: 1638–1642.
    1. Meulenkamp TM, Gevers SK, Bovenberg JA, Koppelman GH, van Hylckama Vlieg A, Smets EMA. Communication of biobanks’ research results: what do (potential) participants want? American Journal of Medical Genetics Part A, 2010; 152A: 2482–2492.
    1. Bunin GR, Kazak AE, Mitelman O. Informing subjects of epidemiologic study results. Children's Cancer Group. Pediatrics, 1996; 97: 486–491.
    1. Mancini J, Genre D, Dalenc F et al Participants’ uptake of clinical trial results: a randomised experiment. British Journal of Cancer, 2010; 102: 1081–1084.
    1. Sarradon‐Eck A, Sakoyan J, Desclaux A, Mancini J, Genre D, Julian‐Reynier C. “They should take time”: disclosure of clinical trial results as part of a social relationship. Social Science & Medicine (1982), 2012; 75: 873–882.
    1. Dudas RA, Lemerman H, Barone M, Serwint JR. PHACES (Photographs of Academic Clinicians and Their Educational Status): a tool to improve delivery of family‐centered care. Academic Pediatrics, 2010; 10: 138–145.
    1. Wilson SE, Baker ER, Leonard AC, Eckman MH, Lanphear BP. Understanding preferences for disclosure of individual biomarker results among participants in a longitudinal birth cohort. Journal of Medical Ethics, 2010; 36: 736–740.
    1. Paasche‐Orlow MK, Taylor HA, Brancati FL. Readability standards for informed‐consent forms as compared with actual readability. The New England Journal of Medicine, 2003; 348: 721–726.
    1. Beskow LM, Burke W, Fullerton SM, Sharp RR. Offering aggregate results to participants in genomic research: opportunities and challenges. Genetics in Medicine: Official Journal of the American College of Medical Genetics, 2012; 14: 490–496.
    1. Jaipaul CK, Rosenthal GE. Are older patients more satisfied with hospital care than younger patients? Journal of General Internal Medicine, 2003; 18: 23–30.
    1. Ratnayake P, Wakefield CE, Meiser B et al An exploration of the communication preferences regarding genetic testing in individuals from families with identified breast/ovarian cancer mutations. Familial Cancer, 2011; 10: 97–105.

Source: PubMed

Спонсоры и соавторы

Заболевания

Медикаментозные вмешательства

Подписаться