Supporting informed clinical trial decisions: Results from a randomized controlled trial evaluating a digital decision support tool for those with intellectual disability
Autoři:
Lauren A. McCormack aff001; Amanda Wylie aff002; Rebecca Moultrie aff001; Robert D. Furberg aff003; Anne C. Wheeler aff002; Katherine Treiman aff001; Donald B. Bailey, Jr. aff002; Melissa Raspa aff002
Působiště autorů:
Public Health Research Division, RTI International, Research Triangle Park, North Carolina, United States of America
aff001; Center for Newborn Screening, RTI International, Research Triangle Park, North Carolina, United States of America
aff002; Health Quality & Analytics, RTI International, Research Triangle Park, North Carolina, United States of America
aff003
Vyšlo v časopise:
PLoS ONE 14(10)
Kategorie:
Research Article
doi:
https://doi.org/10.1371/journal.pone.0223801
Souhrn
Background
Informed consent requires that individuals understand the nature of the study, risks and benefits of participation. Individuals with intellectual disabilities (ID) have cognitive and adaptive impairments that may affect their ability to provide informed consent. New treatments and clinical trials for fragile X syndrome, the most commonly known inherited cause of ID, necessitate the development of methods to improve the informed consent process. The goal of this study was to compare the efficacy of a digital decision support tool with that of standard practice for informed consent and to examine whether the tool can improve decisional capacity for higher functioning individuals.
Methods
Participants (N = 89; mean age = 21.2 years) were allocated to the experimental group (consenting information provided via the digital decision support tool), or the comparison group (information provided via standard practice). Participants were assessed on four aspects of decisional capacity (Understanding, Appreciating, Reasoning, and Expressing a choice). We used regression analyses to test the impact of the tool on each outcome, repeating the analyses on the higher functioning subsample.
Results
No differences existed in any domain of decisional capacity for the sample in full. However, participants in the higher IQ subsample who used the tool scored better on Understanding after adjustment (β = 0.25, p = 0.04), but not on Appreciating or Reasoning. No differences by experimental group existed in the decision to join the hypothetical trial for the full sample or higher functioning subsample.
Conclusions
A decision support tool shows promise for individuals with fragile X syndrome with higher cognitive abilities. Future studies should examine the level of cognitive ability needed for sufficient understanding, whether these findings can be translated to other clinical populations, and the impact of the tool in larger trials and on trial retention.
Klíčová slova:
Autism – Clinical trials – Cognition – Cognitive impairment – Decision making – Medical doctors – Reasoning – Working memory
Zdroje
1. Faden RR, Beauchamp TL, King NMP. A history and theory of informed consent (Online ed.). New York: Oxford University Press; 1986.
2. Institute of Medicine. Informed consent and health literacy: Workshop summary. Washington, DC: The National Academies Press; 2015.
3. National Science Foundation. Federal policy for the protection of human subjects. Fed Regist. 2017;93(23): 7149–7274.
4. Appelbaum PS. Understanding “understanding”: An important step toward improving informed consent to research. AJOB Primary Research. 2010;1(2): 1–3. doi: 10.1080/21507716.2010.499322
5. Gillies K, Duthie A, Cotton S, Campbell MK. Patient reported measures of informed consent for clinical trials: A systematic review. PLOS ONE. 2018;13(6): e0199775. Epub 2018/06/28. doi: 10.1371/journal.pone.0199775 29949627
6. Beskow LM, Dombeck CB, Thompson CP, Watson-Ormond JK, Weinfurt KP. Informed consent for biobanking: Consensus-based guidelines for adequate comprehension. Genet Med. 2015;17(3): 226–233. Epub 2014/08/22. doi: 10.1038/gim.2014.102 25144889
7. Lentz J, Kennett M, Perlmutter J, Forrest A. Paving the way to a more effective informed consent process: Recommendations from the Clinical Trials Transformation Initiative. Contemp Clin Trials. 2016;49: 65–69. Epub 2016/06/22. doi: 10.1016/j.cct.2016.06.005 27327780
8. Anderson EE, Newman SB, Matthews AK. Improving informed consent: Stakeholder views. AJOB empirical bioethics. 2017;8(3): 178–188. Epub 2017/09/28. doi: 10.1080/23294515.2017.1362488 28949896
9. Joffe S, Cook EF, Cleary PD, Clark JW, Weeks JC. Quality of informed consent in cancer clinical trials: A cross-sectional survey. Lancet. 2001;358(9295): 1772–1777. Epub 2001/12/06. doi: 10.1016/S0140-6736(01)06805-2 11734235
10. Wood AM, White IR, Thompson SG. Are missing outcome data adequately handled? A review of published randomized controlled trials in major medical journals. Clin Trials. 2004;1(4): 368–376. Epub 2005/11/11. doi: 10.1191/1740774504cn032oa 16279275
11. Appelbaum PS. Assessment of patients’ competence to consent to treatment. N Engl J Med. 2007;357: 1834–1840.
12. Flory J, Emanuel E. Interventions to improve research participants’ understanding in informed consent for research: A systematic review. JAMA. 2004;292(13): 1593–1601. Epub 2004/10/07. doi: 10.1001/jama.292.13.1593 15467062
13. Beskow LM, Lin L, Dombeck CB, Gao E, Weinfurt KP. Improving biobank consent comprehension: a national randomized survey to assess the effect of a simplified form and review/retest intervention. Genet Med. 2017;19(5): 505–512. Epub 2016/10/14. doi: 10.1038/gim.2016.157 27735922
14. Grady C, Touloumi G, Walker AS, Smolskis M, Sharma S, Babiker AG, et al. A randomized trial comparing concise and standard consent forms in the START trial. PLOS ONE. 2017;12(4): e0172607. Epub 2017/04/27. doi: 10.1371/journal.pone.0172607 28445471
15. Kim EJ, Kim SH. Simplification improves understanding of informed consent information in clinical trials regardless of health literacy level. Clin Trials. 2015;12(3): 232–236. Epub 2015/02/24. doi: 10.1177/1740774515571139 25701156
16. Rowbotham MC, Astin J, Greene K, Cummings SR. Interactive informed consent: randomized comparison with paper consents. PLOS ONE. 2013;8(3): e58603. Epub 2013/03/14. doi: 10.1371/journal.pone.0058603 23484041
17. Kraft SA, Constantine M, Magnus D, Porter KM, Lee SS, Green M, et al. A randomized study of multimedia informational aids for research on medical practices: Implications for informed consent. Clin Trials. 2017;14(1): 94–102. Epub 2016/09/15. doi: 10.1177/1740774516669352 27625314
18. Stacey D, Légaré F, Lewis K, Barry MJ, Bennett CL, Eden KB, et al. Decision aids to help people who are facing health treatment or screening decisions. Cochrane Database Syst Rev. 2017.
19. U.S. Food and Drug Administration. Use of electronic informed consent, questions and answers: Guidance for institutional review boards, investigators, and sponsors. Silver Spring, MD: U.S. Food and Drug Administration; 2016.
20. Synnot A, Ryan R, Prictor M, Fetherstonhaugh D, Parker B. Audio-visual presentation of information for informed consent for participation in clinical trials. Cochrane Database Syst Rev. 2014;(5): CD003717. doi: 10.1002/14651858.CD003717.pub3 24809816
21. Noll S, Trent MW, editors. Mental retardation in America: A historical reader (the history of disability). New York: New York University Press; 2004.
22. Powers L, Dinerstein R, Holmes S. Self-advocacy, self-determination, and social freedom and opportunity. In: Lakin KC, Turnbull A, editors. National goals and research for people with intellectual and developmental disabilities. Washington, DC: American Association on Mental Retardation; 2005.
23. Freedman RI. Ethical challenges in the conduct of research involving persons with mental retardation. Ment Retard. 2001;39(2): 130–141. Epub 2001/05/09. doi: 10.1352/0047-6765(2001)039<0130:ECITCO>2.0.CO;2 11340962
24. Horner-Johnson W, Bailey D. Assessing understanding and obtaining consent from adults with intellectual disabilities for a health promotion study. J Policy Pract Intellect Disabil. 2013;10(3). Epub 2013/11/14. doi: 10.1111/jppi.12048 24223054
25. Fisher CB, Cea CD, Davidson PW, Fried AL. Capacity of persons with mental retardation to consent to participate in randomized clinical trials. Am J Psychiatry. 2006;163(10): 1813–1820. Epub 2006/10/03. doi: 10.1176/ajp.2006.163.10.1813 17012694
26. Dye L, Hare DJ, Hendy S. Capacity of people with intellectual disabilities to consent to take part in a research study. Journal of Applied Research in Intellectual Disabilities. 2007;20(2): 168–174. doi: 10.1111/j.1468-3148.2006.00310.x
27. Raspa M, Wheeler AC, Riley C. Public health literature review of Fragile X Syndrome. Pediatrics. 2017;139(3): S153–171. Epub 2017/08/18. doi: 10.1542/peds.2016-1159C 28814537
28. Hall SS, Burns DD, Lightbody AA, Reiss AL. Longitudinal changes in intellectual development in children with Fragile X syndrome. J Abnorm Child Psychol. 2008;36(6): 927–939. Epub 2008/03/19. doi: 10.1007/s10802-008-9223-y 18347972
29. Loesch DZ, Huggins RM, Hagerman RJ. Phenotypic variation and FMRP levels in fragile X. Mental Retardation and Developmental Disabilities Research Reviews. 2004;10: 31–41. doi: 10.1002/mrdd.20006 14994286
30. Baker DW, DeWalt DA, Schillinger D, Hawk V, Ruo B, Bibbins-Domingo K, et al. “Teach to goal”: Theory and design principles of an intervention to improve heart failure self-management skills of patients with low health literacy. J Health Commun. 2011;16(sup3): 73–88.
31. Sullivan G, Craske MG, Sherbourne C, Edlund MJ, Rose RD, Golinelli D, et al. Design of the Coordinated Anxiety Learning and Management (CALM) study: Innovations in collaborative care for anxiety disorders. Gen Hosp Psychiatry. 2007;29(5): 379–387. doi: 10.1016/j.genhosppsych.2007.04.005 17888803
32. Hooper S, Hatton D, Sideris J, Sullivan K, Hammer J, Schaaf J, et al. Executive functions in young males with fragile X syndrome in comparison to mental age-matched controls: Baseline findings from a longitudinal study. Neuropsychology. 2008;22(1): 36–47. Epub 2008/01/24. doi: 10.1037/0894-4105.22.1.36 18211154
33. Ornstein PA, Schaaf JM, Hooper SR, Hatton DD, Mirrett P, Bailey DB Jr. Memory skills of boys with Fragile X Syndrome. Am J Ment Retard. 2008;113(6): 453–465. Epub 2009/01/09. doi: 10.1352/2008.113:453-465 19127656
34. Bailey DB Jr., Raspa M, Wheeler A, Edwards A, Bishop E, Bann C, et al. Parent ratings of ability to consent for clinical trials in Fragile X Syndrome. Journal of Empirical Research on Human Research Ethics. 2014;9(3): 18–28. Epub 2014/11/26. doi: 10.1177/1556264614540591 25422596
35. Schmitt LM, Shaffer RC, Hessl D, Erickson C. Executive Function in Fragile X Syndrome: A Systematic Review. Brain Sci. 2019;9(1). Epub 2019/01/19. doi: 10.3390/brainsci9010015 30654486
36. Bailey DB Jr., Raspa M, Olmsted M, Holiday DB. Co-occurring conditions associated with FMR1 gene variations: findings from a national parent survey. Am J Med Genet A. 2008;146A(16): 2060–2069. Epub 2008/06/24. doi: 10.1002/ajmg.a.32439 18570292
37. Ligsay A, Hagerman R, Berry-Kravis E. Overview of targeted double-blind, placebo-controlled clinical trials in Fragile X Syndrome. Fragile X Syndrome. 2017: 401–418.
38. Wheeler AC, Wylie A, Raspa M, Villagomez A, Miller K, Edwards A, et al. Decisional capacity for informed consent in males and females with Fragile X Syndrome. J Autism Dev Disord. 2019. Epub 2019/03/03. doi: 10.1007/s10803-019-03930-4 30825084
39. Ryan RM, Deci EL. Self-determination theory and the facilitation of intrinsic motivation, social development, and well-being. Am Psychol. 2000;55(1): 68–78. Epub 2001/06/08. doi: 10.1037//0003-066x.55.1.68 11392867
40. Furberg RD, Ortiz AM, Moultrie RR, Raspa M, Wheeler AC, McCormack LA, et al. A digital decision support tool to enhance decisional capacity for clinical trial consent: Design and development. JMIR Res Protoc. 2018;7(6): e10525. Epub 2018/06/08. doi: 10.2196/10525 29875084
41. Sansone SM, Schneider A, Bickel E, Berry-Kravis E, Prescott C, Hessl D. Improving IQ measurement in intellectual disabilities using true deviation from population norms. J Neurodev Disord. 2014;6(1): 16. doi: 10.1186/1866-1955-6-16 26491488
42. Appelbaum PS, Grisso T. MacArthur Competence Assessment Tool for Clinical Research (MacCAT-CR). 1st ed. Sarasota, FL: Professional Research Press; 2001.
43. Roid GH. Stanford Binet Intelligence Scales (SB5). Fifth ed. Itasca, IL: Riverside Publishing; 2003.
44. Woodcock RW, McGrew KS, Mather N. Woodcock Johnson III tests of achievement. Rolling Meadows, IL: Riverside Publishing; 2001; 2007.
45. Sheslow D, Adams W. Wide range assessment of memory and learning. 2nd ed. Lutz, FL: Psychological Assessment Resources, Inc; 2003.
46. Rutter M, Bailey A, Lord C. Social communication questionnaire. Los Angeles, CA: Western Psychological Services; 2003.
47. Lord C, Rutter M, Dilavore PC, Risi S, Gotham K, Bishop SL. Autism diagnostic observation schedule. Second ed. Los Angeles, CA: Western Psychological Services; 2012.
48. Esbensen AJ, Rojahn J, Aman MG, Ruedrich S. Reliability and validity of an assessment instrument for anxiety, depression, and mood among individuals with mental retardation. J Autism Dev Disord. 2003;33(6): 617–629. doi: 10.1023/b:jadd.0000005999.27178.55 14714931
49. Yan EG, Munir KM. Regulatory and ethical principles in research involving children and individuals with developmental disabilities. Ethics Behav. 2004;14(1): 31–49. Epub 2004/12/08. doi: 10.1207/s15327019eb1401_3 15580725
50. Lipkus IM. Numeric, verbal, and visual formats of conveying health risks: Suggested best practices and future recommendations. Med Decis Making. 2007;27(5): 696–713. Epub 2007/09/18. doi: 10.1177/0272989X07307271 17873259
51. Fischoff B. Communicating risks and benefits: an evidence-based user’s guide. Brewer NT, Downs JS, editors. Washington. DC: Government Printing Office; 2012.
52. Fagerlin A, Zikmund-Fisher BJ, Ubel PA. Helping patients decide: Ten steps to better risk communication. J Natl Cancer Inst. 2011;103(19): 1436–1443. Epub 2011/09/21. doi: 10.1093/jnci/djr318 21931068
Článek vyšel v časopise
PLOS One
2019 Číslo 10
- S diagnostikou Parkinsonovy nemoci může nově pomoci AI nástroj pro hodnocení mrkacího reflexu
- Je libo čepici místo mozkového implantátu?
- Pomůže v budoucnu s triáží na pohotovostech umělá inteligence?
- AI může chirurgům poskytnout cenná data i zpětnou vazbu v reálném čase
- Nová metoda odlišení nádorové tkáně může zpřesnit resekci glioblastomů
Nejčtenější v tomto čísle
- Correction: Low dose naltrexone: Effects on medication in rheumatoid and seropositive arthritis. A nationwide register-based controlled quasi-experimental before-after study
- Combining CDK4/6 inhibitors ribociclib and palbociclib with cytotoxic agents does not enhance cytotoxicity
- Experimentally validated simulation of coronary stents considering different dogboning ratios and asymmetric stent positioning
- Risk factors associated with IgA vasculitis with nephritis (Henoch–Schönlein purpura nephritis) progressing to unfavorable outcomes: A meta-analysis
Zvyšte si kvalifikaci online z pohodlí domova
Všechny kurzy