Decisional Capacity for Informed Consent in Males and Females with Fragile X Syndrome

Wheeler, Anne; Wylie, Amanda; Raspa, Melissa; Villagomez, Adrienne; Miller, Kelsey A.; Edwards, Anne; DeRamus, Margaret; Appelbaum, Paul S.; Bailey, Donald B.

doi:10.1007/s10803-019-03930-4

Cited by 7 publications

(19 citation statements)

References 40 publications

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“…Among the full sample, and among the higher IQ subsample specifically, understanding (i.e., perceiving and retaining information related to the decision to join the clinical trial) was a strong predictor of appreciation (i.e., ones’ ability to link the decision to one’s own situation). This is consistent with our previous research [38] using an expanded sample of participants from this study, and with the results of a study by Fisher et al [25], which found that capacity to consent depends on the ability of the information to be understood by participants. These findings support the need for consent materials to be delivered in a way that augments participant understanding, thus maximizing participants’ ability to make a personal, informed decision based on their own circumstances, beliefs, and wishes.…”

Section: Resultssupporting

confidence: 94%

“…how many times per day they will have to take medicine) and what they would need to do in the doctor’s office (i.e., have their blood drawn). This is consistent with findings from our earlier research using paper-based materials with this study population [38]. While high quality evidence is lacking about the impact of audio-visual interventions on the consent process, some hypothesize that it may help individuals reduce anxiety associated with the experience [20]; this may be an indirect benefit associated with increased knowledge, greater appreciation and satisfaction with the information provided.…”

Section: Resultssupporting

confidence: 89%

“…Our earlier work also demonstrated the importance of repeating information and using alternative ways to convey knowledge, suggesting that scaffolding can improve retention and ultimately understanding [38]. Although the quality of the existing evidence is low, according to a systematic review of audio-visuals that informed consent interventions, the overall body of evidence suggests that an audio-visual strategy may slightly improve knowledge levels [20], which is the case with our findings.…”

Section: Resultssupporting

confidence: 73%

“…This work was done using paper-based consent materials with the written text read aloud by a research assistant. In analyses reported elsewhere, we found that working memory, verbal memory, and oral presentation of information were significant predictors of understanding scores and that study participants were more likely to understand concrete elements of the trial, such as what they would need to do, but still struggled with abstract concepts such as randomization [38]. In the second aim, we examined the impact of a multimedia decision support tool for individuals with FXS who were deciding whether to participate in a hypothetical clinical trial.…”

Section: Introductionmentioning

confidence: 98%

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Supporting informed clinical trial decisions: Results from a randomized controlled trial evaluating a digital decision support tool for those with intellectual disability

McCormack¹,

Wylie²,

Moultrie³

et al. 2019

PLoS ONE

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BackgroundInformed 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.MethodsParticipants (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.ResultsNo 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.ConclusionsA 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.

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Section: Resultssupporting

confidence: 94%

Section: Resultssupporting

confidence: 89%

Section: Resultssupporting

confidence: 73%

Section: Introductionmentioning

confidence: 98%

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Supporting informed clinical trial decisions: Results from a randomized controlled trial evaluating a digital decision support tool for those with intellectual disability

McCormack¹,

Wylie²,

Moultrie³

et al. 2019

PLoS ONE

Self Cite

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“…A large body of research has examined the capacity to consent in individuals with Alzheimer or other forms of cognitive impairment [85][86][87][88], and a handful have examined decisional capacity in individuals with intellectual disability [89][90][91]. This literature, combined with the ethical, legal, and social issues raised in this study, will help to inform the proper consent models for EHR researchers interested in recruiting participants who have a genetic condition that may result in intellectual disability.…”

Section: Summary Of Findingsmentioning

confidence: 99%

Ethical, Legal, and Social Issues Related to the Inclusion of Individuals With Intellectual Disabilities in Electronic Health Record Research: Scoping Review

et al. 2020

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Background Data from electronic health records (EHRs) are increasingly used in the field of genetic research to further precision medicine initiatives. However, many of these efforts exclude individuals with intellectual disabilities, which often stem from genetic conditions. To include this important subpopulation in EHR research, important ethical, legal, and social issues should be considered. Objective The goal of this study was to review prior research to better understand what ethical, legal, and social issues may need further investigation when considering the research use of EHRs for individuals with genetic conditions that may result in intellectual disability. This information will be valuable in developing methods and best practices for involving this group in research given they are considered a vulnerable population that may need special research protections. Methods We conducted a scoping review to examine issues related to the use of EHRs for research purposes and those more broadly associated with genetic research. The initial search yielded a total of 460 unique citations. We used an evaluative coding process to determine relevancy for inclusion. Results This approach resulted in 59 articles in the following areas: informed consent, privacy and security, return of results, and vulnerable populations. The review included several models of garnering informed consent in EHR or genetic research, including tiered or categorical, blanket or general, open, and opt-out models. Second, studies reported on patients’ concerns regarding the privacy and security of EHR or genetic data, such as who has access, type of data use in research, identifiability, and risks associated with privacy breach. The literature on return of research results using biospecimens examined the dissension in the field, particularly when sharing individualized genetic results. Finally, work involving vulnerable populations highlighted special considerations when conducting EHR or genetic research. Conclusions The results frame important questions for researchers to consider when designing EHR studies, which include individuals with intellectual disabilities, including appropriate safeguards and protections.

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Evaluating an informed consent process designed to improve inclusion of adults with intellectual disability in research

Strickler¹,

Havercamp²

2023

Research in Developmental Disabilities

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Decisional Capacity for Informed Consent in Males and Females with Fragile X Syndrome

Cited by 7 publications

References 40 publications

Supporting informed clinical trial decisions: Results from a randomized controlled trial evaluating a digital decision support tool for those with intellectual disability

Supporting informed clinical trial decisions: Results from a randomized controlled trial evaluating a digital decision support tool for those with intellectual disability

Ethical, Legal, and Social Issues Related to the Inclusion of Individuals With Intellectual Disabilities in Electronic Health Record Research: Scoping Review

Evaluating an informed consent process designed to improve inclusion of adults with intellectual disability in research

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