Rewriting the Rules of Informed Consent
Historical and Ethical Perspectives on Research
Over the past 50 years , members of the Disability Rights Movement have advocated for equal opportunities and rights for all people with disabilities , from accessibility and safety in architecture to equal access to employment opportunities ; in the case of clinical trials participation , they are reclaiming an area in which they were once victimized . Research involving individuals with intellectual and developmental disabilities has a complicated history . The horrors of Nazi medical experimentation on this population may seem buried in the past , but , as recently as the early 1970s , individuals with disabilities were used involuntarily as research subjects . This history is fresh in the minds of researchers who have a deeply ingrained wariness of risking – or even appearing to risk – taking advantage of individuals with disabilities .
“ Ethicists are very protective because there was a long history of doing research on people who basically had no right to refuse , including handicapped children in state institutions ,” Jeffrey P . Spike , PhD , professor at the McGovern Center for Humanities and Ethics and former director of the Campus-Wide Ethics Program at the University of Texas Health Science Center in Houston , told ASH Clinical News .
Dr . Spike cited the Willowbrook State School in Staten Island , New York , as one example . From the late 1950s to the early 1970s , researchers at Willowbrook deliberately infected children with intellectual disabilities with live hepatitis virus to study the effects of gamma globulin treatment . They justified inoculating healthy children because 90 percent of the children in the school developed hepatitis soon after their arrival anyway .
Looking back at the Willowbrook experience , ethicists point out the immorality of the experimentation : Children were involuntarily injected with the virus , and they had no chance to benefit from the procedure – only the chance to be harmed .
“ Conducting ethical trials in this population means striking a balance between protecting potential participants who might be easily manipulated and respecting their ability to make their own decisions like anyone else ,” Dr . Spike explained . In larger institutions , investigators might call for an ethics consultation to help determine a patient ’ s capacity to consent or clarify a gray area in which participation in clinical research may be inappropriate .
“ Everything hinges on whether individuals can understand and appreciate the risks they are taking ,” he added . “ We never want participants to say afterwards that they really didn ’ t know what could happen or that something could hurt this much , or for such a long time .”
REFERENCE
Robinson WM , Unruh BT ( 2008 ). The hepatitis experiments at the Willowbrook State School . In : The Oxford Textbook of Clinical Research Ethics ( pp . 80 ). New York , NY : Oxford University Press .
“ Some patients want to be included because they ’ ve so often felt excluded from other facets of community life .”
— KATHERINE E . McDONALD , PhD
participating in research , Dr . McDonald ’ s group conducted individual interviews and focus groups with 16 adults . 3 In general , the participants were interested and even excited about the possibility of participating in clinical research , with a few caveats – they wanted to learn about the research from people they trust , choose the people with whom they would discuss the research , and talk with peers about the decision to enroll or not .
“ Some patients want to be included because they ’ ve so often felt excluded from other facets of community life , but they want to be seen as active , giving , civic-minded members of society ,” Dr . McDonald said . “ Even when they understand they may not personally benefit from the research , they want to be able to help other people who are similar to them .”
Like everyone weighing the decision of whether to enroll in a trial , though , they also want to know that the research is valuable . After reviewing a study with researchers , participants were asked questions to assess their level of understanding of the study ’ s objective ; if participants expressed that they felt the study would be void of benefit , they declined to enroll .
Trust also permeates the conversations Dr . Taub has with parents who are considering enrolling their children with DS in pediatric leukemia studies ; a good reputation and experience in the area are also deciding factors .
“ Parents of children with disabilities tend to talk to one another ,” he said . “ If the parents know you have experience and are invested in finding answers to help their child and other children , they will likely be more receptive to the idea of having their children participate in a study . It assures them that you ’ re not just ‘ experimenting ’ on their child .”
( Parents ’ hesitation to involve their children in clinical research – and , similarly , some researchers ’ hesitation to ask patients to participate – is rooted in the complicated history of research in this population . See the SIDEBAR for the historical and ethical perspectives on research for individuals with intellectual and developmental disabilities .)
The Downside of Overprotection
The attitudes of researchers , Institutional Review Board members , and those in charge of conducting and monitoring research play a substantial role in shaping the knowledge base about the treatment of individuals with disabilities . When Dr . McDonald and authors interviewed these “ gatekeepers of science ” about their opinions on greater trial inclusion , most want more people with disabilities to participate – again , with some caveats . 4
All survey respondents agreed that , to consent to enroll in a clinical trial , participants need to understand “ the prospect of encountering both benefits and burdens from participating in research ” and have the necessary support to participate . However , respondents expressed varying levels of comfort with the degree of autonomy and protections that research participants should be granted .
“ Gatekeepers who perceived greater similarity and shared humanity between people with and those without intellectual disability endorse opportunities for adults with intellectual disability to make decisions about and take part in research ,” the authors wrote . “ Conversely , gatekeepers who support the protection of adults with intellectual disability in general perceived a greater need [ for them ] to receive support specifically in making decisions about research participation and want to see them more protected therein .”
Competing factors are at work : Individuals with intellectual disabilities are potentially at risk for exploitation within the research process because of their limitations and relative social powerlessness . At the same time , avoiding these issues via blanket exclusion from research denies such individuals the opportunity to contribute to and benefit from scientific research .
The authors suggested that “ the tendency to protect adults with intellectual disability by assuming reduced autonomy ” may be doing more harm than good . “ Restrictive attitudes may create subtle barriers to community inclusion ,” they wrote .
Facilitating Inclusion
Making the clinical trial recruitment and enrollment process more accessible and inclusive is a relatively straightforward process , Dr . Raymaker said . “ It ’ s really a question of figuring out how you tailor your process to accommodate a population that is outside of the originally intended population .” But , she conceded , it “ can be difficult work to do .”
And , because the issue affects such a small percentage of the overall population , it can be hard to get people to care enough to try . Her hope is that the tools and tricks that have been developed by those intimately involved in this issue will “ go viral .” New researchers will learn from the work of experienced investigators , then pass along that knowledge to a few more researchers , and so on .
Enrolling individuals with intellectual disabilities takes extra time and effort ; for instance , modifying informed-consent forms with pictures or simplified written explanations ( see FIGURE , page 59 ). It can also require outside help , including family members or researchers who are specially trained to communicate with people with intellectual disabilities .
“ Maybe participants just need time to discuss participation privately with their parents or guardians ,” Dr . Raymaker offered . “ You need to give them the opportunity to do that .”
Parents and legal guardians can also serve as a proxy for patients to provide their consent to participate in a trial , much like the “ assent-consent process ” that is used for enrolling children into clinical trials .
Above all , investigators should keep in mind that “ the purpose of Informed consent is not to get individuals enrolled in the study , but rather to give them the information they need to make that decision for themselves ,” Dr . Raymaker said . — Debra L . Beck ●
REFERENCES
1 . Horner-Johnson W , Bailey D . Assessing understanding and obtaining consent from adults with intellectual disabilities for a health promotion study . J Policy Prac Intellect Disabil . 2013 ; 10 ( 3 ).
2 . Taub JW , Berman JN , Hitzler JK , et al . Improvement outcomes for myeloid leukemia of Down syndrome : a report from the Children ’ s Oncology Group AAML0431 trial . Blood . 2017 ; 129:3304-13 .
3 . McDonald KE , Kidney CA , Patka M , et al . ‘ You need to let your voice be heard ’: research participants ’ views on research . J Intellect Disabil Res . 2013 ; 57:216-25 .
4 . McDonald KE , Keys CB , Henry DB . Gatekeepers of science : attitudes toward the research participation of adults with intellectual disability . Am J Ment Retard . 2008 ; 113:466-78 .
60 ASH Clinical News September 2017