Research on individuals with intellectual and developmental disabilities continues to be an underinvestigated area of medicine. Even after the Surgeon General's report (Office of the Surgeon General, 2002) emphasizing the need to develop specific, achievable goals to improve the quality of life of people with intellectual and developmental disabilities, there is insufficient research with these individuals.
Now more than ever, it is imperative that adequate research efforts be focused on the population of individuals with intellectual and developmental disabilities in order to better understand and define their specific needs, and, ultimately, to improve their quality of life. With the evolution of modern medicine and the introduction of novel medical concepts and treatments, these individuals are living longer (McCausland et al., 2010), but they continue to face clear challenges in maintaining good health because they have poor health status compared to individuals in the general population (Lennox, Beange, & Edwards, 2000). Researchers have shown that the needs of this population differ significantly from those of the general population and that they are at an increased risk of developing comorbidities. However, their specific needs have not been well-defined (Parish, Moss, & Richman, 2008). To date, few large epidemiological studies have been conducted to assess the prevalence of specific medical conditions in individuals with intellectual and developmental disabilities (Beange, McElduff, & Baker, 1995). Furthermore, only 14% of individuals with intellectual and developmental disabilities currently live in public or private institutions (Smith, Lakin, Larson, & Salmi, 2011), making community-based research increasingly important.
Spasticity Research With Individuals Living in Institutional Settings
Individuals who have intellectual and developmental disabilities and cerebral palsy, traumatic brain injury, and spinal cord injury are especially vulnerable to developing a neurological condition called spasticity, which can result from any damage to the central nervous system. When left untreated, spasticity can cause serious health problems, and its impact in people with intellectual and developmental disabilities can be considerable. Therefore, our research group previously investigated the prevalence of spasticity in individuals with intellectual and developmental disabilities living in an institution by surveying all residents of a single large state-operated developmental center (Charles et al., 2010; Pfister et al., 2003). Thirty-five percent of residents at that institution were found to have spasticity, which in most cases (75%) significantly interfered with one or more functions or care tasks. A comprehensive spasticity clinic was developed in which standard spasticity therapies, including neurotoxin injection, intrathecal baclofen, and physical/occupational therapy, were used and an outcomes study (Charles et al., 2010) documented the results. One year after treatment initiation, range of motion and muscle tone measured by the Modified Ashworth Scale (Bohannon & Smith, 1987) were improved. More importantly, the length of time to complete a care task and the perceived difficulty in performing this task were significantly decreased for incontinence care, dressing, wheelchair positioning, transfer, and hygiene, as rated by direct caregivers who were not aware of the purposes of this study. The key finding from the Charles et al. study was that meaningful improvements in function and care delivery could be achieved by treating spasticity in people with intellectual and developmental disabilities. Furthermore, spasticity of average duration over 40 years that had, in many cases, already caused significant musculoskeletal changes, was responsive to treatment.
Spasticity Research in the Community
Although the deinstitutionalization movement has been overwhelmingly beneficial in many respects, its effect on access to appropriate care and other factors that influence health (and, in turn, quality of life) is unknown. Previous reports have suggested that these individuals have poor access to medical care, especially in specialties such as neurology. This may be partially due to the fact that, unlike large institutions, community homes do not have medical directors who routinely assess the residents' health status. Movement to the community may also be associated with variations in disease severity or burden. Because individuals with the most severe disabilities typically reside in institutions (Evenhuis et al., 2004), for instance, it is reasonable to hypothesize that the prevalence of spasticity would be lower in community-dwelling individuals.
Building on our prior research in institutions, we designed a subsequent study to investigate the prevalence of spasticity and need for treatment in a community setting. All adults receiving support from the Middle Tennessee Division of Intellectual Disabilities Services and living in community-based dwellings in Davidson County, Tennessee, were eligible for participation. After obtaining approval from the Vanderbilt University Institutional Review Board (IRB) and Middle Tennessee Division of Intellectual Disabilities Services, an explanatory letter and informed consent document were sent to 100 potential participants (or their legal guardian, if applicable) selected randomly from a list of 986 people. Over the course of this study, however, we encountered various barriers that resulted in only 7% of informed consent documents being returned. Ultimately, we were unable to achieve target enrollment, forcing the study to close prematurely. Although no meaningful conclusions regarding the prevalence, impact, or access to care for spasticity in people with intellectual and developmental disabilities are possible, our experience did illustrate important barriers to informed consent for research participation (and, in turn, improved health care delivery) that warrant investigation.
Although we did not receive much communication from proxy decision-makers, several decision-makers for potential subjects called our office to discuss the study. We were not in any way seeking to make a statement for or against deinstitutionalization, but several decision-makers were very apprehensive about the study and were concerned that the purpose of the study was to oppose deinstitutionalization. These conversations may reflect an underlying suspicion of research studies with individuals who have intellectual and developmental disabilities; this skepticism is not without reason. Unethical and unmonitored research with this population was common until 1979, when the National Commission issued the Belmont Report (Bird, 2010). This lingering distrust in research demonstrates the critical need to educate the general public, especially decision-makers for individuals with intellectual and developmental disabilities, on the benefits and imperative nature of researching the needs of these individuals and to re-instill confidence in medical research.
Barriers to Conducting Research in the Community
One of the most significant barriers we encountered relates to informed consent discussions, which are especially difficult with adults who have intellectual and developmental disabilities. Because these individuals are uniquely vulnerable, investigators aiming to conduct research with this population must take special care to protect patient privacy and ensure that potential participants are fully informed about research procedures and associated risks and benefits (Nunez-Wallace, Gill, Harrison, Taylor, & Charles, 2010). An augmented informed consent document and process should be designed to ensure these goals are achieved, but these precautionary measures lengthen and complicate the informed consent process (Ouellette-Kuntz et al., 2005). Ultimately, the consent process designed to protect adults with cognitive impairments often prevents these individuals from participating in meaningful research aimed at improving health and quality of life (Ouellette -Kuntz et al., 2005).
Another barrier to informed consent involved communication with multiple individuals and small community-based facilities. The ability to establish relationships with a single medical director and to organize large staff meetings to provide education and training and address concerns was instrumental in the success of our previous research at a state-operated developmental center. In previous studies researchers have found greater success in enrollment and completion of research procedures when there is a central point of contact within the care facility (Lennox et al., 2004). Medical directors significantly help with the informed consent process because they see patients on a daily basis, have been involved in patient care for long periods of time, and are often considered neutral, unbiased health providers. Proxy decision-makers may, therefore, more openly approach the informed consent process. In contrast, we encountered difficulty when attempting to establish relationships with caregivers, patients, and proxy decision-makers necessary to conduct community-based research. This may be true in both research and delivery of standard care; coordination with community-based health care systems is perceived as more difficult because specialist physicians do not have influence within the organization and have little administrative help (Evenhuis et al., 2004). In fact, staff in community settings may be suspicious of outside providers and the intent of the research and may actually act as an additional barrier that physicians must overcome in order to access patients (Lee, 1993).
This study was also affected by the heavy workload often encountered by caregivers. Individuals with intellectual and developmental disabilities may rely on caregivers for direct care and assistance with activities of daily living. Because there is often only a single caretaker responsible for people living in each home, community organizations and staff generally face difficult and overwhelming workloads (Lennox et al., 2005). Because workers in community settings have less time and resources than those in an institutional setting, they are understandably reticent to accept optional responsibilities with no immediate benefit, such as research participation. Thus, researchers must allot additional time to educate caregivers on the value of the study to the participants (if any) and possible contributions to medical knowledge.
One final barrier unique to intellectual and developmental disabilities research is that investigators must often rely on others for crucial procedures. For example, in the present study, the university IRB required that initial contact with potential participants be made by the state rather than the study team in order to protect privacy. The Middle Tennessee Division of Intellectual Disabilities Services graciously accommodated this request and assigned an employee to mail out the informed consent document, but this individual was not a trained research professional and did not possess the knowledge required to answer questions about spasticity, our aims for the study, or the potential contribution of the study to medical knowledge. Further, the caregiver was asked to perform this time-consuming task in addition to regular responsibilities. Patients are more likely to consent to a study when the research team meets with them personally to discuss study procedures and answer questions (Lennox et al., 2004), which we were prohibited from doing prior to enrollment. Had patients been contacted by a member of the research team invested in completion of the project and knowledgeable about the study and the medical condition to be investigated, it is likely that the enrollment rate would have been higher. Although this subject recruitment process was put in place to protect the privacy of potential participants, it ultimately contributed to the failure of the study.
While attempting to conduct community-based research (and ultimately address unmet healthcare needs), we encountered significant barriers that affected the completion of the study. We are not the first researchers to encounter extreme difficulties in recruitment of subjects with intellectual and developmental disabilities. Siegel and Ellis (1985) found that 11 of 42 investigators encountered recruitment difficulties due to restricted access to subjects with intellectual and developmental disabilities. In a separate clinical trial investigating the effectiveness of antipsychotic drugs in adults with aggressive challenging behavior living in the community, Oliver-Africano, Murphy, and Tyrer (2009) were able to recruit only 86 subjects; their theoretical maximum enrollment was 1,800. They concluded that researchers in very few randomized studies with individuals who had intellectual and developmental disabilities were able to recruit more than 100 subjects. In order to include community-dwelling adults with intellectual and developmental disabilities in meaningful evidence-based research, investigators must aggressively initiate progressive and purposeful change. The solutions we propose center around three basic principles: education, trust, and public support and advocacy for clinical research.
First, investigators seeking to begin research in the intellectual and developmental disabilities population must (a) understand the greater importance of educating participants, decision-makers, and caregivers regarding the aims of individual projects, the conditions they seek to treat, and the potential risks and benefits of participation; (b) acknowledge that the risk–benefit ratio of a particular study may be very different from a procedurally identical study conducted in a nondisabled population due to poorer overall health (Krahn & Drum, 2007), multiple concurrent therapies, less convenient research environments, and different definitions of clinically meaningful treatment benefit; it is the responsibility of investigators to educate participants on the benefits of participation and knowledge that directly apply to the population of individuals with intellectual and developmental disabilities; (c) expect and factor into research protocols and budgets the increased time and resource burden of managing a potentially complicated and demanding subject recruitment process; investigators should be aware of these difficulties prior to beginning a study so that they can plan accordingly and anticipate problems; (d) work with IRBs to agree upon a subject recruitment process specific to the study that offers potential subjects the opportunity to participate in meaningful research while preserving potential subject privacy and avoiding coercion; (e) personally meet with members of the organizations responsible for the subject registries in order to identify potential key workers within the organization and develop effective working relationships with them; these individuals could assist the researchers with subject recruitment and help ensure that the study recruited an adequate number of subjects (Lennox et al., 2005).
Second, in accordance with the Surgeon General's Report identifying healthcare barriers, every effort should be made to include individuals with intellectual and developmental disabilities, their caregivers, and their family members in the design and conduct of clinical research. The needs of these individuals should not be overlooked because they play integral roles in successful recruitment of subjects for the study.
Finally, investigators must have the opportunity to personally discuss their research with potential participants and their proxy medical decision-makers so the consent process will result in a personalized, unbiased, and informed decision based on a complete understanding of study procedures and potential contributions to the scientific community.
Research focused on community-dwelling adults with intellectual and developmental disabilities is increasingly important given the continuing transition to group homes. Differences in severity of intellectual and developmental disabilities, environment, access to care, and overall state of heath limit generalizability of previous research conducted in institutions to these patients. Without research that specifically targets community-dwelling individuals with intellectual and developmental disabilities, their unmet needs cannot be identified and addressed, even though in many cases there are already practical and readily available solutions. Our experience illustrates that inclusion of this underserved population in well-intentioned and meaningful research can be impaired by logistical issues and reluctance to participate stemming from misconceptions held by investigators, patients, family members, and caregivers. In accordance with Lennox et al. (2005), efforts to reach out to this community should not be abandoned despite these difficulties. The potential benefits of doing research with individuals who have intellectual and developmental disabilities are more important than the inconvenience and difficulty imparted on research personnel during subject recruitment.
This study was funded by an unrestricted research grant to Vanderbilt University from Allergan, Inc., Irvine, CA. Charles, who served as the principal investigator, has received income from Allergan for education and consulting services. The results of this study do not necessarily support the views of Allergan or its representatives.
Anna L. Molinari, BA, Research Associate, Vanderbilt University Medical Center, 1161 21st Ave. S., Nashville, TN 37232. Chandler E. Gill, BS, Medical Student, Stritch School of Medicine, Loyola University, 2160 S. First Ave., Maywood, IL 60153. Henry M. Taylor, MD, Medical Director, Clover Bottom Developmental Center, Middle Tennessee Division of Intellectual Disabilities Services, 275 Stewart's Ferry Pike, Nashville, TN 37214. P. David Charles, MD (corresponding author e-mail: firstname.lastname@example.org), Chief Medical Officer, Vanderbilt Neuroscience Institute, Associate Professor and Vice-Chairman of Neurology, Assistant Dean for Admissions, Vanderbilt University Medical Center, 1161 21st Ave. S., Suite A-1106 MCN, Nashville, TN 37232.