During an outcomes study of spasticity treatment at a developmental center for 62 residents with profound intellectual disabilities, either botulinum toxin A (BTX-A), intrathecal baclofen (ITB), or both were recommended with physical and occupational therapy. Conservators consented to BTX-A more than ITB (p = .021). Court-appointed conservators were more likely to provide consent for treatment than family members (p = .026). Nonparents consented more than parents (p = .009). Finally, Caucasian conservators were more likely to consent to treatment than African American conservators (p = .036), but ethnicity of the resident did not influence consent. Gender of resident or conservator did not influence rate of consent. This report highlights disparities in surrogate consent giving for individuals with intellectual disabilities and indicates a need for more research to ensure that this vulnerable population has access to appropriate treatments.
Intellectual disability is defined as a developmental disorder that begins before Age 18 years with intellectual functioning level below average, accompanied by limitations in function in two or more adaptive skill areas (American Psychiatric Association, 2000). Severity is classified by IQ scores: mild (55–70 IQ), moderate (40–55 IQ), severe (25–49 IQ), and profound (<25 IQ; American Psychiatric Association, 2000). The American Association on Intellectual and Developmental Disabilities (AAIDD) also defines intellectual disability as a disability characterized by significant limitations in intellectual functioning as well as adaptive behavior originating before Age 18. The adaptive behavior covers three skill types: conceptual, social, and practical skills. Conceptual skills include language and literacy, number concepts, and self-direction. Social skills include interpersonal skills, social responsibility, and self-esteem; and practical skills include activities of daily living. AAIDD stresses that in defining and assessing intellectual disability, professionals must consider community environment typical of the individual's peers and culture, linguistic diversity, and cultural differences in the way people communicate, move, and behave (AAIDD, 2009). Individuals with profound developmental disabilities can be severely restricted in mobility, incontinent, and/or require complete care and can experience other comorbid conditions at higher rates (Beange, McElduff, & Baker, 1995).
Spasticity is defined as a velocity-dependent increase in the stretch reflex and may limit motor control and functional performance, thereby contributing to increased disability (Tuzson, Granata, & Able, 2003). Spasticity is present in about one third of patients with intellectual disability living in developmental centers (Pfister et al., 2003). It is imperative to maintain comprehensive care in this population and address interventions for the prevention of morbidity and premature death (Sullivan et al., 2006).
In cases where persons with intellectually disability have legal conservators as decision makers, the presence of intellectual disability is not grounds for withholding treatment in indicated situations, and health care decisions must be made based on the patients' best interests through informed consent (Sullivan et al., 2006). Informed consent is a process by which a person authorizes medical treatment after understanding the nature, indications, risks, and benefits of the treatment (LeBlang, 1995). Racial disparities in access to health care, treatment, and participation in clinical trials remain significant (Blendon, Aiken, Freeman, & Corey, 1989; Bliss et al., 2004; Murthy, Krumholz, & Gross, 2004). African Americans' knowledge of a history of racial discrimination in medical research has resulted in a reluctance to participate in clinical trials, perhaps due to a lack of trust (Corbie-Smith, Thomas, Williams, & Moody-Ayers, 1999; Shavers, Lynch, & Burmeister, 2002; Shavers-Hornaday, Lynch, Burmeister, & Torner, 1997). There is a paucity of literature, however, regarding similar disparities among surrogate decision makers for persons with developmental disabilities.
From 2002 to 2006, we conducted an outcomes study at a state residential facility. The objective was to determine the functional improvement in individuals with intellectual and developmental disabilities after comprehensive spasticity treatment, and those results are published elsewhere (Charles et al., 2010). The purpose of this article is to describe the differences in surrogate consent rates with regards to demographic and relationship factors of individuals who were approached for participation in the outcomes study.
Our study focused on a residential developmental center that provided complete services for residents with intellectual and developmental disabilities. Every resident at the facility had a legally appointed, medical decision maker. The majority of these conservators were family members, most often parents. When a relative was not available to serve in this capacity, an unrelated person employed by an independent agency was appointed.
In 2002, a comprehensive spasticity management program was initiated to expand treatment options for residents at the center (Pfister et al., 2003). This program included initial examination by the study physician of every resident of the center to assess the presence and extent of spasticity. If spasticity was present, functional goals were identified by therapists and direct care staff familiar with the individual. A treatment recommendation was then given to each resident that included one or both of the following: botulinum toxin type A (BTX-A), which was administered through intramuscular injection and/or intrathecal baclofen therapy (ITB), which required a surgical procedure to implant a programmable pump that delivered medication through a catheter into the cerebral spinal fluid space. The conservator for each resident for whom spasticity treatment was recommended was initially contacted by letter from the center's medical director with two informed consent documents as enclosures. One consent form was for the recommended spasticity treatment, and the other was for participation in an outcomes survey designed to document and publish the impact of comprehensive spasticity treatment. The letter emphasized that participation in the outcomes survey was not required to receive treatment. An invitation was included for the conservator to call or visit the center's medical director with any questions about the recommended treatment or survey participation. If the medical director did not receive a response from the conservator within several weeks of the initial letter, telephone contact was attempted. If this attempt failed, a second letter was sent. Last, if the conservator could not be contacted by this manner, they were considered to have declined consent. All conservators were approached in an identical manner by the same medical director without regard to race or gender. Survey enrollment began in March 2002 and closed in November 2004. All residents of the center had comprehensive health insurance. The consent status reported here reflects the status at the end of the enrollment period. Results were analyzed using the chi-square test with one degree of freedom.
There were 205 people living at the developmental center when the spasticity treatment program was first implemented. Of these, 93% had been diagnosed with profound intellectual disability. Each resident recommended for spasticity treatment had a legally appointed conservator who was either nonrelated or a family member. All people consenting to treatment also consented to survey participation. There were six variables considered: treatment recommended, relation to resident (legally appointed conservator, parent, and nonparent), race of resident, race of conservator, gender of resident, and gender of conservator. There were 42 recommended BTX-A treatments and 20 recommended ITB treatments. These numbers include the 12 recommended treatments that consisted of both BTX-A and ITB. Of those 12, six conservators consented to both treatments, three conservators denied consent for both treatments, and three conservators consented to BTX-A but not ITB. For purposes of data analysis, combined treatments were considered as individual data points and consent rates were calculated for BTX-A and ITB. Conservators gave consent to BTX-A significantly more often than ITB (83.3% vs. 55.0%, p = .021) (Table 1). There were 51 conservators who were family members, and 11 who were nonrelated conservators. Nonrelated conservators gave consent significantly more often than family members (100% vs. 68.6%, p = .026). There were 21 conservators who were nonparents, including family members and legally appointed conservators, and 41 who were parents. Nonparents gave consent significantly more often than parents (95.2% vs. 65.9%, p = .009). Out of the 51 conservators who were family members, 10 were family but nonparents and 41 were parents. Family members who were nonparents did not give consent significantly more often than parents (90% vs. 65.9%, p = .13). Out of the 62 residents, 19 were African American, 41 were Caucasian, 1 was Asian American, and the remaining individual's race was not available. The latter 2 were excluded from these analyses due to small sample size and missing data, respectively. The consent rate for Caucasian residents was not significantly different than the consent rate for African American residents (80.5% vs. 63.2%, p = .132). Out of the 62 conservators, 59 had available information about race. There were 15 African American and 44 Caucasian conservators. Caucasian conservators gave consent more often than African American conservators (81.8% vs. 53.3%, p = .036). Out of the 62 residents, 36 were female and 26 were male. Consent was granted for 80.6% of females and 69.2% of males (p = .23). There were 60 conservators whose gender information was included; 2 residents had both parents as medical decision makers, and these 2 were not included in the gender analysis. Out of these, 42 were female and 18 were male. Fifteen (83.3%) male conservators and 71.4% of female conservators granted consent (p = .263).
There are significant disparities in consent rates of the different groups we analyzed. Conservators who were legally appointed were much more likely to give consent than conservators who were family members. In both cases, the expectation of a surrogate decision maker is to act in the individual's best interest. The difference in consent rates between family and legally appointed conservators could be attributed to the family member's relation to the resident, which would encompass emotional and cultural values related to the intimacy of this relationship (Corbie-Smith et al., 1999; Emanuel & Emanuel, 1992; Murthy et al., 2004). In clinical practice, physicians commonly rely on the next of kin as decision makers for patients who are not able to represent themselves, and patients usually prefer their next of kin to decide for them if they cannot do so (Veatch, 1984). In the United States, some states give relatives decision-making rights without being legally appointed (Schoeman, 1985). However, increasing disruption of traditional family structures might limit the appropriateness of this system and pose a high risk for abuse (Puchalski et al., 2000). Although the implications of having a family member versus a legally appointed conservator as the decision maker for an individual who is intellectually disabled are beyond the scope of this article, our results underscore the importance of this area of study.
Conservators were more likely to consent to BTX-A than ITB, a more invasive treatment. This was true whether residents were recommended for a single treatment or both. Intuitively, an option to forego additional risks, which one would expect from a surgical procedure, would result in a preference for BTX-A.
Another important finding in this research was the effect of race on the likelihood of giving consent for appropriate treatment. It was reassuring that the race of the resident was not associated with a difference in consent giving. The race of the conservator, however, manifested an important influence. Caucasian conservators were more likely to give consent than African American conservators. Unfortunately, racial disparities in proxy decision making have not previously been studied in people with intellectual and developmental disabilities. There have been several studies that have documented a lack of enrollment of African Americans in clinical trials and distrust in the health care system, perhaps due to ongoing patters of discrimination (Blendon et al., 1989; Bliss et al., 2004; LeBlang, 1995; Sullivan et al., 2006). Minorities are more likely to express concerns about exploitation, dishonesty regarding risks of experimental treatment, and motivations of researchers (American Medical Directors Association, 2007; Bliss et al., 2004; Puchalski et al., 2000). Some studies have suggested that socioeconomic factors are associated with limited access to clinical trials due to a low income and general limited access to health care (Sullivan et al., 2006). However, in a study of participation in cancer clinical trials, African Americans were well represented in prostate cancer trials compared with breast, lung, and colorectal cancer trials (Blendon et al., 1989).
Due to the sample size of this study, it was not possible to test all variables as independent predictors of consent decision. This highlights the importance of larger studies on the topic. There is a paucity of literature on disparities in consent-by-proxy in people who have intellectual and developmental disabilities. If the disparities seen in this small study are consistent with future large studies, it will be imperative that the process of surrogate informed consent be reevaluated to ensure that conservators are making the most appropriate health care decisions based on all available information.
Although we discuss only consent to spasticity treatment in this brief article, there is a broad spectrum of scenarios where informed consent-by-proxy applies, including research trial participation and life-sustaining therapy. We mentioned that this study showed that African Americans were significantly less likely to give consent and that there have been other studies that show this population is less likely to participate in research trials. It can, therefore, be hypothesized that African Americans may be less likely to give informed consent to research trial enrollment of individuals who are intellectually disabled, whether they are family members or unrelated court-appointed conservators. This could hold true even for minimal risk studies, such as questionnaires or medical record reviews. Another scenario implicated is life-sustaining therapy. Because our sample size was small, we can only speculate that related conservators or African American conservators would be less likely to consent to life-sustaining therapy in cases where they might find further treatment futile. This could potentially be an open door, however, to neglect in persons with intellectual and developmental disabilities, where the meaning of futile can often be subjective and highly controversial.
In summary, we found significant disparities with regards to informed consent-by-proxy at the developmental center in this study. Among these disparities were the relation of the conservator to the resident and race of the conservator. To our knowledge, this is the first report to address these issues in individuals with intellectual disabilities, and the results highlight the need for larger studies of the influence of race and familial relationship on the willingness of conservators to consent to medical procedures and participation in medical research.
Editor-in-Charge: Steven J. Taylor
Karen R. Nunez-Wallace, MD, Resident Physician, Department of Neurology, Baylor College of Medicine, Houston, TX 77030. Chandler E. Gill, Medical Student, Loyola University Chicago, Stritch School of Medicine, Maywood, IL 60153. Courtney H. Harrison, Medical Student, Vanderbilt University School of Medicine, Nashville, TN 37232. Henry M. Taylor, MD, Medical Director, Clover Bottom Developmental Center, Nashville, TN 37214. P. David Charles, MD (E-mail: email@example.com), Associate Professor and Vice-Chair, Department of Neurology, Vanderbilt University Medical Center, 1161 21st Ave. South, Suite A-1106 MCN, Nashville, TN 37232.