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Applied Nursing Research
j ourna l homepage: www.e lsev ie r .com/ locate /apnr
Implementation challenges in end-of-life research with adults withintellectual and developmental disabilities
Teresa A. Savage, PhD, RN a,⁎, Teresa ThaliaMoro, AM, LSW, PhD candidate b, Jackelyn Y. Boyden,MS/MPH, RN c,Allison A. Brown, PhD d, Karen L. Kavanaugh, PhD, RN, FAAN e
a University of Illinois at Chicago College of Nursing, Department of Women, Children and Family Health Sciences, 845 S. Damen, Room 858, Chicago, IL 60612, USAb University of Chicago, School of Social Service Administration, 969 E. 60th Street, Chicago, IL 60637, USAc Capital Caring, Education Research, 2900 Telestar Court, Falls Church, VA 22042, USAd Department of Psychology, University of Illinois Urbana-Champaign, 603 E. Daniel St. (M/C 716), Champaign, IL 61820, USAe Wayne State University College of Nursing, Family, Community, and Mental Health, 5557 Cass Avenue, Detroit, MI 48202, USA
a b s t r a c ta r t i c l e i n f o
This project was made possible by the National InstiInstitute of Nursing Research (NINR) P30 grant NR010680⁎ Corresponding author. Tel.: +1 312 355 0210; fax: +
E-mail addresses: [email protected] (T.A. Savage), [email protected] (J.Y. Boyden), [email protected]@wayne.edu (K.L. Kavanaugh).
http://dx.doi.org/10.1016/j.apnr.2014.10.0020897-1897/© 2014 Elsevier Inc. All rights reserved.
Please cite this article as: Savage, T.A., et al.,disabilities, Applied Nursing Research (2014),
Article history:Received 4 August 2014Accepted 4 October 2014Available online xxxx
Keywords:Terminal careIntellectual disabilityResearch design
Although the 4million+people in theU.S.with an intellectual or developmental disability (I/DD) experience thesame life expectancy as those in the general population, end-of-life research including these individuals is lackingand can be difficult to implement. Aswill be described in this paper, it is possible to overcome barriers to success-fully include people with I/DD in end-of-life research. In this paper, the implementation challenges, feasibility,and implications for successful end-of-life research with individuals with I/DD using focus groups are described.Individuals with I/DD were able to discuss their experiences and views about end-of-life care. However, whilepeople with I/DD made valuable contributions to the focus groups, there were several modifications needed inorder to execute this study. In order to gain a complete picture of end-of-life care for people with I/DD, it isimperative to include them in research to the best of their ability. By anticipating issues related to recruitment,the consent process, setting, and support needs of participants, focus groups can be successfully implemented.
tutes of Health (NIH), National.1 312 996 [email protected] (T.T. Moro),du (A.A. Brown),
Implementation challenges in end-of-life reseahttp://dx.doi.org/10.1016/j.apnr.2014.10.002
© 2014 Elsevier Inc. All rights reserved.
1. Introduction
The 4.7millionpeople in theU.S.whohave an intellectual or develop-mental disability (I/DD) (Braddock, Hemp, & Rizzolo, 2009, p. 1) oftenexperience more complex health problems (Hogg, Juhlberg, & Lambe,2007; Hogg & Tuffrey-Wijne, 2008; Krahn, Hammond, & Turner, 2006;Strydom, Hassiotis, King, & Livingston, 2009) and require greater healthservices than the general population (Hogg et al., 2007). Many adultswith mild to moderate I/DD have an equivalent life expectancy (Bittleset al., 2002) and often experience the same diseases related to agingas those in the general population (Haveman et al., 2010). However,end-of-life research that includes the perspectives of people with I/DDremains limited (Tuffrey-Wijne, Hogg, & Curfs, 2007), with mostresearch involving only the perspectives of staff (Todd, 2013;Wagemanset al., 2013; Wiese, Stancliffe, Baladin, Howarth, & Dew, 2012) and isdifficult to implement. People with I/DD are considered vulnerablebecause their capacity to consent to research may be affected by theirdisability, and historically, people with I/DD have been exploited inhuman subjects research. It is often difficult to access vulnerable
populations because they are appropriately protected from exploitationthrough institutional regulations (Chiang, Keatinge, & Williams, 2001).Despite the potential barriers, it is imperative to include people withI/DD in end-of-life research. The objective of this article is to describethe implementation challenges, feasibility, and implications for success-ful end-of-life research with individuals with I/DD using a focus groupmethod. Specifically, issues surrounding participant access and recruit-ment, the consent process, levels of participant protection, communica-tion and understanding and setting will be described. Includingparticipants with I/DD in research is critical, and this study illustratesthat by successfully overcoming barriers, such research is both feasibleand scientifically rewarding.
2. Focus group methodology
Several scholars in the area of I/DDandEOLhave advocated for the useof qualitativemethodology in order to conduct researchwith peoplewithI/DD, whose perspectives often are not represented (Andre-Barron,Strydom, & Hassiotis, 2008; Kaehne & O’Connell, 2010; Llewellyn &Northway, 2008; Tuffrey-Wijne, 2003). This informed, inductive ap-proach is an appropriate first-step methodology to investigate a phe-nomenon with little empirical documentation (Patton, 1990). Focusgroups were used for this research because this methodology hasbeen successfully utilized in both end-of-life research (Aspinal, Hughes,Dunckley, & Addington-Hall, 2006) and in research with people withI/DD (Andre-Barron et al., 2008; Kaehne &O’Connell, 2010) andmay be
rch with adults with intellectual and developmental
2 T.A. Savage et al. / Applied Nursing Research xxx (2014) xxx–xxx
preferable to surveys for individuals with literacy issues (Boyden,Esscopri, Ogi, Brennan, & Kalsy-Lillico, 2009). This approach also facili-tates group synergy which allows investigators to gather a comprehen-sive range of experiences and can yield more information thanindividual interviews alone (Krueger & Casey, 2000).
3. Implementation issues
3.1. Participant access and recruitment
Accessing and recruiting people with I/DD can be challenging for anumber of reasons: (1) this population has been exploited in the past,and agencies and families are appropriately protective; (2) investigatorsmust rely on agencies, often underfunded and understaffed, for providingaccess to and aiding with recruitment; and (3) traditional methods foraccess and recruitment, such as flyers or media ads, may be ineffectivebecause of lack of access to media and literacy difficulties in thispopulation. The research team's previous experiences with the com-munity agencies expedited gaining access to individuals with I/DDand enabled the focus groups to be conducted at the agencies. In addi-tion, all of the research staff had prior experience working with end-of-life research and/or training in working with people with I/DDwhichminimized the need for training in conducting sensitive researchwith a vulnerable population.
In order to minimally disrupt clients' participation in their dayprograms, agency staff asked that the research team not come in tomeet and screen possible client participants. While this method ofrecruitment relies on uncompensated agency staff taking time out oftheir hectic schedules in order to facilitate recruitment, in many waysit provided an additional protection against coercion since staff wereable to inform participants of the study and participants could feel freeto decline. Agency staff did not enroll participants, but facilitatedrecruitment by approaching individuals that they believed met theeligibility requirements. Having agency staff approach clients reducedcoercion on behalf of the client to participate and avoided acquiescencetoward a newperson the clientmaywish to please. Clientswere eligibleto participate if they had experienced the death of someone close tothem within the last 2 years, but not earlier than 3 months ago, wereemotionally ready to discuss their loss, and had the ability to verballyarticulate their thoughts on death and dying in a group setting. Thetimeline of 3 months to 2 years was initially conceived to yield morecurrent, and therefore, perhaps more reliable memories of events.Having such a restricted timeline proved extremely challenging infinding eligible participants, so after the first focus group, when partici-pants relayed events from 15 or more years in the past, participantswere included if they were able to recall events and feelings surround-ing the death of their relative/friend.
4. Human subject protection issues
The staff was explicitly asked not to consult any records or performany assessments because this would be considered “engaging inresearch”. According to our institutional review board (IRB), whenagencies are engaged in research they must obtain their own FederalWide Assurance through the National Institutes of Health, which wouldhave authorized them to establish their own IRB. None of the agencieshad a need or desire to establish their own IRBs, so this step seemedtoo burdensome to them in order to help us screen research participants.It would also have required the agency staff to complete human subjectstraining. Provided the agency staff did not consult any client records orperform any client assessments, they could help in recruiting.
4.1. Consent process
Major challenges in the consent process included assessing clients'capacity to consent, and assuring that the process was free of coercion
Please cite this article as: Savage, T.A., et al., Implementation challenges indisabilities, Applied Nursing Research (2014), http://dx.doi.org/10.1016/j.ap
and acquiescence. Prior to study approval, the IRB requested detaileddescriptions of how IQand capacity to consentwould be assessed. Initialcapacity was presumed if the client was his or her own guardian, andunderstanding was further assessed during the consent process byasking open ended questions. For clients who had guardians, theconsent of the guardian was first obtained, followed by assent fromthe client. In addition, agency staff were asked to identify, using theirworking knowledge only, clients whom they believedmet the inclusioncriteria. In order to use vernacular familiar to the IRB, the investigatorsdescribed the potential client participants as having “moderate intellec-tual disability”. However, within the disability community the term“support needs” is preferred rather than using functional level. Thebroad functional category of “moderate”was usedwith the understand-ing that individuals with greater support needs would not be selectedby agency staff given the requisite cognitive and communicationabilities needed to participate.
The PI had a concern that the template language of the consent formscould be indelicate to the potential participants; the PI wanted to avoidthe phrase “You are being invited to participate in this research becauseyou have an intellectual disability…” The title on the consent form was“End-of-Life Care for People with Intellectual and DevelopmentalDisabilities”, but the template sentence was re-written to say “We areasking you to participate to hear your views on end-of-life care.” Therewere also references to being a user of an agency serving peoplewith intellectual/developmental disabilities rather than using a labelof intellectual disability.
In order tominimize the potential for coercion and acquiescence, theconsent process consisted of several phases. Agency staff initiallyapproached potential participants, and if the clients agreed to learnmore about the study, they were given a copy of the consent form andstudy information sheet. In cases in which participants were not theirown guardians, agency staff contacted the guardians, and once consentwas obtained, client participants were contacted for assent. Both theconsent forms and information sheets used less technical languagethan the standard IRB template consent form, and the informationsheet additionally used pictures. According to Krueger and Casey(2000), a minimum of four participants are needed for a focusgroup. Thus, once there were at least four clients expressing interestin participating, a focus groupwas scheduled at the agency. As a secondstep, prior to the start of the focus group, the research teammet face-to-face with each client in order to read the consent or assent form aloudand answer questions. This occurred during the first 10–15 minutes ofthe focus group. Once all questions were addressed and forms signed,the moderator reviewed the study, discussed the importance of notrevealing to others what was said in the group, and reinforced theclients' rights to refuse to answer any questions and withdraw fromthe study at any time. Ideally, more time should have been allotted forthe group since the consent process took at least 20 minutes.
4.2. Levels of participant protection
The research team built in six procedural levels of protection toensure the focus group experience would not be distressing, and tominimize participants' distress if it occurred. First, the agency per-sonnel were asked to select only individuals they believed wouldbe emotionally ready to discuss end-of-life issues without acutedistress. Next, participants were informed of the purpose of thefocus group and the nature of the questions. Third, the researchteam monitored the group for signs of distress, such as tears or agi-tated behaviors and planned to manage distress with compassionand discretion. For example, if clients became visibly sad, their emo-tions would be acknowledged, and the moderator would ask if theyneeded to take a break. Having multiple team members (PI, PD andRA) present in the focus group added a fourth level of protection,as they could co-facilitate and/or monitor the groups for signs of dis-tress. The choice to have multiple team members present was also
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made in order to minimize disruption to the group if a team memberneeded to attend to a distressed participant. A fifth level of protectionwas, with the client's permission, to inform the appropriate agencypersonnel if someone became distressed during the session. In stayingwithin the research role, the research team cannot provide therapy fordistressed participants. Thus, it was explicitly discussed with andapproved by each agency prior to recruitment that the agency staffwould provide any follow up or provide any referrals to mental healthresources if needed. (Of note, one agency declined to allow clients toparticipate in the study because of concern that participation could pro-voke complicated grief.) However, none of the participants exhibiteddistress during any of the focus groups. For the final step, themoderatorwould check in with everyone at the end of the focus group to provideclosure and make sure everyone was okay.
4.3. Communication and understanding
Individuals with intellectual disability may have difficulties withliteracy and in understanding abstract conceptual issues, such as death.The first issuewaswhether or not clients could comprehend the abstractconcepts associated with end of life, such as death, dying, advance careplanning, and grief, and be able to express their thoughts about thoseconcepts. The research team hired an experienced moderator who wasable to probe to get information and keep clients focused on the ques-tions at hand. Some clientswere not capable of expressing their thoughtsor tracking the conversation, but most were.
Despite careful planning, a few days prior to the first focus group, theresearch team was informed that two of the participants would needassistancewith eating lunch, and therewere two aideswho could providethe assistance. The PI contacted the IRB with this new information,submitted an amendment that included having the aides sign a confiden-tiality agreement thatwould allow them to remain in the roomduring thefocus group, and stressed the urgency of a response given the effort torecruit and schedule the focus group. Within two days, an expeditedreview of the amendment occurred and the confidentiality agreementform was approved. Anecdotally, it was fortuitous that one of the aideswas in the room as she was able to tell us what the client, whom shewas assisting, was saying since no one else in the room could understandthe client.
The second communication issue is related to capturing the discussionverbatim. Traditionally, focus groups are audio recorded and transcribedat a later time. This method was not ideal for this study because someindividuals with I/DD have articulation issues (for example, the clientwho no one but her aide could understand) which makes it difficult todiscern what they are saying from an audio recording. To ensure clearand accurate transcripts, computer-assisted remote transcription(CART) was used. A transcriptionist (or “live writer”) from an agencyproviding CART was present during the focus groups. This transcribercaptured 95% ormore of the conversation, and the cost was comparableto traditional off-site transcription. The transcriptionist and agencywere in full compliance with HIPAA and removed proper nounsand identifiable data before returning the transcripts. Prior to thefirst focus group, the PI spoke with the transcriptionist regardingher experience with people with I/DD. Since she had none, the PIgave her some background, discussed parameters of the focus group,and alerted her to some behaviors, like slight choking or regurgitationthat might occur while clients were eating. Later the transcriptionistvoiced her appreciation for the “heads up” as she was prepared for thedifficulties some clients had while eating, and was especially alert toarticulation problems.
4.4. Setting
In order to facilitate participation all of the groups were conductedwithin the agency setting that was both convenient and familiar toclients. In an attempt to minimally disrupt participant's workshop
Please cite this article as: Savage, T.A., et al., Implementation challenges indisabilities, Applied Nursing Research (2014), http://dx.doi.org/10.1016/j.ap
activities, groupswere scheduled during the lunch hour. Dietary restric-tions, such as diabetes or food allergies were considered when selectingthe meal, which consisted of sandwiches. Many of the individuals withI/DD were in wheelchairs, so the room had to have enough accessiblespace to accommodate them. Another consideration was the need tocool the room during the summer months. The available room waslarge and was cooled by a window air conditioner. There was concernthat the voices of the participants may not be heard with the air condi-tioner running; in addition, each focus group was digitally recordedusing two separate recorders as a quality measure to compare theaccuracy of the transcripts, and the noise of the air conditioner mightobliterate the recording. The research team arrived early enough torun the air conditioner, then it was turned off during the focus groupmeeting, and turned back on once the session ended.
5. Feasibility issues
Although most participants could follow the discussion, describetheir own experiences, and give their opinions, there were a few whowere unable or unwilling to participate. There was one participantwho frequently interrupted wanting to tell a story or talk about some-thing else and would leave and re-enter the room a few times; oneclient only wanted to discuss the deceased entertainer Michael Jackson;and one other client rested her head on the table and remained silentduring the session. There were a few other clients who had disruptivebehaviors at times, but made substantive contributions to the discus-sion, and the moderator was able to maintain the flow of the session.Demographic information was collected from each client at the timethe consent/assent forms were discussed, and the inability to provideinformation was one indicator that the client may not be able to partici-pate. However, we did not exclude anyone once they agreed to partici-pate, and they were given lunch and a $30 Target gift card. For futurestudies, though, it may be helpful to have a way to screen clients fortheir ability to participate, both in terms of their conceptual abilitiesbut also their communication. Given our IRB view that screeninginvolves “engagement in research” of agency staff, the research teamwould need access to do the screening in a way that would be leastdisruptive to the agency activities.
For this particular study, only one agency was used to recruitclients for focus groups. Given that useful data were obtained, andthe participants expressed their appreciation for the opportunity totalk about the topic of death, focus groups can be an appropriatemethod for end-of life research in this population. Despite one agencydeclining to allow their clients to participate, the research teambelievesit could be cathartic for the clients, if topics of death, dying, loss, andgrief are not ordinarily discussed. A balance should be found betweenthe paternalism of protecting clients and the dignity of risk in permit-ting clients to participate.
6. Conclusions and implications for research
In order to understand the end-of-life care experiences for peoplewith I/DD, it is important to include them in research. The focus groupmethod is a useful tool for learning the perspectives of people withIDD on end-of-life care and gives them an opportunity to informresearch while supporting one another by sharing their experiences.Whereas focus groups do present some unique challenges whenworking with people with I/DD, this method is also quite powerful. Itmay be possible that methods used with the general population indoing advance care planning for one's own death, such as living willsor durable power of attorney for health care could be adapted for usewith people with I/DD. Further research may identify best practices inEOL care. Careful attention to recruitment, the consent process, setting,and support needs is necessary for a respectful and successful focusgroup session.
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Acknowledgments
This publication was made possible by the National Institutes ofHealth (NIH), National Institute of Nursing Research (NINR) P30 grantNR010680 (XX and XX, co-PIs). Its contents are solely the responsibilityof the authors and do not necessarily represent the official views ofthe NINR. Thank you to Drs. X, XX and XX for their assistance in con-ceptualizing and implementing the research project.
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