Perez, Lori; Huang, Jennifer; Jansky, Liz; Nowinski, Cindy; Victorson, David; Peterman, Amy; Cella, David
Health‐related quality of life (HRQL) is a multidimensional construct (Aaronson et al., 1991; Spitzer, 1987) and is an important outcome in neurology clinical care and clinical trials (Murrell, 1999). Usually applied to ill populations (Spitzer), the concept of HRQL evokes a sense of holism by spanning domains such as social and emotional functioning, the experience of diseasespecific symptoms and physical limitations, and an individual's general outlook on life. Similarly, the World Health Organization (WHO; 1946) defines health as a condition of total well‐being, inclusive of multiple domains of well‐being, and not only the lack of disease. WHO has developed the following domains as constituting HRQL: physical functioning; social functioning, including relationships with others, and role functioning; and emotional or mental state (WHO, 1946). Researchers in the field agree that assessments derived to measure HRQL should evaluate the actual or perceived impact of a disease or its treatment on at least some aspects of these domains (Cella, 1992, 2000; Cella & Bonomi, 2003) and “in terms that matter most to… people, such as the capacity to function in everyday life” (Ware, 1993, p. 53). In this way, HRQL is, in part, a subjective evaluation, and patient input is a critical source of information about HRQL.
HRQL assessments are particularly important in the evaluation of neurology clinical care and clinical trials, since many neurological disorders are incurable and chronic, and they cause impairment in multiple domains of functioning. Many disorders often progress gradually, with unpredictable and inconspicuous changes in function. As a result, healthcare professionals may underestimate or fail to recognize the full scope of the impact of neurological diseases and related treatment on their patients' HRQL. Despite the relevance of HRQL assessment for neurological illness, the status of HRQL research with neurological populations is relatively underdeveloped, and there remains a paucity of literature on the multidimensionality of HRQL, especially regarding the impact of neurological conditions on various domains affecting the patient (Murrell, 1999).
As a response to this predicament, the National Institute of Neurological Disorders and Stroke (NINDS) has funded a study called Quality of Life Outcomes in Neurological Disorders (Neuro‐QOL), with the objective of constructing a clinically relevant and useful HRQL measurement system for major neurological disorders that affect the U.S. population. This measurement system must be consistent enough across the selected conditions to allow for cross‐disease comparisons and yet flexible enough to capture condition‐specific HRQL issues. To accomplish this, the study aims to develop, modify, and test item banks, or finite sets of questions, that are designed to assess common concepts that traverse virtually all selected diseases. Separate sets of unique, targeted scales will be developed and field‐tested to augment the generic item banks as a means of evaluating symptoms, concerns, or issues that are relevant only to a subset of diseases or treatments. The primary end users of this measurement system will be clinical trialists and other clinical neurology researchers; however, the item banks will also be appropriate for use in clinical practice.
The primary aim of this article is to present findings from a critical step in the Neuro‐QOL instrument development process: the focus group discussions with patients affected by a representative cross section of neurological conditions and their caregivers. As the Food and Drug Administration has noted, the development of patient‐reported outcome instruments is incomplete without patient input (U.S. Department of Health and Human Services, Food and Drug Administration, Center for Drug Evaluation and Research, Center for Biologics Evaluation and Research, & Center for Devices and Radiological Health, 2006). The importance of patient perceptions in assessing HRQL implies that the instrument must be developed using information from patients themselves. The development process must incorporate the input of a wide range of patients with the condition of interest to ensure that the conceptual framework and instrument items are appropriate to that population with respect to age, sex, ethnic identity, cognitive ability, and life experience (U.S. Department of Health and Human Services et al.). Otherwise, the instrument may omit significant information that is important from the patient perspective (Donovan, Sanson‐Fisher, & Redman, 1989).
This article highlights the findings that emerged from patient and caregiver focus groups in relation to HRQL. Specifically, focus groups were conducted across a variety of neurological conditions with patients representing a range of symptomatology and ages. Although several studies have investigated HRQL within a particular neurological condition, significantly fewer studies have examined HRQL across multiple disorders (Arnold et al., 2004). Since different neurological conditions may have a differential impact on HRQL, we included several neurological conditions within one study to facilitate the conceptualization of domains that are common across several neurological disorders, as well as those that are unique to a specific disorder. The authors sought to confirm relevant, and highlight missing, domains in the conceptual framework proposed for the Neuro‐QOL instrument from the patient perspective and provide the research team with direct access to the language participants use when describing HRQL (Detmar et al., 2006; Kitzinger, 1995). Through firsthand accounts and summaries of themes discussed by patients and caregivers, this study offers a unique examination of the experiences and social and environmental construction of HRQL for patients with targeted neurological conditions.
A total of 11 focus groups were conducted. Of these 11 groups, 7 were patient focus groups, representing Alzheimer disease, multiple sclerosis (MS), amyotrophic lateral sclerosis syndrome (ALS), Parkinson disease, stroke, and adult and pediatric epilepsy conditions. Two groups were conducted with MS patients. The research team also conducted focus groups with caregivers of Alzheimer disease, stroke, and pediatric epilepsy patients because these patients may be unable to reliably report their subjective perceptions of HRQL due to cognitive impariment or age. In total, 83 people (64 patients, 19 caregivers) participated in the focus groups. Of the patients, 32 were males and 32 were females; of the caregivers, 7 were males and 12 were females.
The age range in the adult patient focus groups was 25‐84 years (M = 55.41, SD = 13.70), and for the pediatric patient group, it was 14‐20 years (M = 15.83, SD = 2.23). For the adult caregiver groups, the age range of their care recipients was 40‐82 years (M = 62.75, SD = 9.63). For the pediatric caregiver group, the age range of their care recipients was 11‐18 years (M = 13.17, SD = 2.79).
Focus Group Guides
Semistructured focus group guides were developed to assess participants' experiences related to HRQL, specific to three participant populations: pediatric patients, adult patients, and caregivers of patients. All guides began with broad, open‐ended questions, allowing participants to free‐list responses for their definition of HRQL and what areas of HRQL are most impacted by their disease, the treatment, or both. The guide then progressed to questions regarding specific HRQL domains, such as physical functioning, emotional functioning, social aspects, and treatment effects, which have been shown to be relevant in the literature. Caregivers were instructed to respond from the perspective of their care recipient.
Participants were recruited through various settings (e.g., hospitals, clinics, patient advocacy associations) in several metropolitan and rural cities across the United States to ensure a sample of participants that represented socioeconomic, geographic, and ethnic diversity. Physicians and clinic staff aided in the recruitment process by identifying and obtaining consent from prospective participants according to a standard protocol. Eligibility criteria were as follows: adult patients and caregivers were age 18 years or older, pediatric patients were between 12 years and 17 years, 11 months; patient or care recipients were diagnosed by a physician with one of the study conditions; and all participants were cognitively and physically able to participate in a focus group, as determined by recruitment staff. Prior to the focus group, all adult participants and parents of children in the pediatric focus groups completed a consent form, and pediatric participants completed an assent form. When applicable, physicians and clinic staff obtained HIPAA (Health Insurance Portability and Accountability Act) authorization from participants.
Conduct of focus groups.
Focus groups were disease specific, which provided a sense of rapport for participants, as they all experienced similar symptoms of the same condition. This also permitted moderators to ask more probing questions, allowing an in‐depth analysis per condition, as well as the ability to examine themes across conditions. An experienced moderator and comoderator facilitated the focus group sessions. All focus groups were completed within 2 hours. At the conclusion of the focus group, participants received a $50 cash incentive and a $10 travel reimbursement.
Audio recordings of the focus groups were transcribed verbatim and edited to remove identifiers. The audiorecording and transcription for half of the focus groups were then compared to ensure accuracy of transcription. Members of the research team read all of the transcripts thoroughly to become acquainted with the data and imported the data into NVivo 2.0, a qualitative data analysis and management software package.
Based on the general principles of grounded theory, two coders from the research team analyzed the data via three techniques: coding, memo writing, and the constant comparative method (Chamberlain, 1999; Glaser & Strauss, 1967). The coding phase of the analysis evolved through three stages: open, axial, and selective coding (Strauss & Corbin, 1998). During the open‐coding phase, coders broadly coded all passages of text relevant to the research questions. In the axial‐coding phase, coders refined the broad codes, deleted extraneous categories, and merged categories that were redundant. During the selective‐coding phase, coders further developed the codes through deliberate, directed selection and finalized categories and the relationships between them.
Throughout the coding process, the coders organized categories and subcategories into a codebook, containing the name of the code, a definition, and illustrative quotes. Coders met regularly to discuss the evolution of the codebook, confirm the accurate interpretation of the data, deliberate on codes that did not appear to fit into the original codebook, discuss the emergence of new categories, and rectify issues with the application of codes. With each iteration of coding, the codebook was refined, and codes were merged, expanded, or divided as agreed upon.
The research team also instituted memo writing throughout the analysis to assist with the maintenance and organization of codes, notes, and theory development. Memos were used to note any reflections, biases, questions, observations, or hypotheses that emerged from transcripts or codes. Throughout all phases of coding, we used the constant comparative method, a “hallmark of grounded theory studies” (Chamberlain, 1999, p. 187) that involves comparing incidents, participants, or categories and finding similarities and differences between them (Glaser, 1992). These comparisons are necessary to identify and develop categories, as well as to define relationships between categories. In addition, the research team evaluated the data against Lincoln and Guba's (1985) criteria for assessing the trustworthiness of qualitative research, including credibility and transferability.
By using more than one coder, the variance among coding results was reduced, and a level of control for haphazard or biased subjectivity in the analysis was achieved. An analysis of intercoder agreement checks determined that the coders arrived at the same or similar codes for the study's major themes and subthemes. Intercoder agreement checks were performed on a portion of transcripts. When discrepancies were found, differences were discussed until consensus was reached among the coders and other research team members, and coded text was recoded accordingly.
The themes that emerged from the data are presented in this section and are categorized into HRQL domains and subdomains. Outcomes across conditions are presented first and are followed by outcomes per condition. Although the majority of these themes are presented in the domains that are commonly cited in the literature (e.g., emotional functioning, physical functioning), numerous themes were found that could not be categorized into a singular “traditional” domain (e.g., loss of skills, loss of independence). By presenting the data according to domains, the authors do not wish to imply that these are orthogonal constructs, as the integrated and reciprocal nature of HRQL domains must be recognized.
HRQL Domain Outcomes Across Conditions
The following section narrows the presentation of results to those key domain themes that were endorsed at least once by four or more conditions. These themes, and additional subthemes expressed by a minority of conditions (i.e., three or less), are presented in Table 1, along with the percentages for each. Percentages were calculated as the number of times the theme's code was applied over the number of all codes applied during the focus group discussion. Themes that emerged for less than four conditions are not discussed in detail in the following section, and may warrant further research to understand why they were not mentioned by the majority of groups.
Participants across all focus groups discussed emotional function, which included the subthemes of embarrassment, denial, and anger, as described in the next sections.
Participants who were cognitively affected, such as those in the Alzheimer disease group, stated that they were often embarrassed because they “felt like an idiot” due to their inability to perform familiar tasks. On the other hand, participants who were more physically affected by their condition, such as those in the Parkinson disease or the MS groups, expressed feelings of embarrassment from exhibiting symptoms in public, such as tripping while walking or moving slowly while at a grocery‐store checkout line. For example, one participant from the MS group explained, “Sometimes I used to just drop things, like a glass … and it was just ‘oh whoops.’ It was embarrassing.” Participants from the stroke group also expressed embarrassment because their physical condition made them feel self‐conscious. This was particularly true if their ability to walk was affected, and many stroke caregivers concurred that their care recipients were often embarrassed and refused to use canes or walkers in public, even though they may have used them at home. Similarly, one MS participant stated, “It's like I don't want to bring my cart because everybody will look at me.”
A few participants in the adult epilepsy focus group discussed their feelings of embarrassment, particularly those who had frequent seizures in public or at work. This embarrassment led one participant to isolate himself. He stated, “I don't want to leave home. Less people to talk to, less people to see me make a fool out of myself. You know, it's embarrassing in public.”
Feelings of anger were discussed by numerous participants and expressed most often in the stroke, Alzheimer disease, and Parkinson disease groups.
Several focus groups discussed having experienced feelings of denial. For patients with Alzheimer disease, feelings of denial often occurred at the beginning of the disease progression, when patients retained a greater level of cognitive functioning. Denial was also reportedly a common experience during the beginning stages of MS, with acceptance of their condition occurring later. In contrast, adult epilepsy participants explained that denial continued to occur occasionally after the diagnosis of the condition. This often led patients to become nonadherent to their medication. As one adult epilepsy participant stated, “Okay, so there are times that we all will go into denial, and believe it or not, when you don't take [your medication], you feel good, because you're not drugged down.”
Feelings of anger were discussed by numerous participants and expressed most often in the stroke, Alzheimer disease, and Parkinson disease groups. Although some participants were angry at themselves for their inability to perform certain familiar tasks, others were angry at life for their misfortune. In particular, patients in the stroke focus group stated that they were extremely angry after the stroke, and that they unleashed their anger on their caregivers, particularly when their caregivers tried to provide assistance. Others were angry at the stigma or prejudice they experienced, such as some patients with Parkinson disease who expressed anger when family or friends associated their condition with cognitive decline and impairment.
Physical functioning was a major theme across all of the adult conditions, although to a lesser extent in the Alzheimer disease groups. The subtheme that spanned the majority of focus group discussions was related to gross motor function; however, additional subthemes are also described in the following sections.
Gross motor skills.
Participants representing four conditions (MS, Parkinson disease, ALS, and stroke) felt that their gross motor skills were compromised as a result of their disease. The ability to run, drive, walk, and use stairs was typically affected, but limitations often manifested differently for various conditions. For example, several ALS participants described struggles with leg movements, such as climbing stairs, getting in and out of cars, running, and driving. Examples provided by the MS patients often were related to a loss of feeling and numbness that ranged from interfering with gross motor activities, such as running and walking, to paralysis. Stroke patients and their caregivers described gross motor difficulties due to issues of paralysis down one side of the body, while Parkinson disease patients provided examples of bradykinesia, such as walking at a slower pace, because of the condition. One Parkinson disease participant described bradykinesia as a disconnect between thoughts and body movements: “The brain's saying go, and I couldn't bring my arm down to get my feet, my legs, everything moving all at the same time.”
Sleep was discussed as a problem related to four of the conditions. MS participants cited sleep interferences due to bladder issues and leg sensory complaints such as a “pins and needles sensation.” Stroke patients also attributed sleep problems to leg sensory complaints. Insomnia was discussed by the Parkinson disease patients and the Alzheimer disease caregivers. In addition, several caregivers from the Alzheimer disease group discussed a related sleep issue that was unique to their care recipients: sundowning syndrome.
Social Role Participation
Adult participants from the ALS, MS, epilepsy, and stroke groups, as well as participants from the pediatric epilepsy group, discussed how their social role participation changed as a consequence of their conditions. Subthemes of this domain are described in the sections that follow.
Participation in meaningful activities.
Respondents from all of the groups expressed the importance of socializing and participation in meaningful activities, such as volunteering, dancing, attending support groups, and traveling. Unfortunately, many of these participants could no longer participate in these activities because of their conditions.
Some participants expressed this change in relation to their roles within their social groups. One ALS participant explained that because he could no longer work, his relationship with coworkers, who were once the center of his social circle, was now limited and distant because he did not interact with them on a daily basis. Similarly, participants from the pediatric epilepsy group who were homeschooled explained that they did not have opportunities to interact with their peers. One MS participant described the impact of her condition on her social relationships with friends:
I can't go out to the dances every weekend, I used to go there and I'd sit there and watch everybody dance, but I couldn't dance. I was like, “Why am I doing this?” So, therefore, I cut back on doing all those things I loved to do…. I don't get to see my friends as much because I'm not going to the events, so I feel again that I'm being “robbed,” you know, I'm being robbed of my life.
Conversely, participants from the stroke groups discussed how they continued to participate in meaningful social activities by enrolling in stroke studies to better the lives of other stroke patients, doing volunteer work, and attending church.
Issues related to work.
A number of focus group participants discussed experiencing various issues related to work as a result of their conditions. For example, several participants in the MS, adult epilepsy, stroke, and pediatric epilepsy groups described problems with retaining or finding work. Several of the MS participants described how they were forced to end their employment because of memory or visual problems. In addition, participants from both the adult and pediatric epilepsy groups experienced difficulty with getting to work because some states refuse to issue drivers' licenses to people with epilepsy. Adult epilepsy patients also experienced discrimination when applying for employment.
Participants from various groups expressed the importance and challenges related to interpersonal relationships with family, friends, and romantic partners. For example, participants from the stroke, MS, and pediatric epilepsy groups stated that interacting with friends was important for maintaining HRQL. Given that ALS is a rapidly progressive and invariably fatal disease, participants in this group spoke of the inability to spend time and participate in activities with family members. Issues related to mortality and family relationships emerged as one father explained about his relationship with his son:
We used to play football in the yard. I can't. I would like to teach him how to drive. I don't think I'll be able to do that. That was my quality of life, to spend time with my children, and it's being ripped right away from me.
Participants in the MS and adult epilepsy groups also expressed difficulty with dating and finding romantic partners because of their conditions. One MS patient described how symptoms such as fatigue often limited social activities: “I never had dates because who wants to tell their date they have to go home at 8 o'clock?” Similarly, an adult epilepsy patient described having difficulty with finding a partner who was not frightened by her seizures:
They can just about deal with everything until you get to the grand mal seizures. When you get to that, then it's frightening to them. You can tell them you have a pacemaker, tell them you have diabetes. The minute you say grand mal seizures, now they're afraid of you.
Loss of Skills
Participants in groups representing five of the six adult conditions (Alzheimer disease, ALS, Parkinson disease, MS, and stroke) reported a loss of skills as a result of their conditions. The loss of skills included those related to work, hobbies, driving, physical activity, and managing household finances. For the majority of participants in the Alzheimer disease groups, these skills were often related to cognition, such as memory. The stroke caregivers also expressed difficulties their care recipients had in managing finances and performing work. Several MS participants spoke with sadness when describing their loss of ability to perform certain skills that they previously enjoyed, such as dancing.
For the ALS and Parkinson disease groups, the majority of skill loss was related to fine motor function, which prevented these patients from engaging in hobbies or work requiring dexterity. As one ALS participant explained, “When I retired in 1999, my hobby was photography…. For retirement, I got an expensive camera, two cameras. So that's a downer, you know. You have all this equipment, professional equipment…. I can't even trip the shutter on my Bronica camera.”
Participants across the adult epilepsy, Parkinson disease, and pediatric epilepsy groups stated that they often felt stigmatized because of their conditions. For example, several participants from the adult epilepsy focus group felt stigmatized, particularly when having seizures in public or at work. Patients from the stroke focus group often felt judged for their impaired speech, walking, and use of braces or wheelchairs. One person from the Parkinson disease group also expressed that she felt that the public “associates a mental deficiency [with Parkinson's].” Caregivers of pediatric epilepsy patients stated that their care recipients often felt stigmatized in school, by both students and teachers.
Participants discussed other dimensions of stigma. For example, MS and Parkinson disease participants felt stigmatized due to the transient nature of their symptoms. Thus, there were times when their symptoms were not always obvious, leading others to doubt the existence or the severity of the condition. One participant with MS explained that because she used her motorized cart to prevent fatigue, rather than an inability to walk, “people look at [her] like, ‘What are you doing in that? You're fine.’” A Parkinson disease participant concurred, “When your medicine is ‘on,’ and everything is working, people question if this guy has Parkinson's.”
Participants representing four conditions expressed difficulties with communication‐language. The most common complaint discussed during the Alzheimer disease, ALS, and stroke focus groups was a difficulty forming words. Some Alzheimer patients explained that they were able to form words, but the words often were not what they had expected to say. One participant explained, “What'll come out of my mouth is entirely different than what I was going to say.” A stroke caregiver explained that his wife, “in the middle of a sentence, will have to stop to think what word she needs to use next.” Although participants from the MS group did not share the common experience of difficulty forming words, they did express problems with slurred speech. Additionally, several participants in the MS group and the caregiver stroke group explained that the difficulty with communication‐language was exacerbated by fatigue or stress.
Perceived Cognitive Function
Patients across the MS, Parkinson disease, stroke, and Alzheimer disease conditions deliberated issues related to perceived cognitive function. One‐fifth of the Alzheimer disease discussion was devoted to memory problems, which was expected, considering the devastating effect of Alzheimer disease on cognitive processes. Alzheimer patients and caregivers provided several examples of memory problems, including difficulty remembering directions, where they lived, or family members. Some caregivers described memory problems as inconsistent, while others described memory problems as consistent and from a specific point in time. For example, a caregiver stated that his mother‐in‐law “remembers nothing from a year‐and‐a‐half ago. We have a granddaughter who spends most of her life with us … and she still asks, ‘What's her name?’” Other caregivers explained that their care recipients were unable to remember specific steps for routine behaviors, such as walking or eating.
MS patients also experienced memory problems, which often interfered with work and interpersonal relationships, such as “when you're dating Charles and you call him Bob.” Parkinson disease patients and stroke patients also reported memory difficulties, particularly with short‐term memory. A participant from the stroke focus group explained that his short‐term memory was “on a temporary pause,” and he often had difficulty remembering certain instances, such as part of a conversation.
Participants in the Alzheimer disease patient group also described difficulty performing tasks because of impaired cognitive function. For example, a participant explained that “I sit down, I know exactly what has to be done and how to do it, but I can't pick up the pencil and do it.” MS patients also spoke of these cognitive function issues, stating that their difficulties often interfered with work, forcing some participants to terminate their employment.
Lack of Independence
Participants from Alzheimer disease, ALS, stroke, and pediatric epilepsy groups articulated a sense of lack of independence as a result of their conditions. Patients and caregivers from these groups provided examples of areas in which patients encounter difficulty, such as dressing themselves, maintaining personal hygiene, or eating without assistance. Participants in these groups also discussed a lack of independence after surrendering their drivers' licenses. As a result, one patient from the Alzheimer disease group explained that she missed driving, and that now she had to depend on others to take her to church, doctor appointments, and other activities. A participant in the stroke patient group further explained that the inability to drive was also a difficult issue:
I don't like to have to depend on people to take me places, you know, and that sort of gets me. I don't have the confidence to travel by myself … they take me, but sometimes you feel like you're dependent, and I don't like that feeling.
Positive Psychological Function
Participants across four conditions reported examples of how they have been able to reach a level of positive psychological functioning despite the negative experiences of their conditions. Although not all subthemes of positive psychological function were endorsed by four or more conditions, we have addressed these subthemes in the following sections because participants provided rich data and described these concepts as qualitatively important to the understanding of HRQL.
Mastery and control.
Participants in the MS, Alzheimer disease, and stroke focus groups reported examples of how they have been able to engage in and manage activities in their world. In order for participants to achieve mastery and control over their surroundings and situation, many expressed the need to first understand their symptoms, how their bodies respond to the environment, and how to best adapt to the situation. MS groups contributed to the majority of this discussion, as participants discussed the need to “learn to listen to your body” and to understand the body's strengths and limitations. For example, one participant stated, “I won't eat or drink if I know that I'm going somewhere in the next hour.” Another stated that it was imperative to plan and strategize throughout the day: “Planning becomes a huge part of every element of having MS. If it's dinner with friends, it's like this is my window. If it's working, these are the 2 hours when I really can focus.” Thus, this participant scheduled activities knowing when her body was best able to adapt to the situation.
Caregivers of Alzheimer disease and stroke patients also described various ways in which their care recipients were able to achieve mastery and control of their lives. For example, one caregiver explained that her husband managed his activities with “compensating strategies” that included maintaining intricate lists and “elaborate schemes for how to manage the losses by writing things down.” Several stroke patients also were able to adapt and find ways to manage their lives. One caregiver explained, “My husband's stroke is on his left side, and he was left‐handed, so he had to learn to do everything with his right hand.”
Spirituality was discussed several times across various conditions. For example, several caregivers of stroke patients explained that their care recipients have become more focused on spirituality and a “closer walk with God.” Several participants in the ALS group also spoke of how their sense of spirituality intensified, which also helped them to reframe their view on mortality. As one person stated,
So I looked at it not so much as a death sentence myself, but I was like thank God, I've got time…. Now I got time to get my house in order. My faith, my faith needed restructuring. I was amazed…. I thought just by being a good guy … that there's your ticket in [claps hands to emphasize], [but] no, I find out, no, it's more than that. So I was blessed that I got … more religious, and therefore … I got my house more in order.
Participants from ALS groups and stroke groups also spoke of how their conditions affected their view on life and how they grew to appreciate life as a result of the condition. Participants described an awareness of living in the moment and a mindfulness of the present. This finding may be due to mortality issues related to ALS and the level of acceptance these participants felt with their condition. As one respondent explained,
I walk out in the backyard with my dog every night … and we look up in the sky and see the stars, and that is like fine wine. I mean, I'm blown away by that now. You know, where I've looked up before and been, oh … those are cool and stuff, but now it's like wow.
Cross‐Condition Themes of In. uence
Our analysis indicated that several themes represent HRQL outcomes as well as potential mediators of HRQL. Further research is needed to determine the overlap, influence, or reciprocal relationship between these themes and HRQL outcomes.
Participants across the Alzheimer disease, adult epilepsy, MS, and Parkinson disease focus groups discussed various aspects of general financial difficulties. A number of discussions were devoted to the challenges of paying for medication, changes in lifestyle, and difficulty with paying daily expenses as a result of their conditions. Termination of employment as a result of the condition was also discussed, which led to additional stress and burden for participants. ALS, adult epilepsy, and MS groups also discussed issues related to disability and social security. These participants had trouble filing for disability, either because of complications with paperwork, an extended waiting time to receive their disability checks, or denial of disability benefits without explanation. Others explained that they often faced stigma and prejudice when completing disability forms, particularly if they did not have symptoms that were immediately visible or apparent.
Participants often described a variety of strategies that they used to improve their HRQL by managing their symptoms to alleviate stressors and to achieve a higher state of positive psychological functioning. For example, respondents from all groups displayed a positive attitude. As one participant in the ALS focus group explained to others in the group, “Hopefully, you can stay positive. I think that's been one of the things that's been an attribute for me to be able to handle it, just a positive attitude every day.”
Participants also used humor as a means to alleviate the stress of their conditions, as was discussed in the Alzheimer disease, ALS, adult epilepsy, and MS focus groups. A participant in the MS group explained, “If I'm going up the stairs and I stumble, and there's somebody behind me, I'll turn around behind me and say, ‘Hey, what's the pushin’ for?’… [to] put the humor back on this.” Several patients also expressed that they were able to make themselves feel better by making other people laugh. Other topics pertaining to coping strategies included using spirituality as a means to reach positive psychological functioning, having a sense of purpose, engaging in creative and physical activities, and adhering to a routine schedule.
A third factor that appeared to influence patients' HRQL related to various types of social support. A discussion of the positive support from family members and friends was apparent in all focus groups. All groups stated that their families provided various means of support, and that most were comfortable with asking and receiving help from their families. One participant in the ALS group stated, “And you know you can impose on family. You don't like to impose on someone who's not family.” Many did not realize that they had this level of support until after the onset of their condition, which often brought families closer together. As one woman in the Alzheimer disease group explained,
My oldest daughter and I have sort of traded places. I was a businesswoman all my life, but I lost it, the ability to do a lot of things, and so I couldn't even balance my checkbook anymore; and then I handed it over to my daughter, and she's done a beautiful job…. I don't know what I'd do without her.
Participants reported that the support they received from their family and friends brought enjoyment and stability and mitigated many of the stressors that they experienced as a result of their conditions. This has also been expressed in the literature, as these conditions may preclude patients from participating in social activities, leading to decreased interaction with friends and ultimately to social isolation (George & Clipp, 2000).
Themes Speci. c to Condition Subsets
The following themes were found to be related to small subsets of conditions, with a few being unique to specific conditions.
Participants from the Alzheimer disease, stroke, and Parkinson disease groups discussed chronic changes in mental states, behaviors, or personality as a result of the condition or treatment effects, especially related to HRQL. Examples that emerged from the focus groups include depression, anger, withdrawal from social interactions, less patience, and a short temper.
Participants having Parkinson disease, MS, and pediatric epilepsy described how their treatments have an effect on their HRQL. Whereas pediatric epilepsy and Parkinson disease participants discussed changes in mood being a significant effect of treatment, MS participants mainly described treatment effects related to their physical condition. For example, some MS participants stated that their medication increased their energy to the point that they left “skid marks on the ceiling,” while others experienced fatigue or even panic attacks. Other treatment effects discussed in the groups included weight gain, stomach irritability, changes in energy level, lack of taste, depression, and flu‐like symptoms.
Participants in both the MS and ALS groups discussed their feelings related to the uncertain and unpredictable nature of their conditions and “not knowing how your disease is going to progress.” Some MS participants described how “fatigue kind of impacts [their] schedule.” As one explained, “You never know whether you're going to get the stuff done you want to get done.” Similarly, an ALS participant stated,
You wake up and it's, well, what's going to go today? [When I] started out, I was getting fasciculations in my leg, and that was it. Then pretty soon I was limping. Then I had a drop foot. And then I got it in this leg, and then I got it in this arm.
This theme was exclusive to ALS, which was to be expected, given the nature of this disease. Participants spoke in a factual manner about living wills and decisions about distributing their possessions and preparing for the future. As one person stated, “I know that's going to come down the road, but you know, no matter what you make for it, it's coming. It's a train.”
The theme of self‐acceptance was particular to MS patients and addressed a unique aspect of positive psychological functioning. For these participants, reaching a level of selfacceptance and coming to terms with the condition was imperative in order to adjust and discover ways to control their condition. Many participants stated that self‐acceptance was one of the hardest and most important lessons learned from MS.
Themes specific to Alzheimer disease.
There were several themes found to be exclusive to Alzheimer disease, including fear, loss of initiative, and living in a state of distorted reality. Examples of this distorted reality varied, including this description provided by one caregiver:
I cannot watch news programs anymore … because halfway through … [my mother will] be there crying … what's happening on the television is real, right around her, taking place right there. A bomb blowing off in Israel, or somewhere in the Middle East, that's happened outside. That happened right around and in her home, and she's in fear.
Participants spoke of other fears, including a fear of being lost, particularly when leaving the home and driving. They also discussed a loss of motivation or initiative, considered a well‐established feature of this condition (Alzheimer's Association, 2006; Tractenberg, Weiner, Patterson, Gamst, & Thal, 2002).
Findings from the Neuro‐QOL focus groups demonstrate the complexity of HRQL issues across and within major neurological conditions. The results showed that although the HRQL impact of neurological disorders and their treatments was discussed across all conditions, the relative impact on the different HRQL domains varied by neurological condition. All patients described their HRQL in terms of physical, emotional, cognitive, communication, and social dimensions of life. Findings, however, revealed that the salience of certain HRQL domains is greater for some neurological conditions in comparison to others. For example, results suggest that the impact of treatment effects on HRQL is greater for patients with Parkinson disease, MS, and pediatric epilepsy than for patients with other conditions. This finding may represent the number of moderately effective treatments for symptom management of these neurological conditions. Similarly, discussions of mortality were exclusive to patients with ALS, a progressive neurodegenerative and ultimately fatal disease where treatment is largely palliative. These findings suggest that although HRQL is multidimensional and comparable across neurological disorders, the contribution of specific domains to overall HRQL may differ among disorders.
Participants described achieving positive experiences, such as valuing heartening moments with family and reaffirming spiritual values, by enduring the challenging aspects of the illness.
Results also showed that descriptions of positive experiences were often interwoven with distressing accounts of illness. Distress was often associated with physical limitations, loss of skills, social isolation, stigma, and discrimination. Interestingly, participants described achieving positive experiences, such as valuing heartening moments with family and reaffirming spiritual values, by enduring the challenging aspects of the illness. This simultaneous experience of positive and stressful domains of life experience has been reported in other qualitative research with cancer patients (Breaden, 1997) and adds more evidence for a complex coexistence of states (Reynolds & Prior, 2003). In addition, many of the themes that resulted from focus group discussions pointed to the patients' use of strategies that influence the experience of HRQL domains. Positive psychological functioning and related subthemes were discussed as a domain, as well as a mediating coping mechanism, often as a way to help patients reach self‐acceptance with their diagnosis and redefine their identity. On the one hand, several participants discussed the use of humor, creative activities, and positive attitude to manage their symptoms, conditions, and life experiences. On the other hand, financial hardship was another theme that was discussed not as a domain, per se, but as a mediating factor associated with emotional stress.
Although the focus groups were one of several sources of information used to inform the development of the Neuro‐QOL tool, the study was limited to a finite number of neurological conditions. To minimize this limitation, the research team used purposive sampling methodology to select conditions that had a range of symptomatology and affected a range of ages. Another limitation concerned the exclusion of patients with severe physical or cognitive impairments from this study. Unfortunately, this is a limitation of many neurological studies and requires further exploration. To address this, caregivers of patients with severe impairments were recruited into the focus groups to learn about their care recipients' HRQL. In addition, the small number of focus groups does not support the generalizability of the findings to all neurological conditions, but they do support the end goals of the study as a whole. As such, these findings will provide a rich context against which potential questions, scales, and item banks can be assessed for relevancy and demonstrate the ability to use limited resources in generating generic and specific domains across multiple conditions.
The results of these focus groups highlight certain future implications, such as the need for further exploration of the factors that mediate HRQL domains. For example, although the cost of illness in relation to HRQL has been explored for a few neurological conditions, such as MS (De Judicibus & McCabe, 2005) and ALS (Nelson, Trail, Van, Appel, & Lai, 2003), similar research with other neurological conditions is lacking (Whetten‐Goldstein, Cutson, Zhu, & Schenkman, 2000). In addition, there is little to no research documenting the process whereby neurological patients maximize their HRQL through coping strategies and social support. The knowledge gained from a better understanding of factors that mediate HRQL can be incorporated into theoretical models that form the foundation for interventions or policies directed toward the improvement of HRQL.
The findings also highlight the value of patient input in the development of patient‐reported outcome measures. Historically, HRQL instruments circumscribe the life experiences of patients into stereotyped responses that have little or partial meaning for the intended respondents. This study is among the first to use qualitative methodology to explore HRQL among a wide cross section of neurological conditions. Qualitative methods permit an in‐depth exploration often not possible with quantitative methods, which may separate behavior from its social and environmental context. The use of focus groups to elicit patients' words and experiences provides an indepth examination of domains, allowing patients to define HRQL for themselves and to present personal and meaningful insight. For example, the findings indicate that positive psychological functioning is an important HRQL domain for patients, although this domain is often ignored in other HRQL tools. Findings from this study also suggest that there is a need for a flexible HRQL tool that can measure general as well as condition‐specific HRQL dimensions, and that particular dimensions may have a greater impact with certain conditions. In addition, focus group participants emphasized the interrelated, fluid nature of HRQL domains, which also must be captured in HRQL tools.
The multiple focus group approach allowed for an examination of HRQL issues that were relevant for myriad neurological conditions as well as specific to the experiences of particular neurological patient populations. The findings support the need for the creation of a HRQL measurement tool that is flexible, multidimensional, and generic as well as specific. The focus group data will inform the development of the Neuro‐QOL tool, which seeks to incorporate subjective patient‐reported outcomes of functioning, such as social, physical, and mental well‐being. Ultimately, the research team envisions the end product as a clinically relevant and psychometrically robust patient‐reported outcomemeasurement tool that is responsive to the needs of researchers and patients in a variety of neurological disorders and settings, with a particular emphasis on measuring outcomes in clinical trials.
This study was supported by a grant from the National Institute of Neurological Disorders and Stroke (NINDS; Contract No. HHSN265200423601C) to create the Neuro‐QOL tool discussed in this article.