Disease Management and Measurement
The impact, management, and research needed on specific symptoms of multiple sclerosis (MS) are discussed in the previous chapter. This chapter discusses the conditions of life with MS from a more general perspective and emphasizes research that can improve the lives of people with MS. It covers psychosocial and physical adaptations, as well as a variety of health care issues. At the moment of being diagnosed, the patient is forever transformed into a “person living with MS.” Even in the absence of signs or symptoms this person will forever after live with the knowledge that he or she can be unpredictably impaired. Sometimes the person will recover, sometimes not. For most, living with MS will become one of the major challenges of life. People with MS need to solve problems ranging from finding the right button hook, to getting dressed each morning with limited use of their hands, to specifying “reasonable accommodations” for continuing to work.
Improving the lives of people with MS rests on better understanding of their needs and their successes, specifically research into the conditions of life with MS, which requires objective, reliable research tools. The most essential tools for measuring the conditions of life with MS are the various survey instruments that measure abilities to function and quality of life, which are discussed in the latter half of this chapter. These tools not only provide for objective assessment of the needs of people with MS, they are also an essential element of measuring the effectiveness of any sort of therapeutic intervention—be it a rehabilitation process, a self-help program, or a disease-modifying therapy. Quality-of-life measures can also reveal aspects of the disease process that are not readily captured in standard clinical measures and might reveal insights into the underlying disease
mechanisms. For example, pain, fatigue, and memory impairment are often among the earliest manifestations of the disease, yet they were virtually unrecognized as symptoms for many years. These symptoms are prevalent, troubling, and yet correlate only weakly, if at all, with neuropathology as revealed by current neuroimaging techniques. Quality-of-life measures might also provide more sensitive outcome measures of clinical efficacy of new therapies, and importantly, they measure the outcomes that concern patients the most.
Given the millions of people who will be living with MS now and in the future, it is important that the focus on curing MS not come at the expense of efforts to address the disruptions that pervade routine daily activities, personal relationships, family life, work responsibilities, and social involvement.214
LIVING WITH MS
If a cure were found, would I take it? In a minute. I may be a cripple, but I'm only occasionally a loony and never a saint. Anyway, in my brand of theology, God doesn't give bonus points for a limp. I'd take a cure; I just don't need one. A friend who also has MS startled me once by asking, “Do you ever say to yourself, ‘Why me, Lord?' ” “No, Michael, I don't,” I told him, because whenever I try, the only response I can think of is, “Why not?”
Many people with MS cope remarkably well, others less so. Understanding the traits and conditions that enable people to cope so well can provide insights necessary to help others, but obtaining these insights is complex. Conducting an investigation about people with MS similar in depth and scope to the multisite Medical Outcomes Study, a series of multisite studies funded by the Robert Wood Johnson Foundation in the early 1990s,175 would be invaluable, but also methodologically and logistically challenging and enormously expensive.
A growing body of research describes the lives of average people living in the community with MS. Many of these studies involve surveys, the application of various psychometric instruments (functional status measures, quality-of-life indices, and other instruments targeting specific topics), and qualitative methods (reviews of in-depth interviews or focus groups). With the exception of the Expanded Disability Status Scale (EDSS; see Appendix D), there is little consistency in the survey forms or instruments used across studies. In addition, despite the experiential insight gained from well-conducted qualitative studies,41 they rely on very small samples from specific subpopulations, and the results cannot be assumed to apply to the full range of people with MS. Some studies have involved large numbers, but they tend to focus on single, well-defined issues (for example, employment). Perhaps most importantly, most studies treat people with MS as a homogenous group, as opposed to groups of people with markedly different disease states.51
“You Have MS”
Numerous studies have conclusively documented that communication between health professionals and patients is often problematic.106,107 For people with lifelong conditions such as MS, initial contacts with health care professionals are especially important and can shape patients' attitudes about their disease and expectations about the role of clinicians. Sometimes problems result from general insensitivity, probably not specific to MS per se.
People with MS who write about their experiences invariably highlight the moment when they were first given their diagnosis. It is a pivotal, life-altering moment that remains vividly etched in their memories. Very often, these memories are filled with anger at the way the diagnosis was delivered. Sally Ann Jones, in her mid-fifties with MS, described how her doctor told her the diagnosis many years previously:84
The doctor spent about a minute and a half with me and then he said, “The bad news is, Mrs. Jones, you have MS. The good news is, when I saw you before, I wrote down three potential diagnoses in my notes. If you'd had either of the other two diagnoses, you would be dead by now.” Back then, he never mentioned that to me. I said, “Why didn't you tell me?” He said, “The symptoms of the other diagnoses would have been so bad, you would've had to return, and I didn't want to upset you unnecessarily.” And with that, he left. He didn't tell me what to do. He didn't say, “Do X.” He didn't say, “Come back in six weeks.” He just left. Period. He spent about 10 minutes, beginning to end. I was absolutely in shock.
Because the symptoms are so often insidious, vague, and nonspecific, there can be a long period during which the patient knows something is seriously wrong but is unable to receive definitive answers. The uncertainty is highly stressful, and patients later often resent the long period of concerned waiting or the attitude of a physician who felt it was kinder to offer vague explanations when she or he was strongly suspicious the problem was MS.130 In 1995, Murray wrote, “I have never heard a patient express gratitude for being kept ignorant of his or her diagnosis, but I have heard many express anger and disappointment for being kept in the dark.... While patients are in the dark about their diagnosis, they are unwell without legitimacy, expected to function normally without the physician-sanctioned ‘sick role,' and often under the suspicion of family, friends, employers, and even themselves, of being hypochondriac or malingerers.”139
One study identified four major themes of the experience of being diagnosed with MS: whispered beginnings (earliest signs and symptoms, understood only in retrospect); echoes of silence (worrying, wondering, and waiting, reinforced by relapsing and remitting patterns); hearing the words spoken and beginning to tell others; and refocusing their lives.100 During the period of whispered beginnings, people often tried to minimize what they were experiencing, attributing it to stress. During the echoes-of-silence period, patients frequently felt anxious,
lonely, and worried, sometimes trying various remedies (for example, multivitamins, stopping smoking, drinking lots of water, exercising). Finally, the actual moment of being told, “You have MS,” was readily recalled by the participants. They described trauma-filled reactions such as “being in shock, feeling numb, and really stunned” when they heard the news. For some, the crisis of this response lasted for moments; for others, it lasted days to weeks. The experience of being told the diagnosis of MS brought the study participants to a time of being “emotionally wounded.”100
These experiences emphasize not only the value of better diagnostic tools discussed in Chapter 2, but also the need for a better understanding of communicating with patients in the face of uncertainty at the initial stages of diagnosis and—once the diagnosis is confirmed—in the face of delivering devastating information.
Yet the issues surrounding diagnosis are changing. Patients today spend less time waiting for a diagnosis than they used to, largely due to improved magnetic resonance imaging (MRI) technology and its earlier, more frequent use. Once they receive their MRI results, however, they are faced with uncertainties and decisions that previous patients were not. One is the problem of interpreting abnormal MRI scans. Although the scans might confirm a diagnosis of MS, they still provide little information about the prognosis for impairment or disability. Another is the decision whether to start any of the disease-modifying therapies. Patients must weigh the immediate disadvantages of being yoked to a regular medication schedule with possible side effects that diminish their quality-of-life against the potentially great, but nonetheless uncertain, future benefit. In essence, it is impossible for a patient to make a fully informed decision. Patients can only guess, but the burden of decision rests with them. Therapies appear to be most effective if they are begun early in the disease—at a time when many patients might be most reluctant to assume the burden of treatment.
Disease-modifying therapies for MS were first approved in 1993 but have been widely used only in the last few years. In sum, much of the literature on the psychological aspects of living with MS, almost all of which predates the wide use of disease-modifying therapies, must be reconsidered in light of current options, which are themselves in flux. This does not, however, alter the fact that people with MS must adapt and readapt throughout their lives to MS.
MS generally starts just as people are beginning their independent lives, families, and careers. Many will live with the disease across the full spectrum of their adult lives. Repeatedly over time, they adjust, with varying success, to its myriad disabling consequences. MS is a disease that teases with its unpredictably waxing and waning course—debilitating people one month, restoring function the next. Sometimes losses are insidious, gradually mounting and only dimly
perceived until people finally recognize their presence and permanence. For some, living with MS can be like balancing, precariously, on a razor's edge. Others do well, experiencing only fleeting symptoms over many years. S. Kay Toombs, a philosophy professor who has had MS with progressive impairment over 25 years, observed:214
When the body refuses my commands, it suddenly appears to have a will of its own. Rather than being that which enables me to carry out my projects in the world, my body is an obstacle that I must overcome. I may, for example, have to manually pick up my leg if I am to negotiate a small step, or pay exquisite attention to the way my body feels in terms of pain, fatigue, stiffness, if I am to engage in ordinary activities.
Bodily attentiveness becomes an integral aspect of the illness experience. With permanent physical impairment, one must daily compensate for the body's disabilities and explicitly allow for its limitations. Even when there are lengthy periods of remission, the chronically ill remain uneasily attuned to the way the body feels and moves—always “on guard” for signs of an impending recurrence.
Research suggests that the experiences and feelings of people with MS are often more positive than clinicians or others expect. One study administered six instruments (Areas of Daily Functioning Questionnaire; Ways of Coping Checklist; Global Coping Scale; Thoughts About Suicide Probe; Anger-Fear-Depression Questionnaire; and Global Self-Esteem Scale) to 125 people with MS.48 Although almost 60 percent reported having felt they needed professional psychological help at some point since their MS diagnosis and about 30 percent reported that they were not coping well and currently needed professional help, the most striking finding was that, despite the obvious challenges faced by people with MS, most successfully adapt to the disease. This finding was replicated across all the measures included in the study, including self-rated quality of marital, family, and friend relationships; objective measures of emotional functioning, thoughts of suicide, and self-esteem; and direct self-assessments of current coping with the disease.48 Another study involving 629 women with MS found that compared to normative controls, they displayed significantly better interpersonal relationships and stress management, although they had significantly lower physical activity and spiritual growth compared to normative controls.199 A study of 64 MS patients in Finland found that although they reported feeling physically limited and insecure, many also noted that having the disease helped clarify personal values and enhanced personal growth.115
People vary widely in their coping styles and in their perceptions of how much control they have over what happens to them.160 Coping is defined by Lazarus and Folkman as “constantly changing cognitive and behavioral efforts to manage specific external and/or internal demands that are appraised as taxing or exceeding the resources of the person.”114 Styles of coping with stress are often
dichotomized as “problem focused” or “emotion focused.” Problem-focused behaviors entail confronting the problem, seeking information about how to manage it, and devising strategies to deal with it. Emotion-focused strategies generally involve denial and escape or avoidance, or reconfiguring the problem to make it more positive. While most people use both strategies, one style usually predominates. Studies in other diseases suggest that patients who use problem-focused techniques make better adjustments and have better outcomes than those who use avoidance and denial.11,37,99,172,173,203,204
Remarkably few investigators have inquired into the mechanisms that underlie success and failure in adapting to MS.2 One prospective study that followed the adaptations and coping strategies of 27 patients found that escape avoidance coping strategies predicted future depression, but this was not a robust finding because the investigators reported that the correlation was not apparent in a follow-up study of the same patients.2 Another study of 433 people with MS did not find a significant association between coping strategies and emotional well-being.231 The same study noted that study subjects' coping styles depended on their level of uncertainty about their symptoms, treatment, relationships with caregivers, or future plans. Subjects used more emotion-focused coping when they were more uncertain and problem-focused coping when they were less so.
Psychological adjustment to MS is complex and multiply determined. Eklund and MacDonald48 found that people with MS reporting better long-term adjustment tended to use “adaptive denial” as their coping mechanism at the time of diagnosis. By comparison, those reporting “confrontational coping” (obsession) and escape or avoidance (complete denial) adjusted relatively poorly. In addition, those who had thought through answers to the question, Why me?, were less likely to be depressed in the future. A study of 43 people who had MS for four years or longer (15 years on average) found no relationship between the extent of physical impairment and coping style.12 Murray139 suggests that MS patients use both problem- and emotion-focused coping styles, but the three most common strategies are problem focused: trying to maintain some sense of control; trying different problem-solving approaches; and looking objectively at problems from different angles. Coping strategies can change over time, with emotion-focused methods predominating when patients sense things are beyond their control.139 One limitation of most studies on coping is that most study participants are identified well after the onset of MS so any consequences of adaptational style would have been in operation for a number of years.42
The chronic and uncertain nature of MS makes tremendous demands on the coping skills of people with MS and those who care for them. People adjust with varying degrees of success, relying on different coping strategies, often switching from one to another as their circumstances change. A better understanding of which of those strategies (and under which circumstances) foster more successful adaptations to life with MS would enable nurses, social workers, psychologists, physical therapists—indeed, anyone who counsels people affected by MS—to
provide greater assistance in helping people manage their disease. Likewise, people with MS would directly benefit from information about what coping strategies will most enhance their personal sense of well-being.
Change in Attitudes over Time
Finally, given the chronic nature of MS, patients' attitudes toward the disease—and even themselves—can vary widely and change over time. Toombs wrote:
Bodily change may have different meanings for the same individual at different points in time. The inability to walk has a different significance for me now than it did when I first contemplated this loss of possibility as a thirty-year-old woman who was used to “standing on my own two feet.” And the meaning of this change is different for me than it is for my friend who was a marathon runner before being diagnosed with MS.214
People of all ages with MS experience emotional distress and diminished sense of well-being when illness disrupts their life-styles, activities, and interests. However, older people with MS appear to be less adversely affected than their younger counterparts.43 It is not known why this is so, but it could result from age differences in psychological disengagement, expectations about the age appropriateness of chronic disabling illness, or some other psychological processes. Little research has examined changes in people's attitudes over time or the factors associated with such shifts. These changes could alter perceptions of the value of medical therapy, the role of health care professionals, and interventions to address functional impairments.
Duration of disease does not appear to be associated with psychosocial adjustment: “longer duration is not manifested in greater psychological distress, worry, body image, depression, or guilt.”51,110 One interpretation is that the longer people have MS, the better they are at accepting the physical consequences and the less likely it becomes that MS will affect their emotional well-being.
Impact of Self-Perceptions and Attitudes
Patients, regardless of their illness, also vary in their perceptions of how much control they have over their lives. People with a strong internal “locus of control” strive to exert influence over what happens to them. They believe they are responsible for what happens to them by their own efforts to control the situation. In contrast, those with an external “locus of control” tend to hold outside institutions, other people, or “fate” responsible for what happens to them. A study of 94 people with MS found that increasing sense of personal control was associated with better psychosocial well-being, and two-thirds of participants reported positive psychosocial adjustment.42 An investigation involving 100
people with MS found that those with an internal locus of control were still largely ambulatory and self-sufficient after 25 years compared to externally focused patients.226 These studies could not directly investigate the reason for those differences.
Self-perceptions and attitudes about their lives have important consequences. Toombs notes, “Negative societal attitudes towards illness and disability not only diminish self-esteem, making it hard to accommodate loss of bodily function, but such attitudes cause concrete hardships in terms of the disruption of personal relationships, loss of employment opportunities, inability to obtain health insurance, and so forth.”214 These self-perceptions might vary according to the cause of disability. In a study of 25 wheelchair users, Avillion8 found much lower self-esteem among people disabled by MS than by spinal cord injury. Eklund and MacDonald,48 however, found that self-esteem reported by their 125 respondents with MS did not differ significantly from that observed in samples of physically healthy people. Other studies also offer contradictory findings about self-esteem of people with MS.139
Living with MS is indisputably stressful, but it does not follow from this that stress triggers exacerbations or hastens disease progression. This issue has been debated for more than a century with strong held views on both sides.139,202 For many people, the link seems obvious. Psychological stress is known to alter immune response, but it should also be kept in mind that these changes are generally small and within normal ranges.77 In June 1999, the American Academy of Neurology (AAN), through its Therapeutics and Technology Assessment Subcommittee, conducted a comprehensive review of studies on the impact of psychological stress on MS done up to that date.66 The AAN committee concluded that research done up to the time of its review had not clearly established that psychological stress can trigger MS onset or exacerbation, but that this is possible. Most published studies attempting to demonstrate a causal link were methodologically flawed (reviewed in 1999 by Schwartz et al.176). Since the AAN review, a well-designed prospective study has been published indicating that patients who experience one or more stressful events have a small-to-moderate increased risk of disease progression.176 However, the risk of stress following physical deterioration is even greater. In other words, patients with a faster rate of deterioration will also have a higher risk of stressful life events and, consequently, will be more likely to experience stress before an exacerbation. The link between stress and disease progression remains frustratingly unclear.
Many patients believe that stress will worsen their disease and blame themselves for exacerbations (or feel that others blame them) when they have not managed to eradicate stress from their lives. People with MS are frequently cautioned to “take it easy” and not “overdo it,” sometimes leading to decreases in
recreational and physical activity that are out of proportion to this level of impairment.200 This inactivity can contribute to worsening functional capabilities, reducing endurance, and disturbing sleep patterns, perhaps also heightening stress. Indeed, Schwartz and her colleagues have suggested that limiting one's activities might actually leave people more vulnerable to stress.176 The effect of this “vicious circle” on quality-of-life and disease progression has received little study. Even independently of a causal link between stress and disease progression, stress management would seem to be an obvious area in which interventions could improve quality-of-life for people with MS, as well as their caregivers.
Family and Social Relationships
As for most people, relationships with family and friends are central to the lives of people with MS. Despite common assumptions, neurological impairment and physical handicap are not the major determining factors of quality of life for MS patients. Patients are affected primarily by their relationships to other people and secondarily by symptoms of fatigue, pain, and cognitive dysfunction.139 Quality-of-life studies in MS patients suggest that there are seven primary factors affecting mental health and quality of life: loneliness-companionship, fatigue, degree of chronic pain, duration of symptoms, stressful life events, self-perceived cognitive deficits, and clinically assessed cognitive deficits.
Evidence about the effect of MS on relationships is somewhat contradictory. Conventional wisdom holds that the stresses of MS end marriages, especially those that are already tenuous, and that divorce rates are double those in the general population.139 For marital relationships, three factors are particularly important: (1) both partners cannot equally “share” the illness experience, despite good intentions; (2) traditional gender roles may be compromised; and (3) having one's partner serve as caretaker raises a complex set of emotions on both sides.115 In one study of 125 people with MS, slightly more than half reported that relationships with their spouses and other family members had changed as a result of the diagnosis (55 and 53 percent, respectively), and somewhat less so with close friends (39 percent).48 However, respondents said that these changes were positive about half of the time. A study involving 629 women with MS found significantly better interpersonal relationships than for normative controls.199 A study by Wassem226 of 100 people with MS found that their divorce rate was less than one-third that of the general population. Several explanations are possible. Patients reported that the problems caused by having MS had brought them closer to their spouses and that they had more time to spend together than when both worked. Women reported less strain in their roles as mother and spouse when MS forced them to stay home rather than work full-time.226 A study of 101 people with MS found they generally had small social networks of about four family members and two friends. Despite this, they perceived “moderate” to “quite a bit”
of love, respect, or admiration from these people. Isolation grew with duration of disease.145
Effect of Spousal Attitudes. At the time of writing this report, only one recent publication has reported on the role of spousal attitudes in the psychosocial and physical functioning of the person with MS. Schwartz and Kraft178 examined 44 people with MS and found, not surprisingly, that patients who saw their spouses as responding negatively to their impairment had poorer general mental health functioning and more symptoms of depression. In contrast, patients who viewed their spouses as encouraging of well behaviors were significantly less depressed. The authors of this study suggested that their findings be used to develop family-based treatment strategies but did not test any interventions. It should also be noted that cross-sectional studies such as this cannot establish causality, for instance, whether the spousal attitudes were a cause or a consequence of the mental health of their partners with MS.
Caregiving. When a partner, family member, or friend becomes the primary caregiver, complex issues arise.139 While the person with MS is the one who needs the most help, the caregiver is inevitably affected, but the needs of the caregiver are often neglected by the health care system. Little information is available about the needs and preferences of caregivers who often must adapt to more restricted roles and heightened limitations on time, privacy, social activities, employment, and finances.139
One study that examined the experiences of nine caregiver wives of husbands with MS described how love and commitment for their husbands often coexist with anger toward the disease.40 As one woman said, “I still love [husband's name] in spite of everything. And it's really hard to love someone so much and watch him deteriorate. I love him anyway....I don't hate him, I hate the disease.” Yet the demands of caregiving easily become overwhelming.40 The wives sometimes admitted they had reached a breaking point. One observed, “There were times when I felt like throwing in the towel.” The women sometimes felt the inequity of their marital relationships and the pressing need for “space.” One woman said, “I just felt like...if I couldn't get away from the situation for just a little bit....I felt like I was about to go stand on a corner and scream. It's not because I don't love him, or not because I resent what I am doing, it's just that I need some space.” Another study of 146 people with MS and their caregiving spouses or partners revealed significant differences in coping styles, depending on the caregiver's gender and health, and the level of dependency of their partner with MS.72
More knowledge about the coping strategies and needs of caregivers can be used to provide information and counseling to help families cope more effectively with the demands of MS. There has been little research in this area or on ways to relieve caregiver burden. Yet relieving caregiver burden might, in fact,
offer a strategy to reduce overall health care costs by supporting various interventions that might delay the time when an MS patient needs full-time institutional care or even forgo this need altogether. This might be achievable by relieving the burden of caregivers in a variety of ways such as by providing respite care as temporary relief for caregivers, household help, by providing counseling and social support to the caregivers, or by providing in-home health care visits. Identifying cost-effective measures to relieve caregiver burden would be of mutual benefit to caregivers, MS patients, and health care providers.
Relationships with Children. Parents with MS are especially concerned about the effect of their illness on their young children. Some research has described children of parents with chronic illnesses as constrained, repressed, and antisocial, whereas other studies have noted they are more empathetic and mature in their friendships. More research is needed about the children of parents with MS.115
Crist35 videotaped interactions between 31 mothers with MS and their 8- to 12-year old daughters and compared them to those of 34 mothers without disabilities and their daughters. She found similar rates of receptive, directive, and dissuasive behaviors for mothers with MS and their daughters compared to the control group pairs. Another study examined 35 mothers with MS and their young children, paying particular attention to the effects of exacerbations of the disease.38 When the mothers' symptoms were stable, both mothers and children described similar levels of physical affection. During exacerbations, however, their perceptions diverged. Mothers underestimated the impact of the exacerbations compared to the child's perception. One mother noted:
Well, there's a lot of times, because my patience [is] short and...I'm more concentrating on myself, I'm self-centered at that time and they come in...my youngest daughter is very affectionate...when I am having an exacerbation and she wants to hug and she wants to kiss, I try to respond to her because I know she needs this. This is important to her and it's part of her make-up. There's times when I have to say to her, “Mommy's really not feeling well.”38
Sometimes fathers or other family members take over family responsibilities from the mother, leaving mothers saddened and feeling less involved with their children than they would like.38 Older children often overcome fears with knowledge of their own competence and increasing independence. As one older child said, “She deals with it pretty well and knows her limits. It doesn't really bother me because I don't rely on her for every need. She just needs to take care of herself and I will be fine.”38 Another child described her mother's frustration during exacerbations: “She complains all of the time and we have to get this and get that for her. Then she cries and gets mad because she can't do it. It's really hard to take when you just want a normal life like everybody else.” Little research has examined strategies for working with parents who have MS and their children to handle fears and expectations.
Navigating Community and Societal Attitudes
One of the hardest things in living with MS is dealing with how other people react to your condition—this includes family, friends, caregivers and people in general.
Anonymous survey respondent quoted by Clayton et al.28
The social stigma associated with disabling conditions is well recognized.27,65,148,180,236 Charlton27 argues that there is more discrimination worldwide toward people with mental health and hearing disabilities than those with physical impairments. In the United States, laws passed since the 1970s have attempted to redress systemic inequities against people with disabilities. The 1990 Americans with Disabilities Act followed such federal laws as Section 504 of the Rehabilitation Act of 1973, which made it illegal for organizations that received federal funding to discriminate against anyone “solely by reason of...handicap” and the Technology-Related Assistance for Individuals with Disabilities Act of 1988 (P.L. 100-407, the “Tech Act”), reauthorized in 1998, which aims to heighten access to products for improving function and independence.
The extent to which people with MS have litigated under these various provisions or faced overt discrimination is unclear. Despite federal and state laws, environmental and architectural barriers remain widespread.157 Some communities are undoubtedly more accessible than others. Murray139 believes that people with MS are more likely to have fulfilling lives when they reside in urban areas or near friends, theaters, museums, shopping areas, and community agencies, than when they live in isolated regions. The experience of rural residents with MS has been little studied.
People with MS, like others with disabilities, face negative societal attitudes that legislation cannot eradicate. Murray139 observed, “The stigma of a disabling disease becomes a disturbing negative influence on MS patients as they become recognizable in the community. They are very conscious of their ataxia, aware that many will think them intoxicated; ashamed of their canes and wheelchairs, when a short time before they felt themselves healthy members of society.” People with MS themselves have articulated these sentiments:
When you kind of walk unbalanced and people think you're drunk and you start to stagger or you start to slur your words because the nerve ending in your mouth can't pronounce the right words, people look at you and cannot understand why you are acting that way. Some people also say that you are using the MS as an excuse for being tired, for getting out of things.147
When strangers observe that I am in a wheelchair, they make immediate judgments about me as a person. Most assume that I am dependent on others and unable to engage in professional activities. In perceiving that my legs no longer work, strangers also conclude that my intellect has been likewise affected.214
How people with MS respond to these societal attitudes and whether their responses reflect life-style choices and participation in the community is unclear. Mairs is a writer who leads an active life and travels widely, including overseas. Toombs is a university professor who also travels and publishes scholarly works. Both women are “privileged,” accomplished women with recognized achievements and reasonable financial resources. How others with MS, especially those with less education and lower incomes, face societal perceptions and potential discrimination is unknown.
Critique of Research on Living with MS
An overarching view of the state of research in this area suggests the following:
Studies are generally small and do not distinguish among different MS disease states, with authors noting that their results cannot be applied to the full spectrum of people with MS.
Investigators come from various clinical and social science disciplines and tend to publish in journals specific to their disciplines, which are likely to be read primarily by people in those specific disciplines (such as neurology, physical medicine and rehabilitation, nursing, nursing rehabilitation, psychology, occupational therapy, sociology).
Frequently, however, more than one study has targeted a particular topic, so results can be compared and preliminary opinions formed about the generalizability of conclusions. Yet it is unclear whether investigators are aware of or draw from studies performed by those in other fields because they often fail to cite each others' work.
Most studies on coping and adaptation among people with MS rely on instruments developed and validated only among nondisabled populations.
In summary, although many studies address pertinent topics, the research community tends to operate within narrow disciplinary “silos,” and relatively little cross-fertilization seems to have occurred. Given the breadth of the issues involved and the diversity of research techniques appropriate to exploring them, this situation is understandable. Nevertheless, these disciplinary boundaries obscure just how much is, in fact, known about the lives of people with MS. Multidisciplinary work is relatively rare.
Although many issues about living with MS have been addressed, important gaps remain. As noted below, some topics have received little attention. Obviously, the major area that has yet to be studied involves strategies for improving the lives of people with MS: although articles identify problems, they only speculate about how to address them. Trials of proposed interventions to ameliorate
psychosocial and quality-of-life problems for people with MS and their families are rare.
Research strategies aimed at improving the ability of people with MS to adapt and function should be developed in partnership with research practitioners, managers, and beneficiaries; toward this end, the National Multiple Sclerosis Society (the MS Society) should convene a series of forums to identify the most pressing needs experienced by people with MS. The goal of such a series would be to define research needed to identify ways to help people with MS adapt to their illness and enhance their ability to function. The committee did not include the expertise to develop a research agenda to meet the needs experienced by patients. The committee recommends that the MS Society work in partnership with people with MS to better define their information needs and to guide the development of specific research strategies that will identify the most effective approaches to communicating this information. A series of forums could provide the needed perspective to defining these strategies and should include the following constituencies:
patients and their families;
health care providers;
allied heath professionals, such as physical therapists, occupational therapists, and social workers;
health services researchers, including survey scientists and clinical epidemiologists;
social scientists, including sociologists, anthropologists, and psychologists; and
representatives of organizations of patients with other disorders that present some of the same challenges faced by people with MS
As a disabling disease of young adulthood, MS inevitably disrupts careers.159,195 A 1992 survey of people with MS in the United States found that roughly 40 percent were employed.24 Although the unemployment rate for people with MS is clearly high (even at the time of diagnosis), it is important to consider unemployment rates relative to the base rate for the general population. These rates vary over time, place, and general characteristics of the population, such as educational level, as well as disease-related characteristics such as level of impairment and form of MS. For instance, a community-based study conducted in 1991 that included almost all of the people with MS in Olmsted County, Minnesota, found that more than twice as many people with MS were employed (53 percent)164 as reported in other U.S.170 and Canadian surveys.54,91 Two points are important to note. First, Olmsted County is an affluent community, with a low overall unemployment rate that was less than half of the national average at the
time of the study. Second, it included all people in the region with MS, not just patients attending a specialized MS clinic. Many people with MS—most likely those who are coping most effectively and most likely to be employed—will not see neurologists regularly. Although it has often been reported that within five years of diagnosis, 70 to 80 percent of people with MS are unemployed,147,170 it is important to know that this figure is based largely on data collected in 1975101 and might be biased by the fact that fewer women were employed than during their childbearing years than is the case today. A 1998 study found 72 percent unemployment among people with MS in the United States, compared to 36 percent unemployment among the general adult population.165 Forty-nine percent of unemployed people with MS in that survey indicated they were unemployed because of their disease rather than choice.
A number of factors are correlated with unemployment in MS. Women; people with severe physical impairment, visual impairments, ambulatory problems, or cognitive dysfunction; and people in lower socioeconomic strata are less likely to be employed than other people with MS.159,170 In general, employment rates are lower for people in wheelchairs or for those with steadily progressive disease, both of which apply to people with MS.48
Recent research has focused on barriers to employment and factors that might allow people to continue working. Barriers to employment for people with MS can be divided into three categories: personal (disease characteristics and an individual's educational and employment background); societal (physical inaccessibility of job sites, public attitudes, and inadequate job accommodations); and programmatic (vocational rehabilitation and policies promulgated by programs such as Social Security or Medicaid in the United States).147 Spastic paresis, which is slight or incomplete paralysis due to spasticity, is a major cause of the loss of ability to work.57 Managing fatigue and workplace demands is also difficult, particularly when the job requires working on a deadline, excessive walking, handling and grasping objects, working a full eight-hour day, or standing. Strategies have been suggested for patients to help them manage fatigue at the workplace (see Box 4.1). Incoordination and sphincter disturbances are significant factors, with many patients having more than one impairment.57 Other challenges include environmental factors such as excessive temperature; physical barriers such as entrances, stairs, and steps; and personal difficulties with remembering, writing, thought processing, vision, speaking, and communicating.170 It has been suggested that more people with MS could work if there were better access to the workplace or if they could work from home.57
Work not only provides income, it often defines the adult identity.212 A study of 604 people with MS described the emotional toll of having to leave work because of their illness.115 Those who had left jobs commented they had “lost a part of me” or that they no longer felt “a sense of being needed and of purpose.”24 Often they felt “out of sync” with their employed peers or were forced to give up traditional roles that were important to them, such as when husbands with MS left
Energy Conservation in the Workplace for Patients with MS
SOURCE: Stolp-Smith et al.195
work and wives had to take a job. Some people reported positive aspects to retirement. For example, some respondents noted that retirement had decreased their stress and permitted more time with family and friends,24 and women reported less strain in their roles as mother and spouse when MS forced them to stay home rather than work full-time.226 The experience of being diagnosed with MS can cause a person to redefine attitudes and self-perceptions, and to some degree, the high unemployment rate among people with MS might reflect changes in their own attitudes more than barriers presented by others.
For those with progressively degenerative disease the future becomes not only problematic but intensely threatening....I have known many MS patients who have abandoned life goals and projects simply because they received the diagnosis of MS and without regard to actual physical incapacity.
S. Kay Toombs214
One study of 72 people with MS reported a positive association between “learned helplessness” and unemployment, but no significant association with marital status or education.129 As with other cross-sectional studies, the causes underlying this association cannot be inferred from these data.
Many people with MS who lose or change their jobs to accommodate their condition suffer associated financial losses:24 70 percent of the low-income families, 43 percent of the medium-income families, and 24 percent of the high-income families in one survey reported having trouble meeting health care needs, basic living needs, or both. Rodriguez and colleagues reported that 77 percent of the patients responding to their survey maintained their usual financial standard without external support.164 Again, these data are from an affluent community, which might account for the relatively large percentage of people who were able to accommodate the financial burden of the disease.
Financial setbacks diminish not only the ability to live comfortably, but also the ability to afford medical care, services, home adaptations, or the equipment required to manage progressive impairment. Thus, the determination of when people qualify for disability compensation is paramount in MS, where disability can come and go and the decision process can take up to 18 months.111,195 The U.S. Social Security Administration (SSA) is currently rethinking how it determines qualification for its much-criticized disability insurance (SSDI) and supplemental security income (SSI) programs (see appendix F). The 1990 Americans with Disabilities Act (P.L. 101-336) makes discrimination on the basis of disability illegal and guarantees equal employment opportunities and access to public accommodations for persons with disabilities. The U.S. Supreme Court has heard several cases from people claiming disabilities under the Americans with Disabilities Act (ADA), and has publicly struggled with how to define disabilities in these cases.70 What these cases might portend for people with MS, especially those who primarily experience fatigue or sensory impairments, is unclear. It is also unclear whether this situation is worse for people with MS than for those with other disabilities. Physicians who provide services to people with MS often serve as sources of information and should be able to help patients deal with issues surrounding disability, employment, and the ADA.195
With the exception of developments that address the specific workplace needs of people with MS (see discussion of assistive technology), the most important advancements in employment issues will likely come through changes in national policies. While employment is an important contributor to individual well-being, it seems likely that the most effective way to improve employment opportunities for people with MS is within the broader context of the disability rights movement.
MEASURING FUNCTIONAL STATUS AND QUALITY OF LIFE
Quantifying functional status and quality of life for persons with MS is essential for several reasons. Given the chronicity and uncertain course of MS, tracking its impact over time can assist with care of individual patients, suggesting short-term prognoses and the need for various interventions. Tabulating these findings across individuals offers insight into the burden of MS-related disability within populations—information that is increasingly used to set research, health, and social policy priorities. Examining the trajectory of functioning and quality of life over time defines the patterns of progression and expands understanding of the clinical epidemiology of MS. Finally, functional status and quality of life are critical end points in measuring the effectiveness of therapy, for both clinical trials and routine patient care. Although disease activity can be measured by biological markers (such as lesions identified by MRI), these are only surrogate measures for the ultimate goal of improving patients' lives.
Over the last several decades, researchers have developed hundreds of measures of functional status and quality of life. Although the boundaries between the two sets of measures blur, functional status measures typically focus on observable behaviors and might or might not incorporate the patient's viewpoint, whereas quality-of-life measures are designed to assess the perceptions and feelings of individuals in relation to their disease and, necessarily, rely on patient self-report.64 Virtually all instruments used to measure health-related quality of life include a measure of physical function.150 Indeed, some observers find such distinctions spurious, preferring global measures of patient well-being that cut across various dimensions, and categorize quality-of-life measures as any measure that defines health beyond traditional biological function indicators.79,223
Both functional status and quality of life can be measured using generic or disease-specific survey instruments.152 Generic measures allow for broad comparison across studies, interventions, and types and stages of disease, while disease-specific measures focus on issues that are most relevant to individuals with the disease.79 Disease-specific measures are likely, but not guaranteed, to be more responsive to changes in a patient's condition.78
The wide variety of approaches to outcome measurement used in MS leads to problems when comparing studies. For example, in clinical trials of disease-modifying drugs for MS, different investigators have used different outcome measures in different populations. Direct comparisons of the trials are thus intrinsically imprecise, with the result that comparisons of the drugs are based more on expert opinions than on evidence.169 Functional status and quality of life are concepts that can be viewed in many different ways, and no single universally valid measure is likely to emerge.13,151 Nevertheless, standardized assessment methods that are multidimensional, quantitative, and include cognition evaluation are needed, particularly for the evaluation of patients receiving therapy.169 With these goals in mind, the National Multiple Sclerosis Society's Clinical Outcomes Assessment Task Force generated a list of the ideal characteristics of a clinical outcomes measure (Box 4.2).56
The modern outcomes movement, which developed in the 1980s, established the principle that the consequences of a medical intervention for its recipient should be a major criterion in determining its value.162 This reflects the awareness that biological standards of evidence alone do not completely depict the effects of a medical procedure. The recent interest in assessing outcomes of care has increasingly sought the patient's own voice, coalescing into the burgeoning field of health status and quality-of-life assessment.
Eliciting patients' own values about their health states is central to quality-of-life measurement—the values that two people place on a given health state can differ widely. For example, older people tend to be health optimists, having more
Characteristics of an Optimal MS Clinical Outcomes Measure
Adapted from Fischer et al.56
favorable health perceptions than their levels of physical functioning objectively allow.105 One study of gravely ill patients found that their health values varied widely and could not be clearly predicted based on their current state of health.215 Being heard is especially important for persons with disabilities. Those in what others may perceive to be “poor” health place a relatively high value on their own health since they have adjusted their life-styles and expectations to take account of their condition. This may be particularly true of young disabled men and women, since one-quarter of this group of respondents describe their health as “poor” yet value it as “good.” Conversely, young people who describe themselves as “healthy” can be reluctant to value their health near the top because they have high expectations about what being in the “best imaginable health state” involves.45 Health care professionals often see patients only in the setting of an
office or hospital unit and might conclude that the effects of MS on the person's personal life will be measured by the elements in the neurological examination.139 However, most aspects of health-related quality of life are only weakly related to measures of neurological impairment.55 Interestingly, several studies have found that the duration of MS is not consistently related to quality of life—either positively or negatively.6
Health status measures generally rely on extensive data obtained directly from patients, encompassing numerous attributes such as severity of illness, physical capabilities, psychosocial and emotional functioning, sense of well-being, and health-related quality-of-life.73,118,119,193,196,223 Numerous quality-of-life measures exist. A review of 75 articles involving quality-of-life measurement found that 159 different instruments were cited. Each article used anywhere from 1 to 19 instruments, with a mean of 3 per article.64 Some are generic to the extent that they assess health concepts that represent basic human values (Box 4.3);223 others are designed to capture the impact of specific diseases (Box 4.4).
The SF-36, a 36-item measure developed in the Medical Outcomes Study and comprised of eight dimensions, is the most widely used quality-of-life scale (Box 4.3).192,193 However, four of its eight dimensions cover functional limitations, so it can also be considered a generic measure of functional status.
Concerns About Quality-of-Life Measurement
Despite the obvious appeal of quantifying patients' experiences and perspectives, several caveats arise. First, the explicit effort to be inclusive frequently yields megavariable composite indices.53 The importance of specific variables, such as particular symptoms that are especially troubling or of prognostic importance, can be overwhelmed by numerous other factors incorporated in the composite index. Further, many functional status measures contain subscales (for example, the SF-36 measures eight dimensions), and single numbers do not adequately capture the full range of functional problems. For instance, how should an overall health score be assigned to a person with a serious chronic disease such as diabetes who feels well and functions as a productive person with no role or social limitations?193 As an example, three categories such as self-care, mobility, and physical activity could be satisfactorily combined into one functional status index, whereas personal and role functioning measures are not so satisfactorily collapsed into a single index.194
Certain quality-of-life measures have been developed specifically for patients with particular conditions, whereas others are generic. No single method suits all research needs,152 and choosing an approach depends on the specific research question. Mosteller and colleagues recommend routinely using both condition-specific and generic methods,137 while Patrick and Deyo recommend using standardized, generic instruments with disease-specific supplements.152 The SF-36, a generic instrument, reportedly yields reliable and valid results when
Examples of Generic Quality of Life (QoL) Questionnaires Used in MS
MEDICAL OUTCOME STUDY SHORT FORM 36 (SF-36)132,224
SICKNESS IMPACT PROFILE (SIP)14
NOTTINGHAM HEALTH PROFILE82
tested in people with MS, although it fails to capture important information about their quality of life that can be captured in MS-specific measurements.219 The authors of that study recommend supplementing generic measures of health-related quality of life with MS-specific scales.
Health status measures do not perform equally well across the entire spectrum of impairments or within selected patient populations. The Medical Outcomes Study general health survey (MOS-20) was largely designed and tested among ambulatory patients and is biased against documenting a decline in the
MS-Specific QoL Questionnaires
DISABILITY AND IMPACT PROFILE (DIP)108
FUNCTIONAL ASSESSMENT OF MULTIPLE SCLEROSIS (FAMS)26
MULTIPLE SCLEROSIS IMPACT SCALE (MSIS-29)80
MULTIPLE SCLEROSIS QUALITY-OF-LIFE INVENTORY (MSQLI)55
MS QUALITY-OF-LIFE-54 (MSQOL-54)220
health of severely ill patients, which would include many patients with advanced MS.15,59 When baseline scores are already near the bottom (or floor) of the scale, indicating poor health, further declines are less likely to be captured by the scale. The SF-36, which is similar to the MOS-20 in that it was derived from the same Medical Outcomes Study survey, also has problems with elderly people, poorly educated or impoverished persons, disabled people, and patients with both medical and psychiatric comorbidity.98,131 The SF-36 can produce ceiling effects for healthy, community-dwelling persons, even those who are elderly.3 In general, such floor or ceiling effects are more likely to be found on instruments with small numbers of items.15
Another important issue involves what to do when patients cannot respond themselves for any number of reasons, such as poor health, cognitive impairment, or physical limitations. One common solution is to ask a family member or close friend to serve as a proxy.17,190 The accuracy of a proxy's response in representing a patient's perceptions is, however, potentially suspect. Some studies find that although patients and proxies provide generally comparable assessments of overall health and physical functional status, proxies report significantly lower emotional health, social activity, and satisfaction than do patients.188 Others find that proxies rate both physical and psychosocial dimensions of functional status as more impaired than do patients themselves.21,122 Physicians also frequently underestimated patients' functioning.167
Functional status is a component health status and refers to the ability to perform activities of daily living.168,222 Researchers have developed measures of functional status that address either specific areas of functioning or a range of dimensions related to functioning (see Box 4.5 and Box 4.6). Core components of
Generic Measures of Physical and Social Functioning Used in MS
BARTHEL INDEX (BI)123
FUNCTIONAL INDEPENDENCE MEASURE (FIM)68
MEDICAL OUTCOMES STUDY SHORT FORM 36 (SF-36)132,224
functional status measures typically include basic activities of daily living (feeding, bathing, dressing, toileting, walking) and instrumental activities of daily living (shopping, cooking, cleaning house, telephoning, managing money, using transportation). Most comprehensive functional status measures also encompass cognitive abilities (level of alertness, orientation, long- and short-term memory, capacity for learning and computation), affective health (happiness, anxiety, de-
MS-Specific Measures of Physical and Social Functioning
EXPANDED DISABILITY STATUS SCALE (EDSS)104
FUNCTIONAL SYSTEMS SCALES (FSS) 104
MULTIPLE SCLEROSIS FUNCTIONAL COMPOSITE (MSFC)36,56
MINIMAL RECORD OF DISABILITY (MRD)90
INCAPACITY STATUS SCALE (ISS)55,90
ENVIRONMENTAL STATUS SCALE (ESS)55,90
CAMBRIDGE MULTIPLE SCLEROSIS BASIC SCORE (CAMBS)138
GUY'S NEUROLOGICAL DISABILITY SCALE181,182
IMPAIRMENT SCALE FOR THE EUROPEAN DATABASE FOR MULTIPLE SCLEROSIS (EDMUS)31
pression), and social activities (for example, visiting friends, sexual relationships). The context of measurement affects assessments of functional ability. “Capability” indicates what persons “can do” in controlled settings, whereas “performance” assesses what a person “does do” in everyday life. Capability typically exceeds performance.235
In response to the concern among MS researchers that no currently available outcome measure adequately measured changes in patients' conditions over time, the MS Society's Clinical Outcomes Assessment Task Force developed the Multiple Sclerosis Functional Composite (MSFC) (Box 4.6). The MSFC was designed to fulfill the optimal characteristics of clinical outcome measures previously developed by the task force (Box 4.2). Three measures (leg function and ability to walk, arm and hand function, and cognition) were derived from a statistical analysis of variables measured in six different natural history studies and in placebo groups from nine clinical trials. Preliminary results indicate that the MSFC is responsive to change over one- to two-year intervals, that it is reliable, and that the three dimensions are nonredundant.56 Independent validation studies suggest that the MSFC is reliable,29,30 has good construct and criterion validity,92 and is moderately to strongly associated with quality-of-life as perceived by patients (see Box 4.2 for explanation of terms).135 Wider use of the MSFC should facilitate collaborative data collections and comparison among clinical trials.
Measuring functional status raises several issues. First, as with other measures of health status, many functional status measures are generic in that they are independent of diagnoses or the cause of impairment. Although they do not permit comparisons among different treatments or conditions, condition-specific approaches are best for certain purposes. For instance, the Visual Analogue Pain Scale128 pertains to pain syndromes; the Tinetti Balance and Gait Evaluation211 assesses gait abnormalities; and the Activities of Daily Vision Scale (ADVS) focuses on visual function.126 These condition-specific scales are generally more sensitive to change in specified functions than generic measures. As described above, most investigators believe that generic instruments do not capture the full range of functional status concerns for MS patients.208
The mode of administration (face-to-face interview, mail with self-administration, or telephone interview) can affect responses. In face-to-face interviews, respondents may be reluctant to reveal the extent of their dysfunction. One study using the SF-36 found significant differences in patients' reports over the course of a week, depending on the mode of administration. For four of the eight SF-36 dimensions, face-to-face administration elicited a more optimistic view of functioning than did self-administration.227
Finally, as described above, perceptions about functioning frequently vary depending on who is asked—patients, proxies, or physicians.
The term disability is used in many conceptually different ways throughout the literature. The definition of disability has become so controversial that the World Health Organization published a draft report in 1997 proposing that the term disability be abandoned in favor of the more neutral term activity, to more
comprehensively assess the following three different dimensions: body structures or function, personal activities, and participation in society.233 At the time of this writing, the proposal had not been formally adopted, although the expanded concept of disability reflects current thinking.
Disability can generally be defined as inability or limitation in performing certain roles and tasks that society expects of an individual.87,88,233 Thus, disability reflects the gap between a person's capabilities and the barriers posed by the environment and society, and his or her ability to carry out tasks in the face of these societal and environmental barriers.88 Disability refers to much more than motor control, with which it is often equated. Impairment, which is defined as any abnormality of structure or function affecting the whole body, and handicap, which is any alteration in a patient's status in society, contribute to but are not synonymous with disability.
Not only is disability defined in different ways, it is measured and interpreted in ways that do not allow easy comparison. One problem is that the specific tasks or activities used to identify disability are not defined. As a result, scales that have been developed to measure disability contain different tasks, depending on the interpretation of the developers, and often intrude into the concepts of impairment, handicap, and quality-of-life.208 Although reliability and validity have been established for many of these scales, these tests are performed in different ways on different populations. Comparison of studies that used different measures is therefore problematic.
Finally, functional status measures and disability measures have been criticized as too narrow to be used as outcomes measures by themselves because they do not address the impact of the disease on the patient. The effects of MS on relationships, family, employment, and other aspects of life are not covered by functional status measures but fall under the concept of health-related quality of life.208 With respect to MS, no measure of disability has been shown to be superior to all others.
Expanded Disability Status Scale
The EDSS is the most widely used measure of disability in MS to the extent that it has been used in almost every MS clinical trial for several decades. Yet it is widely disparaged as a flawed tool. The EDSS is not a linear scale; it is bimodal. Patient scores are generally at the low or higher ranges of the scale, with relatively few in the mid-ranges, and patients spend more time at some levels than at others.208 It is not even strictly a measure of disability, because at different points on the scale, it mixes impairment (where it scores limb movement) with disability (where it scores ability to perform daily activities).
Several aspects of the EDSS make it insensitive to change. Once a patient has limited ambulation, other aspects of the neurological exam are not taken into account. Also the EDSS does not measure cognitive impairment, although this
deficiency is common to all scales based on the standard neurological exam. As noted earlier, a patient with MS can be unable to sustain a full day's work due to fatigue or other factors and yet have an EDSS score of zero. The EDSS is also heavily criticized for poor reliability.208 Consequently, even though its wide use should allow the advantage of comparing between studies, such comparisons should be made with caution.
Even though the EDSS is much criticized, there is no consensus on the usefulness of other disability measures for MS. Granger and colleagues68 concluded that the most useful disability scale for MS is the Functional Independence Measure (FIM), but van der Putten and colleagues217 concluded that the FIM and the Barthel Index (BI) were similar in their appropriateness and responsiveness in MS patients.68 Marolf and colleagues reported that both are more responsive to changes in a patient's condition than the EDSS.127 They measured the results of rehabilitation in 100 patients and found that about 30 percent of them showed improvements at the end of a four-week inpatient program, as measured by either the Extended Barthel Index or the FIM. However, only 5 percent of them showed any change in EDSS scores. The EDSS generally correlates with the physical function domains of the SF-36, but not with other aspects such as role functioning or mental health.18,144
The measurement of functional status and impairment is central to all aspects of clinical research on MS, and the development and validation of acceptable measures must remain a priority for MS research (see Recommendation 12).217
Assistive technology refers to any technology that helps support the individual's independence by enhancing or assisting performance of the activities of daily living.187 Assistive technology can be as simple as a bathtub shower grab bar or as complex as a motorized, voice-activated wheelchair. MS patients' needs for assistive technology are as variable and difficult to predict as the disease itself. Their needs can wax and wane with relapses, and the need for specific types of assistive technology will be determined by each patient's unique presentation and sequence of symptoms. The assessment of patients' needs and communication of the options available are thus especially important in MS.
Access to Assistive Technology
Patient reluctance and high costs are factors that hinder individuals from making full use of existing assistive technologies. People can be reluctant to use assistive technology because of the social stigma associated with disability, the
fear that reliance on assistive technology will promote further loss of function, and discomfort about assuming a new identity as a “handicapped person.”83,142
The high cost of assistive technology limits its availability to persons who need it. Since reimbursement is limited even for durable medical equipment such as beds and wheelchairs, the patient may be forced to pay the cost of devices such as scooters or of home modifications such as installing ramps and widening doors.24,83,142 Reimbursement for skilled occupational therapy services to match the needs of individuals with assistive technology is also limited, particularly for MS since the patient's needs vary over time142 (Michael Weinrich, personal communication). Most neurologists do not routinely recommend the purchase of a scooter or wheelchair until lower extremity and balance problems affect mobility, whereas physical therapists generally recommend the use of large equipment much earlier.33 It is not known which criteria will best match the level of individual capacity to the type of mobility aids for support of quality of life, safety, and long-range health needs.
It is important to note that assistive technology might pay for itself by affecting moneys in other areas, for example, when the individual is able to continue working as a result of technology.
Several technology databases are available on the internet to help clinicians and patients identify and locate appropriate technologies. ABLEDATA, sponsored by the National Institute on Disability and Rehabilitation Research, describes more than 25,000 assistive technology products, including the price and company information for each. REHABDATA, an online bibliography of publications, journals, and government reports on rehabilitation, provides information on assistive technology and is run by the National Rehabilitation Information Center and funded by the National Institute on Disability and Rehabilitation Research.187 Several web sites provide recent information on research and events relevant to MS. In addition, telemedicine can help patients with wound management, self-injection support and training, education and support during the initial self-catheterization period, emotional and physical adaptation to increasing disability, and family education.
Technology can enhance independence through assistance with mobility control, environmental control, communication, and employment.96
Novelist Nancy Mairs, who cannot walk because of MS, travels the world in a motorized wheelchair:
Relaxed and focused, I feel emotionally far more “up” than I generally did when I stood on two sound legs....Certainly I am not mobility impaired; in fact, in my Quickie P100 with two twelve-volt batteries, I can shop till you drop at any mall you designate, I promise.125
Fatigue and impaired mobility are frequent problems in MS, and the use of technologies that enhance mobility can provide empowerment and relief to patients.83,142 Mobility aids also reduce the risk of life-threatening and costly injuries, and can be an economical alternative to costly personal assistance and institutionalization.96
A variety of assistive technologies aid mobility for both ambulation and patient transfer.187 For ambulation assistance, canes, walkers, power and manual wheelchairs, scooters, and a spectrum of wheelchair accessories that allow for voice control, a joystick, or head movement are available. Bathtub benches, shower chairs, grab bars on walls, and lift systems assist with transfers to and from the bed or in the bathroom. Aids for access to community include adapted vehicles, portable and permanent driving hand controls, portable and permanent ramps, and wheel chair lifts. Aids for accessing the upstairs of a home include stair lifts, small residential elevators, and wheelchairs that climb stairs. Functional electrical stimulation is a technology that stimulates muscles that are no longer motivated by nerve impulses.153 This technology, originally conceived in the early 1980s, is relatively new, and research is needed to improve its functionality and reliability. Finally, portable and fixed cooling devices help with fatigue management and exercise participation, especially for pool therapy use.103,142
Technology can assist with environmental control in a variety of ways, including controlling the power and output from devices such as electric doors, fans, telephones, TVs, radios, stereos, lights, heating and air conditioning units, and drapes. Low-technology devices such as reachers, alternative types of door and drawer handles, buttonhooks, and kitchen items can be adapted or configured for more functionality. Mouthsticks and head pointers can assist individuals who cannot use their hands. High-technology devices such as electrically powered feeders, electrically powered page-turners, or environmental control units are also available. Permanent or temporary home environment modifications, such as lowering kitchen counters and cabinets to wheelchair height, can be helpful. Finally, voice-activated computers can be linked to robotic environmental controls and remote controls.
Since the onset of MS occurs in the years when most people are deeply involved with their careers, technologies that allow people with MS to keep their jobs are especially important. The most frequently cited reason that people with MS left their jobs was physical inability to perform job tasks; the use of a wheelchair by MS patients was also associated with unemployment.170 Adaptive equipment has been related to continued employment among persons with increasing
physical disabilities, as adaptive keyboards, voice control systems, and adaptations of the workstation expand the range of tasks that people can perform.48 Aids for effective seating and positioning such as adapted desk chairs and chair cushions reduce pressure and fatigue, support posture, provide comfort, and enable function.71
Communication becomes increasingly difficult as MS progresses, particularly when vision, speech, and memory are affected. Visual problems are aided by magnifiers and eyeglasses as well as nonoptical aids such as enlarged print, high-intensity lamps, high-contrast objects, and enlarged and tactually labeled aspects of self-care and leisure items. Electronic aids, such as computer software that enlarges text and images, and closed-circuit television devices (CCTV), can increase image size, color contrast, and overall brightness.
In the later stages of MS, the patient's speech can be difficult for others to understand, and the ability to talk can be lost. Technologies that increase the ability of the patient to communicate include speech volume amplifiers, speech enhancers to clarify speech, and text-to-speech voice synthesizers. These devices can increase social interaction and, along with portable home emergency call systems, serve as important safety devices that can be used to call for help when an accident has occurred in the home.
Internet. The computer has opened the doors to enhancing communication for people with MS like no other technology before it. The Internet is the ultimate community access tool and information resource, with no restrictions on mobility or speech faculty beyond access to the technology. There are limitations, but they are surmountable. These limitations include screen size, the standard keyboard and mouse that are difficult to operate when fine motor control is impaired, and the need for a workstation to accommodate wheelchair use or use while in bed. In addition, the Internet can be used to spread false or misleading information, and consumers have to be careful in their search for information from reputable sources. Another limitation of this powerful tool is the digital divide. Many people cannot afford a home computer or lack the skills and knowledge to make use of one.
The Internet can serve health needs in a variety of ways. It can be a source of medical advice and information on the latest research results. It can be used to contact health care organizations for information, scheduling of appointments, and communication with physicians. It allows communication among members of support groups. Also, it is a source of information on goods and services, such as assistive devices, that are on the market. Tasks, such as ordering personal and household items, banking, and paying bills, can be accomplished through computers. Certain services, such as grocery shopping, are often limited to urban
areas and are unaffordable for many people. Nonetheless, such a service might be cheaper than the cost of personal assistants.
Research Needs in Assistive Technology
The greatest needs for the field of assistive technology in MS include strengthening the research on assessing and evaluating patient needs and improving the dissemination of information and equipment to patients to fully address their needs. Persons with MS frequently have little awareness of assistive technology options and professional support available in assistive technology (Peggy Neufeld, personal communication, Box 4.7).
Many specific research questions remain unanswered in the area of assistive technology and MS, and research should focus on ways to maximize the positive effects of available and emerging technologies on quality-of-life. Research on the satisfaction of consumers, therapists, and third-party payers with various products would provide direction for manufacturers of assistive technology to improve available technologies and new designs. Input from MS patients in the design of new technologies is crucial.
Development of effective educational methods for demonstrating the application and operation of assistive technology to health care professionals and people with MS would increase the use of available technologies and increase
Assistive Technology That Would Be Particularly Useful to People with MS
Source: Peggy Neufeld, personal communication.
patient safety (David Krebs, Peggy Neufeld, personal communication). Strategies to disseminate information, appropriate training methods, and self-help strategies to enhance the ability of persons with MS to select assistive technology will help educate both patients and care providers.
The optimal time for the introduction of assistive technology during the rehabilitation or therapy process should be studied. Strategies to reduce the social stigma associated with disability should be examined. Cost factors should be analyzed to determine when the use of assistive technology by people with MS results in economic benefit or loss. Finally, research should take place in natural environments using people with MS as subjects, and the MS consumer voice should be included in every aspect of assistive technology design, research, and policy development.
Support Groups and Activities
There are several centers that provide life-enhancing experience for people with MS. For example, the committee met with Brian Hutchinson of the Jimmie Heuga center in Edwards, Colorado, in the Rocky Mountains. This center offers a variety of programs to promote general health for people with MS through exercise and psychological well-being. Charlotte Robinson, founder of Adventures Within, who also spoke to the committee, leads outdoor adventure tours where people with MS can go rock climbing, rappelling, horseback riding, and whitewater rafting. While participants in these programs enthusiastically extol their benefits, the impact of this type of program has not been measured. One can, of course, imagine innumerable benefits ranging from the rejuvenation offered by any pleasant vacation, to the companionship of other people with MS, to the discovery of unrealized capacities and learning new coping strategies.
Other types of group programs include “wellness” programs in which people with MS work on developing individual coping strategies and share their own knowledge with other participants.
Patient Care Services
One of the greatest fears of the elderly and persons with disabilities is that they will eventually be “warehoused” in an institution where a life with dignity is impossible. As a person living with progressive neurological disease, my greatest fear is not unbearable pain, nor even death. Rather, I am most afraid that my illness will progress to a point where neither I nor my family will be able to cope.214
The options for care of someone disabled by MS range from in-home care provided and financed by the family to publicly funded care in an institutional setting. Most people prefer care that maximizes their independence and comfort, but the cost can be prohibitive for individuals. There are only a few published
reports that analyze the cost-effectiveness of different modes of care. A British study reported that in-home care is more cost-effective than hospital care, even when home care includes a paid nurse.229 However, any analysis of cost-effectiveness must take into account the health care financing system of a particular country. Observations for people with MS who live in countries where comprehensive health care is publicly funded will not necessarily apply to those living in countries where health care is privately funded or where there is a combination of public and private funding. These are national public health policy issues germane to all chronic, disabling conditions and must be addressed at that level and studied in the context of other chronic and disabling conditions.
This is an important and relevant issue in the management of MS because it incorporates acute hospital and neurorehabilitation services together with community-based activities and, in essence, has to bring together medical and social services in a way that meets the complex and ever-changing needs of the person with MS. The key components of such a service have been listed as including the following:
it should offer support at the diagnostic phase;
there should be a high level of expertise;
it should be comprehensive and flexible;
it should be accessible and coordinated; and
it should be linked to a neuroscience center.
Ideally, most services should be community based, with supporting expertise from the acute hospital or rehabilitation center at times of particular need (for example, at diagnosis or at the time of a severe relapse) or complexity (when multiple symptoms interact and intensive inpatient rehabilitation is required). The optimum method of service delivery has not yet been defined, and little work has been done to compare existing services. A recently completed, though not yet published, study carried out in Rome compared two forms of service delivery in a randomized controlled trial of 201 patients with MS. One group (133 patients) received what was described as “hospital” home care, in which patients remained in the community but had immediate access to the hospital-based multidisciplinary team as and when required, while the other (68 patients) received routine care. The range of outcomes, which included EDSS, FIM, SF-36, and measures of anxiety and mood, were carried out at baseline and at 12 months. No difference was seen in the level of disability between the two groups, but the more intensively treated patients had significantly less depression and improved quality-of-life. Another study, comparing the benefits of multidisciplinary MS clinics, general neurology outpatient settings, and community-based general practice in 150 patients (50 in each group) has just commenced in the United Kingdom. Patients will be evaluated at baseline and at three-month intervals for one year, with a range of outcome measures from impairment to self-efficacy.
Rehabilitation is often defined as the process of restoring or developing physical, sensory, and mental capacities in people with disabling conditions—in other words, reversing the disabling process.88 However, this definition is too restrictive for MS or for any other degenerative condition. The World Health Organization has adopted a broader perspective that applies better to people with MS. It defines rehabilitation as an active process by which those impaired by injury or disease achieve a full recovery or, if a full recovery is not possible, realize their optimal physical, mental, and social potential and are integrated into their most appropriate environment. This definition expresses a philosophy that also emphasizes patient education and self-management and is ideally suited to meet the needs of such a complex and progressive disorder. An essential element of this definition is that an overarching goal of the rehabilitation process is to improve a patient's sense of well-being.61
The essential components of successful rehabilitation include expert multidisciplinary assessment (including an assessment of patients' perspectives and circumstances), goal-oriented programs, evaluation of impact on the patient, and evaluation of goal achievement. The wide range of coexisting symptoms in MS produces a complex pattern of disability, including the possibility that treating one symptom might worsen another. Any management strategy has to take this complexity into account. It is also apparent that comprehensive management will invariably require input from different treatment modalities, including the provision of information, patient education, therapy from a range of disciplines, and drug treatment. Finally, the variable and fluctuating nature of MS means that the needs of individual patients will change over time, often quite abruptly, and that these needs will tend to increase over time.
Evaluating Outcomes in Neurorehabilitation
If there is to be ongoing improvement in the process and impact of rehabilitation, there must be an evaluation process. This essential component is probably also the most challenging. It requires the use of outcome measures that are scientifically sound (reliable, valid, and responsive to changes in a patient's condition), clinically useful (short and simple), and appropriate to the sample under study and the intervention being evaluated. In neurorehabilitation, improvements are not sought at the levels of pathology and impairment but rather in disability and handicap (or activity and participation, as proposed by the World Health Organization233) and in the broader, more patient-orientated areas of quality-of-life, coping skills, and self-efficacy. The standard outcome measure in MS therapeutic trials is the EDSS, but it is inappropriate for evaluating rehabilitation, not only because of its scientific limitations, but also because it does not measure many of the dimensions relevant to health-related quality-of-life such as mental
and social function.81 Indeed, the EDSS score can be zero even though a person is unable to sustain a full day of work. Consequently, a number of generic measures of disability and quality-of-life have been utilized in MS rehabilitation.
Integrated care pathways, which detail the expected interventions occurring within a given episode of clinical care, are used to monitor and audit both the process and the outcome of rehabilitation. They can be used to evaluate the rehabilitation process, including goal achievement, and serve as an excellent audit tool206 that can document when goals are not achieved on time; more usefully, however, they can indicate why this has occurred (for example, because of underestimation of cognitive dysfunction or the impact of fatigue).
Evaluating any intervention within the context of a randomized double-blind placebo-controlled trial in a condition as variable and unpredictable as MS is inherently difficult. Yet evaluating interventions aimed at neurorehabilitation—an approach that encompasses such a broad range of approaches and that must, at the same time, be tailored to meet the specific needs of an individual patient—poses still another dimension of challenges for clinical trial design. Chief among these are a lack of detailed description of programs (for example, the number of disciplines involved or techniques employed) and inadequate standardization of an intervention including its duration and location (inpatient, outpatient, or community based). Therapists are reluctant to use control groups, and limited resources often prohibit the use of independent assessors, which is particularly important when masking a patient's identity is difficult. Finally, there is no consensus as to what the most appropriate outcome measures are, and until recently, limited and often-inappropriate tools have been used inconsistently.
Despite these obstacles, a number of recent studies have demonstrated that it is indeed possible to achieve some degree of evaluation for rehabilitation interventions, although many more studies are required. In order to assess the data that these studies have produced, it is useful to consider four distinct levels, moving from the broadest concept of (1) service delivery, to (2) packages of comprehensive rehabilitation, (3) individual components of that package, and finally the most specific, (4) the intrinsic components of the rehabilitation process such as assessment and goal setting. Most studies have focused on comprehensive packages of rehabilitation (predominantly inpatient), but some have addressed individual components including physical therapy and aerobic exercise, and recent studies have compared different forms of delivery of care. There are no published studies of the intrinsic components of rehabilitation as they apply to MS.
The following are two key questions that have to be answered:
1. Is comprehensive rehabilitation effective in reducing disability and handicap and improving quality of life?
2. If so, how long do these benefits last?
The majority of studies have evaluated inpatient rehabilitation programs, which are generally more accessible than outpatient programs to controlled studies. Two studies, both retrospective and relatively small, have compared inpatient rehabilitation with other forms of intervention. One study compared outcomes of 20 pairs of patients who were treated either in an inpatient rehabilitation hospital or in an acute care hospital.161 Outcomes for the two groups were comparable at 16 months, although it should be noted that the acute care hospital was an MS center and thus provided more sophisticated care for MS patients than an average hospital. The other study compared the outcomes of 67 patients who received either inpatient services (in the same rehabilitation hospital as in the previous study) or outpatient treatment provided by visiting nurse services.58 Three months after discharge, the inpatient group showed less disability, as measured by the Incapacity Status Scale (ISS), although both showed gradual worsening, and by 12 months there was no difference between the two groups.
Freeman and colleagues designed a randomized controlled trial that overcame some of the methodological issues discussed earlier.60 Sixty-six patients with progressive MS were stratified at the beginning of the study according to their EDSS, and the treatment group received a short period of inpatient rehabilitation (mean of 20 days). A group of patients who were wait-listed for rehabilitation treatment served as a control. The two groups were matched in relation to age, sex, disease pattern, and duration. Disability (Functional Independence Measure) and handicap (London Handicap Scale, LHS) were measured at the beginning of the study and six weeks later. The treated group showed a significant benefit in both disability and handicap compared to the control group. No change in the EDSS was seen in either group.
A more recent randomized single-blind trial evaluated the benefit of a three-week inpatient rehabilitation program with a home exercise program in 50 moderately impaired ambulatory patients (EDSS 3.0-6.5).189 The control group did only the home exercise program. Patients were evaluated with the EDSS, FIM, and a health-related quality-of-life measure, the SF-36, at the beginning of the study and at 3, 9, and 15 weeks. As in previous studies, neither group showed a significant change in impairment (as measured by the EDSS). Disability, as measured by all of the FIM motor domain scales, was significantly reduced in the rehabilitation group, and this benefit was sustained for six weeks after the three-week program ended (nine-week time point). However, only two subscales, self-care and locomotion, continued to be significantly different between the groups 12 weeks after the program ended. At the end of the three-week period, the rehabilitation group had received significantly greater benefit in the mental health components of the health-related quality-of-life measure. This beneficial difference between groups was still apparent 6 weeks after the end of the rehabilitation program but had disappeared by 12 weeks (15-week time point).
One study, in which 50 patients were studied at three-month intervals for one year after their discharge from an inpatient rehabilitation unit, illustrates the
dependence of the observed outcome of rehabilitation on the measure used. In that study, improvements in disability and handicap were maintained for approximately 6 months, emotional well-being for 7 months, and the physical component of health-related quality-of-life for 10 months.60
Although few researchers have attempted to evaluate outpatient-based rehabilitation in MS, a 1998 study reported that fatigue and the frequency of other MS symptoms in progressive MS patients were significantly reduced after outpatient treatment.44 A group of progressive MS patients was randomly assigned either to an active treatment group of 20 patients who received five hours outpatient therapy a week for one year or to a control group of 26 patients that was wait-listed for, but had not yet received, therapy. The range of outcome measures included an MS-related symptoms checklist composite score, a measure of fatigue frequency, and items from the Rehabilitation Institute of Chicago Functional Assessment Scale. The services provided in the study included therapy aimed at physical function but also involved support for the family as well as the patients, social support, assistance locating community resources, organized recreation, and counseling to improve coping skills. Indeed, the authors suggested that the observed therapeutic benefits might be attributable to the emphasis on social and emotional support.
All 11 studies summarized in a recent review of the outcome of comprehensive inpatient rehabilitation in people with MS indicate a potential benefit in the area of disability.207 It is difficult, however, to combine the results of all these studies. There are major methodological differences among them, and few reach an adequate scientific level. Nevertheless, they all suggest that organized patient-centered multidisciplinary rehabilitation benefits MS patients. The degree of benefit and the extent of carryover have yet to be determined.
Components of the Rehabilitation Package
Few studies have looked at therapy intervention in the management of MS, and the only specific modality examined has been physical therapy. A randomized control trial of inpatient physical therapy (6.5 hours over two weeks) was carried out on 45 patients.62 Outcome measures used in the study included the Rivermead Mobility Index, the Barthel ADL Index, and a visual analogue scale of “mobility-related distress.” The treatment did not show significant benefit on either mobility or ability to perform activities of daily living but did significantly reduce mobility-related distress. However, the same authors have recently demonstrated a significant benefit from outpatient or home-based physical therapy.228 A second study went a step further and attempted to compare two forms of physical therapy.120 This pilot study involved 23 patients, 20 of whom completed the study. They received what was described as an impairment-based “facilitation approach,” while the other group received a more disability-based task-oriented approach. Patients received a minimum of 15 sessions over five to seven weeks.
The outcome measures were mobility based and included the 10-meter timed walk and the Rivermead Mobility Index. Not surprisingly, no difference was seen between the two small groups, but both improved from baseline (p < 0.05).
The impact of aerobic exercise was evaluated in a population of 46 patients with relatively mild MS (EDSS of 6 or less): 21 of the patients were randomly assigned to a 15-week exercise program, while 25 had no exercise over that period.154 The wide range of outcome measures included aerobic capacity, isometric strength, the Fatigue Severity Scales (FSS), the EDSS, and the Sickness Impact Profile (SIP), which measures quality of life. Significant changes from baseline were seen in the exercise group over the 15 weeks in the physiological measures and the physical component of the SIP. There was little sustained change in the psychosocial domain of the SIP and none in the EDSS or FSS.
Although there is good empirical evidence to support coordinated expert service delivery in other diseases, there is little evidence currently available to support this concept in the management of MS, and further studies are required. There are few adequately designed studies to support the role of rehabilitation therapy in MS, but recent studies have confirmed that such assessments are now feasible and urgently need to be carried out.
INFORMATION AND COMMUNICATION
What People Say
Problems of delivering the diagnosis MS were touched upon in the earlier section “You Have MS.” Patients generally say they want concrete information about their diagnosis, even when it is uncertain. Although it has been common for physicians to euphemistically tell a patient he or she has an “inflammation of the nervous system” or something equally vague, this is not what patients say they want (reviewed in 1999 by McGuinness and Peters130). A retrospective study in Britain found that approximately 60 percent of the people with MS in Southampton felt they were not given enough information at the time of diagnosis.133 They also reported being angry over the delay in the prompt provision of a diagnosis. At least part of that delay was due to the inherent difficulties in diagnosing MS, including the diagnostic requirement for more than one episode, and there are no published data to determine whether MS patients' anger at the delay is reduced when the difficulties are explained to them. It should also be kept in mind that most reports on patients' reactions to diagnosis do not reflect current approaches to diagnosing MS. Most of the studies were conducted in the mid-1990s or earlier and were done before MRI scans were as common as they are today and time to
diagnosis was longer. Without more information, it is not possible to know whether patients' frustration arises from a failure to communicate on the part of their physicians or from the reality that MS is difficult to diagnose.
Information for Newly-Diagnosed Patients
Effective communication about MS to the patient and family is especially crucial around the time of diagnosis. Not only is the patient wholly unprepared for such life-altering information, but after this point, many patients will see an MS specialist only rarely. Their experience with health care providers at this point will profoundly influence their future health care choices.
Withholding the diagnosis raises suspicions of physician dishonesty. These experiences may compromise patients' trust of their doctors, heightening fears that physicians will abandon them. Studies of other diseases have shown that such trust is essential to developing strong therapeutic relationships.205,210 Koopman and Scheitzer100 suggested that a supportive care model would help patients with MS and their families during the stresses surrounding the diagnosis, noting: “It is important for patients and their families to experience the first years of MS coupled with caring and consistent support from their MS team....A case management or primary nursing delivery system may be the best option.”100 They did not test the role of a case manager-directed care model in delivering care to people with MS. Interestingly, another study found that patients who had not gone to an MS clinic in the prior two years were more likely than others to “have come to terms with” their disease.6
Although almost every study reports that patients say they would have preferred full disclosure during the diagnostic phase, these are retrospective views of people with MS.57 However, one cannot assume that MS patients would have felt the same at the moment of diagnosis as they do many years later. To avoid the bias introduced by changes in people's perspectives over the passage of many years after their disease has progressed, studies on the outcome or effectiveness of different communications should be done prospectively. The committee found only one prospective study that measured the effect of diagnosis on patients suspected of MS.146 They were divided into three groups, those with MS, without MS, and those whose diagnoses remained uncertain. Diagnostic testing benefited all three groups. It reduced patients' distress over their physical symptoms and their general anxiety, even in those with MS. However, while the study measured the impact of diagnostic testing, it did not provide specific information about how the diagnosis can be imparted most beneficially to patients and their families.
In 1993, the British Society of Rehabilitation Medicine with the support of the MS Society of Great Britain and Northern Ireland published guidelines for imparting the diagnosis (Box 4.8).57 These guidelines represent the consensus of a committee of people with MS, their families, health care professionals, and service providers. Also, while an association with specific health or psychologi-
Guidelines for Delivering the Diagnosis of Multiple Sclerosis
Source: Ford and Johnson57
cal benefits has not been demonstrated, the guidelines are at least reasonable. It would be helpful to know what approach to communicating a diagnosis of MS is most conducive to successful adaptation to the illness and to establishing a productive relationship with one's physician. The MS Society developed a highly informative pamphlet, book, and Web page on information for the newly diagnosed. This information has been widely distributed and is generally believed to be very helpful, but its effectiveness has not been evaluated.
Given that the cause of MS remains unknown and the course of the illness is maddeningly unpredictable, one should consider the possibility that no amount of available information will satisfy. Patients will be frustrated by the lack of information, almost by definition. At the least, the background of unpredictability of the disease course should be considered as a potential factor in determining optimal forms of communication about health-related information.
Patients do not generally want to learn about demyelination when they are diagnosed.10,140 They want to learn how to live. From the patient's point of view, while information about disease mechanisms might satisfy intellectual curiosity, it provides only marginally useful information for living with MS. At the time of diagnosis, it seems only reasonable to deal first with such practical concerns. Patients need to know the extent to which they can continue to fulfill their responsibilities at work and home; how and whether they should alter their family and career plans; reassess their needs, obligations, and desires; choose therapies; and—especially in the United States where health care coverage is fraught with uncertainty—how they are going to meet their future health care needs. Furthermore, with every change in their condition, they will have to solve each of these
puzzles anew. Frequently, neither patients nor their doctors know where to go or whom to ask for advice. Only by understanding the specific hurdles that people face can initiatives be designed for improving the lives of people living today with MS.
Parameters of Effective Communication Strategies
The desire of people with MS for more information is clearly a recurring theme, but there are many questions to answer before this general need is translated into effective communication strategies. What sort of information do people need most, and how is the available information most effectively shared? The first step in addressing these questions is to identify the communication goals—goals that might be defined somewhat differently by patients than by their physicians.
Many of the published reports implicitly assume the goal of short-term patient satisfaction in their consultation with their health care providers, but there are other goals. For example, a more ambitious goal of communicating information about MS is to foster long-term improvements in their health, ability to function, and psychological well-being for people with MS—in short, to facilitate patient adaptations to living with MS. Although this goal is not inconsistent with immediate patient satisfaction, it is also not necessarily a consequence of patient satisfaction with the physicians. This highlights the need to consider the temporal aspects of communication. Long-term goals are often best served by different approaches to communication than those that work best for short-term goals.
Important considerations in effective health communication include who defines the problem (or information need) and who defines the solution (what is to be communicated and how). Identification of both problem and solutions requires broad input—from patients and their families, as well as health care providers (physicians, nurses, physical therapists, and psychologists). While the perspectives of patients, physicians and other providers are likely to overlap in many respects, each will have a greater understanding of different elements of disease management. Ultimately, the most important outcome is improvement in the patient's quality of life.
An important principle that has emerged from recent advances in communication theory and practice is that communication is most effective when it is targeted to specific populations.230 Different strategies work better for groups of different educational, ethnic, and economic backgrounds. This is even more important for people with MS, where the sheer variety and magnitude of the manifestations of the illness add to the already considerable differences among healthy populations. Another key principle of health communication is that no single communication or form of communication will be adequate. A pamphlet mailed to the patient or a scheduled visit with a physician will always be insufficient if it
is the only information provided to someone with MS. Individual differences might also be important in determining optimal communication strategies. Some might absorb information best in group settings, whereas others might absorb information better in a private consultation or via written, videotaped, or computer material.
Other principles of effective communication stem from the specific features of MS, particularly the unpredictability of the disease course and changing levels of impairment. The information needs of people with MS change throughout their illness. As these needs change, optimal communication strategies will also likely change. For example, approaches to communication will be very different during periods of stability when information can be considered carefully, free of pressure to take immediate action, compared to periods of crisis when psychological stress is high and quick action is required. Examples of such periods include exacerbations when a person's ability to function declines, when a caregiver is suddenly unable to meet the person's needs and new arrangements have to be made on short notice, or during a transition state, such as when a person suddenly needs to use a wheelchair or scooter for the first time. (As discussed in the section on assistive technology, information on wheelchair use is sorely needed, but rarely easy to obtain.) Each of these difficulties brings with it new needs for information, and there has been almost no research to identify optimal modes of fulfilling the diverse information needs of people with MS. Finally, for people with MS an important consideration is that their access to information will vary, depending on their mobility (whether they can easily get to a group meeting or clinic), as well as on economic factors (whether they can afford a computer, internet access, scooter, or other adaptive devices).
Although there is a large body of research on optimal communication strategies, empirical research on the information needs of people with MS and effective communication strategies to meet these needs are starkly lacking. Because this is such an overarching need of people with MS, it should be an area of emphasis in MS research. The Institute of Medicine committee did not include the expertise to identify specific research strategies but suggests that the MS Society consider developing a request for proposals (RFP) to assess and address communication needs of MS patients. Ideally, the RFP would target researchers outside the field of MS, but would request that the project be a collaborative effort between experts in health communications and those whose research expertise includes knowledge of MS symptoms and measurement of the quality of life of people with MS. Much of the literature on health communication focuses on healthy populations and is unlikely to be particularly relevant to people with MS. Although the relapsing-remitting form of MS presents the distinctive challenges posed by unpredictable impairment, many of the problems faced by people with MS are also faced by others with other serious and chronic diseases. For example, the research that has been done on improving approaches to imparting the diagnoses of cancer and AIDS would likely add some insight to the commu-
nication challenges posed by MS. The depth of knowledge and rate of progress will improve if the research on communication is not restricted to MS but includes other relevant conditions.
At a minimum, the MS Society should work in an ongoing way with patients to evaluate their information needs and how the information should be presented, including who delivers it (physician, nurse, other), when (at diagnosis, follow-up, once, several times), and how (private consultation, mailings, pamphlets in doctors office, Internet chat groups, participatory group meetings).
The Information Highway
The power of computers to deliver health information continues to accelerate. There is, of course, a quality problem. A recent National Research Council report concluded that it is not adequate for health care delivery per se, but the delivery of information is a distinctive aspect of health care, and for this, there is a wealth of excellent and clearly presented information that should be understandable to any college graduate, much of it supplied by the MS Society (United States), as well as the pharmaceutical companies that market the currently approved disesase-modifying therapies (Berlex, Biogen, Serono, and Teva Marion Partners) and a number of research organizations. This information does not, however, cover all aspects of MS equally well. While there is an abundance of high-quality information about the biological aspects of the disease, as well as descriptions of the symptoms of MS, there is comparatively little information about specific adaptations to assist people with MS in adapting to their illness.
Of course, it is important to emphasize that this invaluable resource is not equally available to all. Only about 40 percent of the population of the United States and Canada were regular Internet users as of March 2000, although this figure is steadily increasing.216 In Europe, the figure varies from country to country, but even in Sweden and Norway where Internet access is greatest, it is still only about 50 percent. Low-income, rural populations, and people with no college education have disproportionately less access to information posted on the Internet, and different approaches to meeting their information needs should be considered. A 1998 survey reported that 42 percent of people with MS surveyed in the United States have Internet access, suggesting that their access is somewhat greater than that of the general population.165
Access to Health Care
Costs and payment policies present major barriers to obtaining mobility-related services. Health insurers typically restrict the numbers of physical therapy
sessions, paying exclusively to restore patients to baseline function. Insurers, with some legitimacy, argue that financing therapy to maintain function or prevent its decline could generate insatiable demand. Despite its older and disabled population, Medicare, for example, limits coverage of interventions targeting functional status, and payment for rehabilitation services depends on continued functional improvements.23 Many of these services have not yet garnered a rigorous “evidence base,” which has seemingly become essential for insurance coverage.
Insurers often deny coverage of mobility aids. A 1990 national survey, not specific to people with MS, found that about 2.5 million Americans needed assistive technologies, including mobility aids, but 61.1 percent could not afford them.109 Almost half of those using assistive devices paid for them entirely out-of-pocket. Improving home accessibility (for example, grab bars, ramps, widened doorways) is also expensive; 78 percent of these improvements came out-of-pocket.109 Insurers routinely require doctors to certify the “medical necessity” of equipment. Despite this assurance, initial requests can be denied,85 requiring lengthy and frustrating appeals. The experience for people with MS is unknown.
Even if coverage is approved, insurance payments may be incomplete. Medicare's Supplemental Medical Insurance (Part B) covers such mobility aids, but beneficiaries must pay 20 percent coinsurance.1 Insurers sometimes deny payment for “lower-tech” equipment, arguing that the patient's rate of functional decline means more sophisticated, costly equipment will soon be needed,225 for example, they might pay for a powered wheelchair but not for a scooter. Other payment policies affect the use of mobility aids. Medicare, for example, limits home health services to people confined to their homes except for medical appointments.76 Medicare beneficiaries who obtain wheelchairs to leave their homes for other purposes will lose home health services. Whether financial barriers are impeding people with MS from obtaining assistive technologies is unknown. Quality-of-life could be diminished by inadequate access to these devices.
Attitudes and environment present barriers, even for health care services access. Justice Department investigations discovered persistent problems even with physical access to health care sites.158 Iezzoni and colleagues86 found low rates of Pap smear and mammography use among women with major mobility impairments; the study controlled for demographic characteristics and differences in access to health care. This fits with qualitative and anecdotal reports about barriers to primary and preventive care for people with disabilities.16,19,39,63,156,197,198
Toombs wrote that her primary care doctor questioned her need for bone density screening for osteoporosis, rationalizing that wheelchair users never fall.213 One internist described her private practice in an upper middle class community, observing that although wheelchair users “probably could get into the building they would not have been able to get into the office and, certainly, would not have been able to get into the examining room.”4 When she moved to a Medicaid health maintenance organization (HMO), she encountered more patients with disabilities. One of her patients, a 45-year-old woman with MS, had
never had a Pap smear; nobody had ever offered her one. When the internist and her assistants tried to move the patient onto the high, unadjustable examining table, they failed. The patient's daughter, familiar with the maneuver, transferred her mother. The internist next ordered a mammogram, but the facility could not serve her because she could not stand up.4 Given the long lives of most people diagnosed with MS, more research on access to preventive and screening services, including physical access to care sites (for example, adjustable examining tables, mammography machines) is needed.
A 1990 survey of 604 families found that almost everyone with MS had some form of health insurance paying an average of 75 percent of the medical bills, but 28 percent reported that the coverage was inadequate for their health care costs.24 Failure of insurance to pay for expensive drugs was particularly problematic, as were insurance exclusions for “preexisting” conditions. Some respondents reported staying in unhappy marriages to retain their health insurance.
Participatory Style with Physicians. The literature outside MS suggests that patients who participate more actively in decision making about their care may do better.93,94,95,166 In one study, patients who assumed control of conversations (for example, asking more questions, attempting to direct the flow of the discussion and their doctors' behavior) during a baseline office visit reported fewer days lost from work, fewer health problems, lower functional limitations due to health, and improved health status at a follow-up visit. Patients seeking more conversational control had, at follow-up, lower blood glucose and blood pressure readings. Diabetes and hypertension management are, however, fairly clear-cut. Exactly how patients' interaction styles might affect treatment choices and outcomes in MS needs more study.
Physicians Dealing with Emotional Distress and Psychosocial Issues. Physicians can be trained to communicate more effectively with patients and, in particular, to deal with areas of emotional distress.166 Physicians who are interested in the psychosocial aspects of disease are much more likely than other physicians to raise these issues.116 Murray139 observed that MS physicians often feel uncomfortable addressing such issues as marital and personal problems; unfortunately, they also do not refer MS patients to other clinicians for these issues. Furthermore, communication must also address patients' interests outside the focus on treating the disease. Little attention has been paid to health promotion activities for persons with MS to enhance their overall well-being and quality of life, although a study of 629 women with MS found that health promotion activities significantly improved quality of life along the spectrum of disability.199
Paradoxically, many health care professionals are ill-equipped to instill hope because they have a depressing view of the disease, perhaps because they often work in hospitals where only the most ill and therapeutically resistant and com-
plicated patients are met. Most MS units now manage a large number of patients in an ambulatory setting, where most of the patients are getting along and the hospital-based medical student, physician, nurse, physical therapist, or social worker sees only the most difficult and most advanced problems in the wards. The availability of the new disease-modifying drugs has certainly increased the optimism of many physicians, but these drugs are still new. With time and experience, patients' and physicians' optimism could become more reserved or even greater. It is too early to know. Further, the new disease-modifying drugs have not been demonstrated to be helpful to the most ill patients.
The role of hope in patients with MS and the role of the physician in providing and supporting reasonable and appropriate hope have often been ignored. Hope is something physicians do not often discuss. Patients often called “hopeless” can be helped in many ways to cope with the ravages of the disease and to see the opportunity despite the limitations they face. We cannot yet cure, but we can help, and we can understand, and we can care.139
Physical activity has long been discouraged for people with MS, in part because they often experience fatigue and thermosensitivity, which causes their neurological symptoms to worsen during exposure to heat.155 Exercise can be particularly difficult for patients with limited mobility and poor balance. Yet many people with MS are probably less active than they could be if they were given proper guidance and access to appropriate facilities. One study compared the activity levels of a group of ambulatory MS patients with those of sedentary control subjects who did not have MS.143 (The median EDSS score of the MS patients was 3.0 [no assistance needed to walk], with a range of 1.5 [no disability] to 6.0 [assistance needed to walk]). When questionnaires asked them to recall their physical activity levels, the two groups reported similar activity levels. However, when their physical activity was measured over seven days using a motion detection device attached to their waists, the ambulatory MS patients were found to be significantly less active than the sedentary controls.
The benefits of exercise for people with MS are many and, conversely, lack of activity can contribute to fatigue and weakness and is a risk factor for many chronic diseases. Numerous studies have shown that physical activity in cancer patients enhances well-being in terms of physical and functional status, as well as psychological and emotional status (reviewed in 1999 by Courneya and Friedenreich34). Aerobic exercise training improves fitness, increases feelings of well-being, and reduces depression and anxiety in people with MS.155 Some aspects of impaired muscle function observed in MS are similar to those seen in healthy people after prolonged periods of inactivity, and deconditioning due to reduced physical activity likely contributes to the loss of muscle function in MS.97,143
Exercise training might thus reduce the loss of muscle function, as well as decreasing fatigue.97,143
Although exercise programs can benefit MS patients, they should be undertaken with care and attention from exercise physiologists, physical and occupational therapists, and physicians. Different types of exercises are best for people with different types of MS and different types of impairment (reviewed in 1999 by Petajan et al.155). For example, patients with motor deficits need an exercise program that takes their impairment into account. Pools are helpful because they are safe and allow for a wider variety of exercises. They also increase body cooling, thereby reducing the problem of thermosensitivity. In general, because increased body temperature can block conduction in damaged nerves, exercise programs should be designed to avoid overheating. An assessment of exercise history, including activities of daily living, and a fitness evaluation should be obtained prior to the initiation of an exercise program. Exercises that increase flexibility, strength, coordination, and balance are particularly helpful. Lack of motivation to exercise is likely, especially when a person does not feel well, and a supportive, proactive, and pleasant exercise environment is especially important. Although exercise training does not appear to influence relapse rates, exercise programs should sometimes be modified or temporarily discontinued during relapses.154
Weakness, fatigue, spasticity, and ataxia make exercise difficult for people with MS, and even brief exercise bouts can cause symptoms to appear or increase, but improvement in fitness not only can help offset these difficulties, it can reduce fatigue and depression and improve quality-of-life.155
All sorts of diets have been proposed to be of benefit in MS: low-saturated-fat with high-polyunsaturated-fat diets, megavitamins, liquid diets, and sucrose-free and gluten-free diets have all been advocated (see Appendix G).185 However, none of them have been rigorously tested under clinical conditions, and none have been supported by an underlying immunological basis.57,209
The rate of MS is generally higher in countries where diets are commonly high in animal fat, animal protein, and meat from nonmarine mammals and lower in countries where people eat more vegetables and fish (reviewed in 1997 by Lauer113).50,112 Yet despite the correlations, there are many confounding variables, and none of these dietary factors have been determined to be either risk factors or prognosticators for the disease.
Vitamin D has also been hypothesized to be related to MS.75 Since needs for vitamin D are met through exposure to sunlight in addition to dietary intake, it has been suggested that vitamin D levels might be a factor in the geographic distribution of MS.49 The evidence is circumstantial, and further investigation is needed. At least in EAE (experimental allergic encephalomyelitis) mice, exog-
enous vitamin D can prevent disease, supporting the idea that there might be some effect in humans, but this remains to be demonstrated.22
There are some data suggesting that large amounts of polyunsaturated omega3 fatty acids, which are prevalent in fish, reduce the severity of relapses and the progress of disability in early cases (but not long-term disability) and some indicating that reduced intake of animal fat and increased intake of vegetable fat and seafood are beneficial. However, most of these data come from uncontrolled, methodologically flawed studies.
There is, however, an emerging body of evidence to indicate that dietary antigens might trigger autoimmune responses in diseases other than MS.25,149,179,186 While this suggests a biological basis to predict an effect of dietary antigens in MS, as yet there are no conclusive data. If dietary influences are modest—which is highly likely considering the lack of clear-cut results to date—or if different foods interact in their effects on MS, obtaining such data would require an extensive clinical trial. In addition, since many patients are taking disease-modifying drugs, it would be statistically more difficult to detect the increased benefit of a modest improvement. Each treatment independently introduces some random variation, meaning that a clinical trial including both dietary changes and medication would have to be larger than a clinical trial with only one of these variables. While a healthy balanced diet is recommended for people with MS,171 the evidence supporting a specific benefit from such for MS remains inconclusive.113 Even if there were no specific benefit from dietary changes, if they resulted in generally improved health or well-being of people with MS, this would be reason alone to recommend them.
Use of Alternative Medicine
Alternative medicine refers to a mix of practices as disparate as herbal medicine, therapeutic touch, imagery, and homeopathy. What unites such treatments is that they are not widely taught in medical schools and are not generally available through physicians or hospitals. The term “complementary medicine” is preferred by some people, because it indicates that alternative therapies can be integrated with conventional therapies. For simplicity, the single term “alternative medicine” is used here to include all aspects of non-traditional medicine. Despite the preferred concept of complementary medicine, the use of alternative therapies is not fully integrated into medical treatment. Most patients who use alternative therapies do not inform their doctors. In 1997, only 38 percent of U.S. users of these therapies informed their physicians.46
The distinction between alternative and conventional medicine is fluid. Over time, an unconventional practice, such as self-hypnosis or yoga, might become conventional and many formerly conventional treatments such as blood-letting have fallen out of favor. One constant of alternative therapies, however, is that they are not subject to the same regulatory protections as conventional drugs.
They are not tested in clinical trials; manufacturer's claims of safety and efficacy are not reviewed by independent parties; and there is no product standardization. For example, in an analysis of ginseng products, even though different brands were labeled as containing equal amounts of the active ingredient, the actual amounts varied as much as tenfold; some brands contained no ginseng at all.5
Prevalence of Use
Despite its medically uncertain status, alternative medicine is an integral part of Western health care systems, if only because it is so widely used. A study of the U.S. population estimated that the number of visits to alternative therapy practitioners was 243 million more than the number of visits to all primary care physicians.46,52 Estimates vary among countries, but independent surveys of Australia and of North American and European countries generally report that more than 40 percent of the general population in the countries surveyed use alternative medicine.46,121,174 Many patients use more than one therapy at a time.136,218 Prevalence of use varies among the population. Twenty-five to 49 year-olds are more likely to use alternative medicine than people who are younger or older, and African-Americans are less likely than other ethnic groups to use alternative medicine.47 Different surveys report different age groups as those who use alternative therapies most extensively.46,134 This inconsistency might reflect international differences, population differences in health status, or differences in the way alternative therapies were described in survey questions.
Use of alternative therapies is not only widespread among healthy people; once people are diagnosed with a serious illness, their use is likely to increase.136,163,174,218 For example, in a study of 480 women with newly diagnosed breast cancer, 28 percent of them began to use alternative therapies.20 On the bright side, and unlike the general population, most of these women informed their doctors.
People with chronic illness consult alternative care providers about three times more often than people without chronic illness,7,134,163 and those with disabilities are 50 percent more likely than the general population to use alternative therapies.102 Compared to people without disabilities, those with disabilities seek alternative therapies significantly more often for treatment of pain, depression, and anxiety.7,102
Not surprisingly, the use of alternative therapies is more widespread among MS patients than among the general population. Although estimates vary, probably three-quarters use an alternative therapy, often more than one.52,141,177,191,232 (Note that some studies report visits to alternative therapy providers, whereas others report the use of alternative therapies. In general, the number of people who visit alternative therapy providers is about half that of those who use alternative therapies, many of whom do so without consulting an alternative therapy provider.)
Because MS is a long-term disease for which conventional medicine has no cure or completely effective treatment, one might reasonably ask, “Why not try alternative therapies?” Even though most herbal remedies have not been proven effective, most are probably not dangerous (reviewed in Angell and Kassirer, 1998),5 but there are other concerns. Because MS symptoms typically wax and wane, false positives are particularly likely. For instance, when a symptom is relieved after some therapeutic intervention, many patients are convinced that the intervention was responsible, even when they know that MS symptoms can wane with no treatment at all. In addition to the illusion of benefit when symptoms spontaneously subside, the placebo response can give patients the illusion that their sense of improvement is due to the specific treatment they tried, when in fact, the placebo effect could have arisen from any treatment. If placebo effects and illusions of benefit have no impact on the disease, they are harmless, but if they encourage the patient to forgo effective treatments, then these effects can be medically harmful. In addition to illusory effects, there are also medical concerns about using unregulated products whose contents are unverified. Finally, MS patients should be particularly cautious about substances that are relatively benign in people without immune disorders but might have negative impacts on the immune responses of MS patients.
The widespread use of alternative therapies has implications for both treatment and research in MS. There are endless varieties of alternative therapies, and it is important to know which of these are used most often and most heavily by MS patients. The use of alternative therapies should be taken into account in all aspects of treatment and assessment—from prescription of symptomatic treatments, to research on health status and quality of life, to identifying the most pressing health information needs of people with MS. Proven or not, alternative therapies are a pervasive dimension of health care among people with MS.
Specific Alternative Therapies
Of all forms of alternative treatment, herbal medicine is the most common (reference 15 in Angell and Kassirer5). Appendix G offers a critique of more than 50 alternative therapies that have been claimed to be beneficial for treating MS. Approaches not included there are described below.
Marijuana. Many MS patients, as well as patients with spinal cord injury, smoke marijuana to relieve spasticity. Controlled studies of the effects of marijuana or THC (tetrahydrocannabinol), the primary active ingredient in marijuana, have generally been inconclusive in MS patients, but none of those studies have included more than 13 MS patients (reviewed in 1999 by the IOM).89 These studies neither disprove nor support claims of the therapeutic value of marijuana's active ingredients. Many MS patients are convinced that marijuana relieves their spasticity and pain. One survey reported that 97 percent of MS patients reported
using marijuana to relieve one or more of their MS symptoms, but it is important to note that the target population of this survey was marijuana users.32 The degree to which these results apply to the general population of people with MS is impossible to know. Further, marijuana and THC can reduce both anxiety and pain, and this might reduce the discomfort associated with spasticity or tremor, although with no direct effect on spasticity. Experiments in EAE mice indicate that THC and related compounds can reduce spasticity and tremor.9 As of this writing, clinical trials testing the effects of marijuana extracts on MS patients are being conducted in Britain. The results of these trials should help clarify the effects of marijuana and its constituent compounds on MS symptoms.
Traditional Chinese Medicine. A variety of approaches—primarily herbal treatments, but also acupuncture—are used in traditional Chinese medicine to treat symptoms of MS, but their effectiveness has not been tested in rigorous clinical trials, and their descriptions are limited to Chinese-language publications.234 There are reports that Chinese herbal treatments can reduce the neuropathology associated with EAE in guinea pigs, but these reports have to be validated through experiments published in journals that are more widely accessible for critical review.
Hypnosis. Hypnosis has been proposed as an approach to improve muscular control in MS patients, but at present, the committee is aware of only scattered case reports of improvement after hypnosis.201 As with every individual case of improvement after some experimental treatment, there is a strong possibility that improvement might be a placebo effect or might have occurred in the absence of any treatment.
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