Long-Term Conditions: Diabetes and ME/CFS
Illness Experience and Health Care
’I have high blood sugars, and Type 2 diabetes is not going to kill me. But I just have to eat right, and exercise, and lose weight, and watch what I eat, and I will be fine for the rest of my life.
No one chooses to have ME — everything changed when I became ill.’
Tom Kindlon (2015)
In this chapter, we review two long-term conditions that are diagnosed in increasing numbers of patients: diabetes and ME/CFS. Neither can be cured using currently available treatments and both require an approach that utilizes the biopsychosocial model. The two conditions can often cause distress, with major reductions in quality of life, and involve significant care commitments from informal, family caregivers. We review each condition in turn, including the causes, the risk factors, the experience of living with the condition, interventions to ameliorate the symptoms and informal carers’ experiences.
Long-term or chronic illnesses have replaced acute illnesses as the predominant disease pattern. Greatly improved longevity and higher cancer survival rates have meant an increased prevalence of cancer, coronary heart disease, AIDS, diabetes and myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS). We reviewed the first three of these conditions in Chapters 21—23. In this chapter, we review the remaining two.
As life expectancy of the human population increases, so does the prevalence of diseases of older age. Long-term illness typically involves restrictions on activities of daily living, and increases in pain, fatigue, depression and anxiety. Patients need to cope daily with a changing array of ongoing symptoms, the life threat involved and the requirements of treatment. Maintaining effective relationships with health-service personnel, family and friends requires many adaptations and there is a need to continuously make adjustments. One of the greatest sources of worry is uncertainty around prognosis.
If one needs a litmus test of the current level of acceptance of the biopsychosocial model (BSM), then the manner in which long-term conditions without medical cures are perceived and managed provides a good indication. Neither diabetes mellitus nor ME/CFS can be cured using available treatments within biomedicine and both require an approach based on the biopsychosocial model. Diabetes has known causes but no known cure. ME/CFS has no known causes and no known cure. Unsurprisingly, ME/CFS receives little attention in the medical curriculum. A study by Stenhoff et al. (2015) found that medical students acquired their knowledge and attitudes from mainly informal sources and expressed difficulty understanding ME/CFS within the biomedical framework. ME/CFS patients were even perceived by some medical students as ’time-wasters’. Teaching about unexplained medical symptoms needs to be integrated into the medical curriculum, but to date this has not been happening. Unfortunately, lack of knowledge among physicians about ME/CFS and other long-term conditions increases the already high levels of distress and uncertainty for these patients and their families.
In the USA in 2012, about half of all adults — 117 million people — had one or more chronic health conditions. One in four adults had two or more chronic health conditions (Ward et al., 2014). Seven of the top ten causes of death in 2010 were chronic diseases. Two of these chronic diseases — cancer and heart disease — together accounted for nearly 48% of all deaths (CDC, 2013a) (see Chapters 21 and 22).
Obesity is a serious health concern, as we have seen earlier in this book. During 2009—2010 more than one-third of adults, or about 78 million people, were obese (defined as body mass index [BMI] ≥30 kg/m2). Nearly one in five youths aged 2—19 years was obese (BMI ≥95th percentile) (CDC, 2013b) (see Chapter 10).
Arthritis is the most common cause of disability (Hootman et al., 2009). Of the 53 million adults with a diagnosis of arthritis, more than 22 million say they have trouble with their usual activities because of it (Barbour et al., 2013).
Diabetes is the leading cause of kidney failure, lower-limb amputations other than those caused by injury, and new cases of blindness among adults (CDC, 2013c).
Chronic illnesses often strike in middle and older age and, while they can prove fatal, most people with a chronic illness live for many years with the condition. Genetic factors and diet-related behaviours are two primary determinants of diabetes, but the causes of ME/CFS remain a complete mystery. The focus of this chapter will be on the psychological aspects of disease management and treatments based on the BPSM. Management of fatal or chronic diseases is a principal feature in the lives of 10—15% of the population. The primary health care system is of great importance in long-term conditions. Patient-centredness has been strongly emphasized in offering care to patients with chronic conditions. Further research is needed on developing applications of the BPSM that are acceptable to patients.
The Chronic Care Model (CCM) was proposed as a possible solution to the rising prevalence of chronic diseases and has been widely advocated in order to improve quality of care for the chronically ill (Wagner et al. 2001). In the CCM, the health care system is viewed as one part of a larger community and health care practice or ’health organization’:
Effective self-management support and links to patient-oriented community resources help to activate and inform patients and families to better cope with the challenges of living with and treating chronic illness. Traditional patient education emphasized knowledge acquisition and didactic counselling. Mounting evidence indicates that while such interventions increased knowledge, they were unsuccessful in changing behaviour or improving disease control and other outcomes. More recent theoretical and empirical research has shifted the focus from patients’ knowledge of the disease and its treatment to their confidence and skills in managing their condition. The interventions that have emanated from this research reinforce the patient’s (and family’s) crucial role in managing the condition, help patients to set limited goals for improving their management of their illness, identify barriers to reaching their goals, and develop a plan to overcome the barriers. (Wagner et al., 2001: 63)
CCM includes formal health care combined with informal self-care and care by family members or friends. The latter occurs almost invisibly with little recognition or financial support from society at large. In all life-threatening conditions, family members can experience significant emotional, economic and social challenges, as can the person with the illness. Caregiving is often seen as ’women’s work’. Research indicates that around 3.1% of females provide informal care compared to around 1.6% of males (Mathiowetz and Oliker, 2005). More than 25 million family members provide informal care to disabled or ill family members in the USA (Levine, 2000). More than half of American women will care for a sick or disabled family member at some point during their adult lives (Moen et al., 1994). The economic value of informal care is estimated at $196 billion (in 1997 dollars), which is equivalent to 18% of the total national health care expenditure (Arno et al., 1999). One survey reported that women are twice as likely to care for someone for more than 20 hours a week and are 1.5 times more likely than men to perform more labour-intensive or intimate care tasks (Levine et al., 2000).
Lee et al. (2003) conducted the study in 54,412 women from the Nurses’ Health Study, a prospective cohort of female registered nurses residing in 11 US states to examine the association between caregiving and cardiovascular disease incidence. The women were aged 46 to 71 years and did not have diagnosed CHD, stroke or cancer at baseline (1992). They collected information on caregiving responsibilities in 1992 and CHD between baseline 1992 and 1996. During four years of follow-up, they documented 321 incident cases of CHD (231 non-fatal cases of myocardial infarction and 90 CHD deaths). In analyses controlling for age, smoking, exercise, alcohol intake, BMI, history of hypertension, diabetes and other covariates, caregiving for a disabled or ill spouse for more than nine hours per week was associated with increased risk of CHD (risk ratio, 1.82; 95% confidence interval, 1.08—3.05). However, caregiving for disabled or ill parents or disabled or ill others was not significantly associated with increased risks of CHD. From the results of Lee et al.’s study, it would appear that informal caregiving for a disabled or ill spouse is a significant risk for CHD. We discuss the psychological aspects of informal caregiving in more depth in the following sections.
The role of homeostasis in the maintenance of health and well-being has been a significant theme in our review of health psychology. Diabetes mellitus is a condition caused by a breakdown of homeostasis, in this case, at a physiological level. In Type 1 diabetes mellitus blood glucose homeostasis ceases to function because the beta cells of the pancreatic islets are destroyed. In Type 2 diabetes mellitus the pancreas loses its ability to secrete enough insulin in response to meals or to control the body’s glucose level. The condition is fatal if left untreated.
What is Diabetes?1
1. This primer text on diabetes is extracted from the US National Diabetes Education Program ’Diabetes Primer for School Personnel’ (2017), which is in the public domain.
Diabetes mellitus is a chronic disease in which blood glucose (sugar) levels are above normal (hyperglycaemia). People with diabetes have problems converting food to energy. After a meal, food is broken down into a sugar called glucose, which is carried by the blood to cells throughout the body. Insulin, a hormone made in the pancreas, allows glucose to enter the cells of the body where it is used for energy. People develop diabetes because the pancreas produces little or no insulin or because the cells in the muscles, liver and fat do not use insulin properly. As a result, the glucose builds up in the blood, is transported into the urine and passes out of the body. Thus, the body loses its main source of fuel even though the blood contains large amounts of glucose. When insulin is no longer made, it must be obtained from another source —insulin injections or an insulin pump. When the body does not use insulin properly, people with diabetes may take insulin or other glucose-lowering medications. Neither insulin nor other medications, however, are cures for diabetes; they only help to control the disease.
Figure 24.1 Main symptoms of diabetes
Source: Mikael Häggström (2009). Public domain
Taking care of diabetes is important. Over the years, ongoing high blood glucose, also called hyperglycaemia, can lead to serious health problems. If not managed effectively, diabetes can affect the blood vessels, eyes, kidneys, nerves, gums and teeth, making it the leading cause of adult blindness, kidney failure and non-traumatic lower limb amputations. Diabetes also increases a person’s risk for heart disease and stroke. Some of these problems can occur in teens and young adults who develop diabetes during childhood. The good news is that research shows these problems can be greatly reduced, delayed or possibly prevented through intensive treatment that keeps blood glucose levels near normal. There are three main types of diabetes.
Type 1 diabetes mellitus (T1DM), formerly called juvenile diabetes, is a disease of the immune system, the body’s system for fighting infection. In people with Type 1 diabetes, the immune system attacks the beta cells (the insulin-producing cells of the pancreas) and destroys them. Because the pancreas can no longer produce insulin, people with Type 1 diabetes must take insulin daily to live. Type 1 diabetes can occur at any age, but onset of the disease occurs most often in children and young adults. Most cases of diabetes in children under age 10 are Type 1. In adults, Type 1 diabetes accounts for 5—10% of all cases of diagnosed diabetes.
The symptoms of Type 1 diabetes are due to an increase in the level of glucose in the blood and include increased thirst and urination, weight loss, blurred vision and feeling tired all the time. These symptoms may be mistaken for severe flu or another rapid-onset illness. If not diagnosed and treated with insulin, the child with Type 1 diabetes can lapse into a life-threatening condition known as diabetic ketoacidosis or DKA. Signs of DKA include vomiting, sleepiness, fruity breath, difficulty breathing and, if untreated, coma and death.
Although scientists have made much progress in predicting who is at risk for Type 1 diabetes, they do not yet know what triggers the immune system’s attack on the pancreas’s beta cells. They believe that Type 1 diabetes is due to a combination of genetic and environmental factors that are beyond the individual’s control. Researchers are working to identify these factors and to stop the autoimmune process that leads to Type 1 diabetes.
Type 2 diabetes mellitus (T2DM), formerly called adult-onset diabetes, is the most common form of the disease. People can develop it at any age, even during childhood. A progressive disease, Type 2 diabetes usually begins with insulin resistance, a condition in which muscle, liver and fat cells do not use insulin properly. At first, the pancreas keeps up with the added demand by producing more insulin. Over time, however, the pancreas loses its ability to secrete enough insulin in response to meals or to even control the glucose level overnight or during periods of fasting. Managing Type 2 diabetes includes lifestyle changes such as making healthy food choices and getting regular physical activity. In addition, people with T2DM may take insulin and/or other glucose-lowering medications to control their diabetes. In the past, T2DM used to be found mainly in overweight or obese adults ages 40 or older. Now, as more children and adolescents have become overweight and inactive, T2DM is occurring in young people.
The symptoms of T2DM in children may be similar to those of Type 1 diabetes. A child or teen may feel very tired or thirsty and have to urinate often due to high blood glucose levels. Other symptoms include weight loss, blurred vision, frequent infections and slow-healing wounds. High blood pressure or elevated blood lipids (cholesterol) are associated with insulin resistance. In addition, physical signs of insulin resistance may appear, such as acanthosis nigricans, a condition in which the skin around the neck, armpits or groin looks dark, thick and velvety. Often, this condition is mistaken for poor hygiene. Some children or adolescents (and adults) with T2DM may have no recognized symptoms when they are diagnosed. For that reason, it is important for the parents/guardian to talk to their health care providers about screening children or teens who are at high risk for T2DM.
The risk factors of T2DM are being overweight, having a family member who has T2DM and being African American, Hispanic/Latino, American Indian, Alaska Native, Asian American or Pacific Islander, including Native Hawaiian. Other risk factors include having a mother who has had diabetes during her pregnancy (gestational diabetes), having high blood pressure, high cholesterol, abnormal lipid levels, polycystic ovary syndrome and being inactive.
For children and teens at risk, health care professionals can encourage, support and educate the entire family to make lifestyle changes that may delay — or prevent — the onset of Type 2 diabetes. Changes include making healthy food choices, reaching and maintaining a healthy weight and engaging in regular physical activity.
Gestational diabetes develops during pregnancy and is caused by the hormones of pregnancy. These hormones can cause insulin resistance or a shortage of insulin. Although gestational diabetes usually goes away after the baby is born, a woman who has had it is at increased risk for developing diabetes for the rest of her life. In addition, the offspring of that pregnancy is at increased risk for obesity and developing T2DM.
Living with Diabetes
’Being diagnosed with diabetes, or knowing someone who is diagnosed with the condition, may throw up many questions about how it fits into your daily life, from how it makes you feel, to managing diabetes at work, or while you are driving. ’The practicalities of living with diabetes can be stressful, but you needn’t put your life on hold’ (Diabetes UK, 2017). Qualitative studies have been carried out suggesting that this description is accurate. One Swedish study even suggests that diabetes is a ’marginal problem’: ’Diabetes in the shadow of daily life: factors that make diabetes a marginal problem’ is one of a relatively small number of studies offering the patient perspective of what it is like living with diabetes (Ågård et al., 2016). The sample consisted of 24 patients (15 male) with diabetes mellitus. They were recruited from a medical outpatient clinic in western Sweden, in an area with a high incidence of immigrants and low socio-economic status. One main interview question was formulated: ’Can you describe what living with diabetes means to you?’ Content analysis placed the responses into meaning units that were categorized into four main themes and eight sub-themes. The following sections discuss these themes using quotes from the study.
Theme 1: ’A Lifelong Follower but not a Real Problem’
Sub-theme 1A: ’No big deal’. The authors reported that respondents did not feel that diabetes was a severe disease but it was perceived as a minor issue, with practical problems such as needing an increased focus on food and medicines, but life in general went on as usual. Those who had had diabetes for a long time had incorporated diabetes management into the routines of their everyday life. Some felt gratitude for being in a country where they had access to insulin and care. ’For me, diabetes is only something I have and I will have it as long as I live. I don’t think so much about it. It is like having a cup of coffee in the morning … or going to the laundry’ (R2; Ågård et al., 2016).
Sub-theme 1B: ’Symptoms, not blood sugar levels, matter’. Another reason for not ascribing diabetes great importance was the lack of symptoms associated with high blood glucose levels. As a result, some respondents felt that they did not bother about complications at the moment, although they knew they could occur in the future. ’The health care unit tested my blood sugar level and once again I had more than 30. However, I had no symptoms of diabetes this time either’ (R16; Ågård et al., 2016).
Theme 2: ’A Trifle in Relation to the Daily Struggle with Difficulties’
Sub-theme 2A: ’Burdening circumstances’. Many said their perception of living with diabetes was dependent to a great extent on ’burdening circumstances and troubles in life’. One middle-aged man felt unable to manage his diabetes because a work project had failed. Another patient mentioned worries about the future when asked about living with diabetes. ’It’s difficult to live with diabetes because every day I get a new problem. I live alone. I can’t get anyone to help me at home. Yes, every time I go to sleep, I pray to God to end my life so that I don’t have to wake up. I don’t see any joy in life’ (R5; Ågård et al., 2016).
Sub-theme 2B: ’Less important compared with other diseases and symptoms’. Apart from diabetes, respondents could have a combination of other diseases. Medical conditions such as cardiac diseases, high blood pressure, inflammations, chronic obstructive pulmonary disease and depression tended to overshadow the importance of having diabetes. Moreover, symptoms (e.g., sleeping problems) and treatment (e.g., for tuberculosis or leukaemia) related to these diseases were perceived as barriers to the control of diabetes. ’Heart failure sets the limit for everything. In my opinion, having diabetes does not have a substantial impact on my capacity. The diabetes is merely something that goes along. It is nothing that really bothers me. I just have to check the values every day and take insulin on demand’ (R10; Ågård et al., 2016).
Theme 3: ’Something Out of One’s Own Control’
Sub-theme 3A: ’Diabetes as a predetermined condition’. Some felt that it had been predetermined for them to get diabetes or because they were a victim of circumstances such as stress, fate, a difficult childhood, conflicts, having lived under a death threat, war and homesickness. Diabetes was ’something evil’ that ’through no fault of their own, had affected them’. As a result, they felt little incentive to adhere to lifestyle advice. ’I’ve always had a poor view of myself because I had a miserable childhood. And, of course, it affects everything; well, that’s why I’m ill’ (R9; Ågård et al., 2016).
Sub-theme 3B: ’No use trying’. A sense of helplessness and lack of responsibility in coping with diabetes led to a sense of resignation. ’plants and leaves of olive trees and honey … treat diabetes because olive leaves are able to regulate sugar … this treats diabetes, hypertension and even high cholesterol … but the problem is that I don’t have anyone who can travel with me so I can bring the stuff home’ (R5; Ågård et al., 2016).
Theme 4: ’Not Worth Giving Up the Good Things in Life For’
Sub-theme 4A: ’Preference for a good life rather than living right’. The majority had received nutritional advice and knew what they should eat and drink. However, some reported not being willing to refrain from tasty food and alcohol to any great extent. The social and enjoyable dimensions of eating were more important than eating the right food. Some felt that their prescribed diet, apart from not being very tasty, made cooking more complicated and meals more boring. ’The diet and the food that I got at the hospital were like getting some kind of medicine. But, at home, you have a family and you want to hang out and you want to live a little, kids come and so on. You want to have a reasonably good life. So I cross the limits sometimes’ (R6; Ågård et al., 2016).
Sub-theme 4B: ’Reasons for not exercising’. Similarly, the majority were quite aware of the positive effects of physical activity, yet some took almost no exercise at all. They said that they were lazy, unwilling to exercise or felt that exercise was boring. It was more convenient to remain on the couch or sit in front of a computer. Other excuses for not exercising were: pain, fatigue, depression, sleep problems, lack of experience, financial problems and/or potentially harmful to the heart. These participants had decided not to change their diet, exercise more or live a highly regulated life. ’I try to go out for a walk now and then. We have also been training at the gym. That makes you fit. However, I guess we’re lazy. We spend lot of time in front of the computer’ (R17; Ågård et al., 2016).
The findings of the Ågård et al. study illustrate the value of qualitative research in providing insight into patients’ attitudes, health beliefs, reasons for adherence or non-adherence, and their ability or willingness to practise self-management skills and knowledge. Data from focus group discussions, together with other kinds of data, can be helpful in understanding patient experience.
Vermeire et al. (2003) reported a qualitative study of T2DM patients’ health beliefs in relation to their illness, their communication with caregivers and the problems encountered in adhering to treatment regimens. Seven focus groups were held for patients recruited by pharmacists in primary care in Flanders, Belgium. The groups focused on their experiences with their treatment in response to four questions: ’1. How did you experience the diagnosis of diabetes? In what ways did diabetes change your life? 2. Diabetes is a chronic illness treated by diet, oral medication or insulin. How are you treated? 3. Your doctor probably decided on your therapy. How do you feel about his/her decision? 4. Do you modify your treatment from time to time? How do you feel about this and do you tell anyone about your decision?’
Themes and sub-themes emerging from the focus group discussions included: poor knowledge; incomplete and conflicting information; uncertainty about physician’s compliance with diabetes management guidelines; little attention to patient’s health beliefs; little attention to patient’s opinion on medicine; paternalism; doctor’s fluctuating tolerance; follow-up inconsistency; patient’s fears about communicating adherence; self-regulation; keeping in touch with one’s own body; observation of body’s functioning; imperceptible evolution of the disease; imperceptible effects of treatment; little encouragement; complexity; insulin: a major crisis; adherence equals losing or gaining control of one’s own body.
A systematic review of qualitative research studies was carried out by Wilkinson et al. (2014). In 37 studies, the main issues impacting on individual self-care were ’communication’, ’education’, ’personal factors’, ’provider issues’ and ’support’. Multiple barriers were suggested as influencing the day-to-day management of diabetes, including poor communication with health care providers. The findings suggested the need for an education programme that allows for incremental knowledge gain and also experiential and vicarious learning and the provision of culturally sensitive care. The authors concluded that people living with diabetes face many issues in their day-to-day management of the disease, compounded by vulnerability to wider situational, cultural and social issues. Self-care ability is a ’dynamic, evolutionary process’, it was concluded, ’that varies from person to person and involves moving from a disease focused existence to maximising life’ (Wilkinson et al., 2014: 111).
As we have seen in earlier chapters, health-related stigma has been strongly associated with both obesity and HIV/AIDS, but it has been less studied with diabetes. Browne et al. (2013) explored the social experiences of Australian adults living with T2DM, with a particular focus on the perception and experience of diabetes-related stigma. They audiorecorded semi-structured interviews, which they subjected to thematic analysis. This study took place in non-clinical settings in metropolitan and regional areas in Victoria. A total of 25 adults with T2DM participated (12 women, median age 61 years, median diabetes duration five years); 21 participants (84%) indicated that they believed T2DM was stigmatized, or reported evidence of stigmatization. Specific themes were feeling blamed by others for causing their own condition, experiencing negative stereotyping, being discriminated against or having restricted opportunities in life. Sources of stigma included the media, health care professionals, friends, family and colleagues. Themes relating to the consequences of this stigma were evident, including participants’ unwillingness to disclose their condition to others and psychological distress. Participants believed that people with T1DM do not experience similar stigmatization.
The long-term complications of diabetes can be severe as a visit to the general surgical wards of any metropolitan hospital will prove. The complications include cardiovascular disease, retinopathy (a disease of the retina which results in impairment or loss of vision), nephropathy, which is damage to the kidneys, and diabetic neuropathy, where nerve fibres are damaged as a result of high blood sugar, most often in the legs and feet. The latter can result in the amputation of one, two or possibly more limbs. People living with diabetes can take steps to prevent the development of long-term complications (see Box 24.1).
Box 24.1 Preventing Complications
There are eight steps that help to prevent complications:
1. Regular check-ups with a health care team — at least once a year.
2. Long-term checks on the diabetes, such as an HbA1c test.
3. Testing blood glucose levels at home regularly, and recording the results, aiming for between 4mmol/l and 8mmol/l* before meals and less than 10mmol/l two hours after meals for most of the time.
4. Achieving and maintaining a healthy body weight.
5. Keeping blood pressure and blood fats (e.g., cholesterol) under control.
6. Eating a healthy balanced diet.
7. Increasing physical activity.
8. Not smoking.
Source: Diabetes UK (2017)
An important trial carried out in the USA, the ’Diabetes Control and Complications Trial’ (DCCT), assigned patients to conventional or intensive therapy from 1983 to 1993. Participants were randomly assigned to receive either the experimental or standard therapy. The experimental therapy involved the use of an intensive insulin regimen designed to maintain near-normal glycaemic levels in the absence of severe hypoglycaemia. The standard treatment was designed to maintain patients free of clinical symptoms related to hyper- or hypoglycaemia while receiving up to two insulin injections daily. Since 1993 the DCCT has been observational, and intensive therapy was recommended for all patients. Clinical treatment goals of T1DM were changed since the Diabetes Control and Complications Trial demonstrated reduced long-term complications with intensive diabetes therapy.
Nathan et al. (2009) provided an analysis of the incidence of long-term complications. Their study was performed after 30 years of observing the DCCT cohort of 1,441 patients and a subset of another relevant cohort (EDC) of 161 patients. The cumulative incidences of proliferative retinopathy, nephropathy and cardiovascular disease were 50%, 25% and 14%, respectively, in the DCCT conventional treatment group, and 47%, 17% and 14%, respectively, in the EDC cohort. However, the DCCT intensive therapy group had substantially lower cumulative incidences (21%, 9% and 9%) and fewer than 1% became blind, required kidney replacement, or had an amputation because of diabetes during that time. These results are important in showing that the frequencies of serious complications in patients with T1DM, especially when treated intensively, are lower than that reported historically.
Interventions for Diabetes Patients
Primary Health Care
The Chronic Care Model (CCM) has been influential in the organization of primary health care and continuity of care between patients, families and professionals. The American Diabetes Association’s (ADA) Standards of Medical Care in Diabetes is updated annually. These ’Standards’ provide the most current evidence-based recommendations for diagnosing and treating adults and children with all forms of diabetes. Recommendations in the 2015 ADA Standards state that: ’Patient-centered communication that incorporates patient preferences, assesses literacy and numeracy, and addresses cultural barriers to care should be used. Care should be aligned with components of the Chronic Care Model (CCM) to ensure productive interactions between a prepared proactive practice team and an informed activated patient’ (Chronic Care Model, 2015: 97).
In making lifestyle changes that are crucial to self-care, ’an informed activated patient’s’ autonomous motivation to self-regulate health-relevant behaviours is important. Koponen et al. (2017) investigated six dimensions of primary health care that are strongly associated with autonomous motivation (self-regulation) for effective diabetes self-management. The study comprised 2,866 patients with T2DM. The six characteristics were access to care, continuity of care, diabetes counselling, trust, patient-centred care and ’autonomy support’ from one’s physician. Koponen et al.’s study showed that of these six quality dimensions of primary health care, autonomy support from one’s physician was most strongly associated with self-regulation for effective diabetes self-management. However, the authors suggest that overall support received from friends, family members, other patients with diabetes and health care professionals may even play a greater role.
Baptista et al. (2016) systematically reviewed studies that evaluated different elements of the CCM in patients with Type 2 diabetes mellitus. They assessed the influence of the CCM on different clinical outcomes. Studies were eligible for inclusion if they compared usual care with interventions that used one or more elements of the CCM. Six out of the 12 studies included in the review showed evidence of the effectiveness of the CCM for diabetes management in primary care as well as significant improvements in clinical outcomes. In six other studies, no improvements regarding clinical outcomes were observed with CCM. The authors concluded that greater benefits could be obtained through combining all elements of CCM.
Some risk factors for T2DM are potentially reversible, e.g., elevated plasma glucose concentrations in the fasting state and after an oral glucose load, overweight and a sedentary lifestyle. One group of investigators hypothesized that modifying these factors with a lifestyle-intervention programme or the administration of metformin would prevent or delay the development of diabetes. The Diabetes Prevention Program Research Group (2002) randomly assigned 3,234 non-diabetic persons with elevated fasting and post-load plasma glucose concentrations to placebo, metformin (850 mg twice daily) or a lifestyle-modification programme with the goals of at least a 7% weight loss and at least 150 minutes of physical activity per week. Metformin hydrochloride lowers blood sugar levels by increasing the sensitivity of muscle cells to insulin, reducing the amount of sugar produced in the liver and delaying the absorption of sugar from the intestines after eating so that there is less of a spike in blood sugar levels after meals. The lifestyle intervention consisted of a 16-lesson curriculum covering diet, exercise and behaviour modification to help the participants achieve the set goals. The participants averaged 51 years of age, and had a mean BMI of 34.0. Two-thirds of the sample were women, and 45% were members of minority groups. The average follow-up was 2.8 years.
The incidence of diabetes was 11.0, 7.8 and 4.8 cases per 100 person-years in the placebo, metformin and lifestyle groups, respectively. The lifestyle intervention reduced the incidence by 58% (95% confidence interval, 48—66%) and metformin by 31% (95% confidence interval, 17—43%), as compared with placebo. The lifestyle intervention was significantly more effective than metformin. The results showed that to prevent one case of diabetes during a period of three years, 6.9 persons would have to participate in the lifestyle-intervention programme and 13.9 would have to receive metformin. While lifestyle changes and treatment with metformin both reduced the incidence of diabetes in persons at high risk, the lifestyle intervention was more effective than metformin.
Diabetes Self-Management Education
Diabetes self-management education (DSME) involves the process of facilitating knowledge, skill and ability necessary for diabetes self-care. Diabetes self-management support (DSMS) is the support that is required for implementing and sustaining coping skills and behaviours needed to self-manage on an ongoing basis. The American Diabetes Association and many other diabetes associations worldwide recommend that all individuals with diabetes receive DSME/S at diagnosis and as needed on an ongoing basis thereafter. The goals of self-management education are to optimize metabolic control, prevent acute and chronic complications and optimize quality of life, while keeping costs acceptable.
Recent studies estimate that of those newly diagnosed with diabetes, fewer than 7% of individuals have private insurance and fewer than 5% of those covered by Medicare participate in DSME. Thus, although the systematic review by Norris et al. (2001) indicated that DSME resulted in clinical improvement, DSME remains under-utilized in diabetes care (Chrvala et al., 2016).
Yet research has revealed significant knowledge and skill gaps in 50—80% among patients with diabetes (Clement, 1995), and ideal glycaemic control (HbA1c <7.0%) is achieved in fewer than half of people with T2DM (Harris et al., 1999). Educational techniques used in DSME have shifted away from purely didactic presentations to interventions involving patient ’empowerment’ with participation and collaboration (Norris et al., 2001).
Norris et al. (2001) systematically reviewed the effectiveness of self-management training in T2DM. They searched for studies between 1980 and 1999, which were original articles reporting the results of RCTs on the effectiveness of self-management training in people with T2DM. A total of 72 studies were identified. Self-management training was observed to have positive effects on knowledge, frequency and accuracy of self-monitoring of blood glucose, self-reported dietary habits, and glycaemic control in studies with short follow-up at six months. Effects of interventions on lipids, physical activity, weight and blood pressure were less definite. With longer follow-up, interventions using regular reinforcement were sometimes effective in improving glycaemic control. The results suggested that interventions involving patient engagement may be more effective than didactic interventions in improving glycaemic control, weight and lipid profiles. Performance, selection, attrition and detection bias were common in the studies reviewed, and external generalizability was often limited. The evidence from the Norris et al. review supports the effectiveness of self-management training in T2DM, particularly in the short term. Larger studies with long-term follow up are needed to confirm the value of DSME.
A new modality for DSME is the smartphone. El-Gayar et al. (2013) reviewed whether diabetes smartphone apps are capable of helping patients with Type 1 or Type 2 diabetes mellitus to self-manage their condition, and identified issues for large-scale adoption. The review covered apps available at the Apple App Store and articles published from January 1995 to August 2012. Findings suggested that apps can support self-management tasks such as physical exercise, insulin dosage or medication, blood glucose testing and diet. Other ancillary tasks included decision support, notification/alert, tagging of input data and integration with social media. The analysis suggested that app usage can improve attitudes favourable to diabetes self-management. However, relatively few people with T2DM are currently making use of the technology due to a perceived lack of additional benefits and ease of use (Scheibe et al., 2015). Improved usability, perceived usefulness and adoption of the technology are necessary for the future adoption of this technology.
Depression is common among patients with diabetes, who have a higher risk of diabetes-related complications such as diabetic retinopathy, nephropathy, neuropathy and macrovascular complications. Ali et al. (2006) conducted a systematic literature review consisting of ten randomized controlled trials and 51,331 patients to estimate the prevalence of clinical depression in adults with Type 2 diabetes compared with those without Type 2 diabetes. The prevalence of depression in Type 2 diabetes patients was 9% higher than in those without diabetes.
More recently, Bogle (2017) carried out a systematic review to determine whether CBT is effective in reducing depressive symptoms and improving glycaemic control among depressed diabetic patients. The results revealed diverse applications of CBT, in which CBT had a positive impact on depressive symptoms, with three studies also finding an improvement in HbA1c, and one also demonstrated improved self-efficacy and self-concept related to successful diabetes management. There is a need for controlled studies with larger sample sizes and follow-up periods of more than six months.
Caring for a Person with Diabetes
Diabetes places a substantial care burden on elderly individuals, their families and society, both through the resulting disability and the substantial periods of time that informal caregivers spend helping to address the associated functional limitations.
Langa et al. (2002) obtained nationally representative estimates of the time, and associated cost, of informal caregiving provided to elderly individuals with diabetes, and determined the diabetes complications that contributed to the need for informal care. Data came from a nationally representative survey of people aged 70 or older (n = 7,443). Those without diabetes received an average of 6.1 hours per week of informal care, while those with diabetes but taking no medication received 10.5 hours and those with diabetes who were taking oral medication received 10.1 hours, and those taking insulin received 14.4 hours of care. Disabilities from heart disease, stroke and visual impairment were predictors of diabetes-related informal care. The total cost of informal caregiving for elderly individuals with diabetes in the USA was estimated to be between $3 and $6 billion per year, similar to previous estimates of the annual paid long-term care costs attributable to diabetes. That figure will have increased to at least $4—8 billion in 2018 prices.
The toll on parents of children with T1DM has been described as ’relentless’. Night-time caregiving with nocturnal blood glucose monitoring (NBGM) tends to increase parents’ anxiety and stress. Monaghan et al. (2009) invited 71 parents of children with T1DM ages 2—6 to complete questionnaires designed to assess the frequency of NBGM, illness characteristics and health outcomes, and parental concern. Approximately one-third reported regularly monitoring their child’s blood glucose level after their child was asleep, suggesting that parents’ nighttime caregiving practices and sleep loss needed clinical attention.
Continuous glucose monitoring (CGM) involves a subcutaneously implanted enzyme electrode that senses interstitial fluid as a proxy for blood glucose levels. The relationship between glucose concentrations in interstitial fluid and blood has generated great interest due to the possibility of gaining up to 288 glucose level readings a day without having to do finger pricks. Pickup et al. (2015) analysed narratives about experiences of using CGM in people with T1DM and their caregivers from an online survey. Questions included duration of CGM, frequency of sensor wear, funding, and a free narrative about experiences or views about CGM.
Pickup et al. identified four themes: (1) metabolic control, (2) living with CGM (work and school, sleep, exercise, nutrition, frequency of self-monitoring of blood glucose [SMBG]), (3) psychological issues and patient/caregiver attitudes, and (4) barriers to CGM use (technical issues, financial issues, attitudes of health care professionals towards CGM). The narratives suggested that the experiences were overwhelmingly positive, with improved glycaemic control, diet and exercise management, quality of life, and physical and psychological well-being, as well as reduced frequency of SMBG. The authors concluded that patients and caregivers both view CGM as a valuable addition to caring for many people with T1DM.
Trief et al. (2017) examined associations between relationship status, relationship factors and outcomes in adults with T1DM; 1,660 participants completed a survey measuring relationship satisfaction and perceived partner support style (active engagement, protective buffering, over-protection). Differences in glycaemic control and adherence for married or partnered couples versus those who were not were not significant. However, having higher relationship satisfaction, and having an engaged, not over-protective, partner was associated with better glycaemic control and self-care. Trief et al. concluded that helping partners support patients and avoiding over-protection may enhance relationship and patient outcomes for adults with T1DM.
What is ME/CFS?
Myalgic encephalomyelitis (ME) or chronic fatigue syndrome (CFS) is one of the most controversial and hotly debated topics in the history of medicine. Even the correct label for the condition is under dispute, as is the nature of the condition itself and everything else to do with it. The condition has also been named ’Systematic Exertion Intolerance Disease’ (SEID), ’Post-Viral Fatigue Syndrome’ (PVFS) and ’Chronic Fatigue Immune Dysfunction Syndrome’ (CFIDS). In this chapter, we use the label ’ME/CFS’. This condition has been characterized by different authorities and organizations in many diverse ways. ME/CFS is a label for a condition that involves prolonged muscle weakness after minor exertion (accompanied by muscle pain) and neurological symptoms indicative of cerebral dysfunction, such as sleep disturbances, headaches and cognitive problems.
The severity of ME/CFS varies from patient to patient, with some people able to maintain fairly active lives. For many patients, however, ME/CFS places significant limitations on their work, school and family activities, and the pattern of symptoms can be unpredictable. According to the International Consensus Criteria (ICC) for ME/CFS, actual fatigue is not a requirement for diagnosis (Carruthers et al., 2011). The ICC report stated:
The label ’chronic fatigue syndrome’ (CFS) has persisted for many years because of the lack of knowledge of the aetiological agents and the disease process. In view of more recent research and clinical experience that strongly point to widespread inflammation and multisystemic neuropathology, it is more appropriate and correct to use the term ’myalgic encephalomyelitis’ (ME) because it indicates an underlying pathophysiology. (Carruthers et al., 2011: 327)
Chronic fatigue can accompany many long-term illnesses. Patients with illnesses such as depression and cancer may be misdiagnosed with ME/CFS. Case definitions for ME/CFS vary between the broad Oxford criteria (Sharpe et al., 1991) and the more specific Fukuda criteria (Fukuda et al., 1994). According to the Centers for Disease Control and Prevention (2017b: 312):
Chronic fatigue syndrome, or CFS, is a debilitating and complex disorder characterized by profound fatigue that is not improved by bed rest and that may be worsened by physical or mental activity. Symptoms affect several body systems and may include weakness, muscle pain, impaired memory and/or mental concentration, and insomnia, which can result in reduced participation in daily activities.
CFS can affect adults, children and adolescents. Between 0.2% and 2.3% of children or adolescents suffer from CFS. In children, particularly adolescents, CFS is more likely to develop after an acute flu-like or mononucleosis-like illness, but gradual onset of illness may occur (CDC, 2017b).
Unfortunately, many patients with ME/CFS find the medical health care they receive insufficient. Patients’ negative reactions might be explained by the types of intervention offered by health care services to patients with ME/CFS (see Box 24.2).
Box 24.2 ’No One Chooses to have ME — Everything Changed When I Became Ill’
I first became ill at 16. Before becoming ill, I lived a very active life. I participated in whatever sport was going — soccer, table tennis, life-saving, tennis, cricket and cross-country running. I was also naturally academic and managed to get good results while still enjoying lots of extra-curricular activities.
All that changed when I became ill with ME.
I can remember how it happened. I developed an infection the day before going on a school trip. I decided to still go. We went to an adventure centre in the west of Ireland canoeing, hill-walking, abseiling and orienteering. It was February and it was cold and rainy throughout the trip — not the place to be when you’re unwell. I was ill for some days after I came home.
I was never the same after that. Anytime I tried to play sports I felt ill afterwards. I found I was struggling to get my homework done.
I struggled on at school and then university for another four years living a hermit-like existence. I had some natural ability (I came sixth in Ireland in the Irish National Maths Contest in sixth year in school) which helped, but found I had virtually no energy for a rounded life.
I went to an orthopaedic surgeon who recommended exercise, so I started going to a swimming pool three or four times a week. I kept appearing to strain muscles so I’d have to try different strokes like back stroke with only one arm. I had over 100 physiotherapy appointments trying to deal with all the muscle and tendon strains I appeared to have. I was desperate to get back to my previous life. I thought that all I needed to do was get fit and my life could go back to normal. Things took a turn for the worse in college. A couple of hours after an exam, my throat swelled up and I could barely swallow. I developed flu-like symptoms and a high temperature.
Looking back, I’d recognise that as a relapse from the mental exertion of the exam. When I tried to go back to exercising a few weeks later, my reaction was much more severe. Even a few lengths of slow swimming led to high levels of pain the next day. I switched to walking, but developed shin splints. I developed tennis elbow in my right arm and tendonitis in my right hand. My body was falling apart. I started trying to write with my left hand but soon developed tendonitis in that. I took a year out hoping I could get back to functioning.
Unfortunately, it was not to be. I developed an almost constant sore throat. By May, I developed pain in my lower stomach, and diarrhoea.
I was referred to a gastroenterologist who after a colonoscopy diagnosed irritable bowel syndrome. This is quite common in ME but I just thought I was very unlucky to have lots of separate health issues.
Finally, by August, I started having difficulty walking short distances. I would get out of breath even from slow walking. My GP referred me to a consultant in the Blackrock Clinic, the late Prof Austin Darragh who sadly passed away earlier this month. After doing some blood tests, he diagnosed me with ME, or post-viral chronic fatigue syndrome, saying I was a classic case.
Source: Kindlon (2015). Reproduced by permission
Not a single, specific cause of ME/CFS has yet been identified. It could well be that ME/CFS has multiple causes or there could be one single cause yet to be identified. The problem is that nobody knows. Conditions that have been suggested as causes or triggers of ME/CFS include infections, immune disorders, stress, trauma and toxins. The controversies around the definition, the cause and the treatment have been hugely emotive and have involved patients, patient organizations, health care and medical organizations, medical, psychiatric and psychology journals, government departments, insurance companies, and even legal tribunals in hotly debated disputes.
Living with ME/CFS
Difficulties with Diagnosis
Uncertainties around diagnosis is one of the major problems for patients, families and their medical advisers. For doctors, diagnosing ME/CFS is complicated because:
1. There’s no lab test or biomarker for ME/CFS.
2. Fatigue and other symptoms of CFS are common to many illnesses.
3. For some ME/CFS patients, it may not be obvious to doctors that they are ill.
4. The illness has a pattern of remission and relapse.
5. Symptoms vary from person to person in type, number and severity.
These factors have contributed to a low diagnosis rate. The CDC (2017b) suggests that of the 1—4 million Americans who have ME/CFS, less than 20% have been diagnosed. However, early diagnosis and treatment of ME/CFS increase the likelihood of improvement. The patient who thinks she/he may have ME/CFS may find it difficult to talk to their doctor or other health care professional about their condition.
ME/CFS can resemble many other illnesses, including mononucleosis, Lyme disease, lupus, multiple sclerosis, fibromyalgia, primary sleep disorders and major depressive disorder. Also, medication can cause side effects that mimic the symptoms of ME/CFS. People may mistakenly assume that they have ME/CFS when they have another illness. An ME/CFS diagnosis can be made only after other conditions have been excluded.
A major, substantive review of the qualitative literature on the lived experiences of ME/CFS patients was published by Anderson et al. (2012). Anderson et al. reviewed and synthesized findings across 34 qualitative studies of people living with ME/CFS. Their analyses included a multi-perspective (e.g., individual, physician, familial) examination of ME/CFS, as well as a comparative analysis of ME/CFS versus other chronic conditions. For patients, illness development influenced identity, reductions in functioning and coping. We base the following description of the living experience with ME/CFS on the findings of Anderson et al. (2012).
People with ME/CFS experience a change in identity throughout the course of their illness that can lead to a shift in body—mind relationships. They experience a disruption in and reconstruction of their identity and also a loss of confidence and self-esteem. Identity disruption is often related to transformations of family roles (e.g., children taking on additional responsibilities).
Fennell (1995) proposed a four-phase theory of ME/CFS illness experience that included periods of crisis, stabilization, resolution and integration. People with ME/CFS go through cycles of health and unhealthiness that can be transformative. Chronicity contributes to the cycle of being overwhelmed by and eventually learning to live with the illness. ME/CFS patients are made accountable for the cause of their illness due to the contested nature of the illness. Shifting accountability from the medical system to the individual patients is one way in which the societal response blames the victim.
Horton-Salway (2001) interviewed a woman with ME/CFS and her partner to identify how ME/CFS is constructed as a physical illness through attributional and identity narratives. Patient narratives make storytelling patterns within a community recognizable.
In an interview study of ME/CFS and fibromyalgia, Asbring (2001) found that: (1) these illnesses can involve a ’radical disruption in the women’s biography that has profound consequences for their identity’, particularly in their work and social life; (2) disruptions are partial rather than total, with different degrees of identity transformation; (3) many women also experience illness gains in relation to the new identity.
Reductions in Functioning
Reductions in functioning across occupational, education, personal or social domains are a characteristic part of living with ME/CFS. Social and economic reductions, personal losses and disruptions, in addition to the physical reductions experienced in everyday life are all commonly experienced. Economic and occupational reductions can be profound as well as multiple losses concerning their jobs and finances, including major disruptions to their careers and financial instability, and, in many cases, unemployment.
The loss of social roles and major disruptions in personal relationships are inevitable for most patients. People with ME/CFS describe reductions in the form of a loss of stamina and ability to develop future plans and changes in relationships, activities and social networks because of the illness.
Coping and Balancing Activity
The emotional and coping responses tend to vary according to which symptoms are present and the severity of the symptoms. For example, people who develope ME/CFS can use coping mechanisms such as seeking support from others, religion and using selective comparisons (’there are other people worse off than me’) to deal with their new illness. It was common for people with ME/CFS to use alternative medications and therapies.
Ray et al. (1993) described four coping strategies that may be used to manage ME/CFS: (1) maintenance of activity, (2) accommodating to the illness, (3) focusing on symptoms, and (4) information seeking. The most common of these themes across the studies reviewed by Anderson et al. (2012) was patient efforts to balance activity. Conflicting suggestions have been made about what is best for patients when it comes to balancing activity as a treatment or coping mechanism. Some patients say that they are able to control symptoms by reducing activity. Many people with ME/CFS practice ’living within [their] limits’ by monitoring and/or self-initiating the restriction of activities, including role constriction in employment.
Pacing oneself seems crucial to many patients. ME/CFS can hinder one’s ability to perform activities or cause the person to avoid activities altogether, which may in turn reinforce ME/CFS symptoms. The idea of balancing activity is supported by the envelope theory (Jason et al., 1999, 2010), which proposes that people with ME/CFS should try to balance their perceived and expended energy levels, thus staying within their ’energy envelope’.
Interventions for ME/CFS
Interventions for ME/CFS are based on theories of the processes that are responsible for the perpetuation of the symptoms. These theories are highly contested and have caused major controversies that have divided patients from doctors and psychiatrists.
Cognitive behavioural theories of ME/CFS assert that cognitions and behaviours perpetuate the fatigue and impairment of individuals with ME/CFS (Wessely et al., 1989, 1991). This assumption is contested by many patients and patient organizations. Vercoulen et al. (1998) developed a cognitive behavioural model of ME/CFS based on these assumptions. However, findings by Song and Jason (2005) suggested that the model was an inaccurate fit for individuals with ME/CFS, although the model fitted individuals with chronic fatigue in psychiatric conditions. In spite of the evidence against it, the socio-cognitive model continues to be cited as evidence supporting cognitive behavioural and graded exercise therapies for individuals with ME/CFS (e.g., White et al., 2011).
Sunnquist (2016) re-examined the behavioural pathway of the Vercoulen et al. (1998) model, which is characterized by causal attribution for symptoms, activity level, and fatigue and impairment, and found no support in the data for the Vercoulen et al. (1998) model. Sunnquist’s study suggested that activity level is an indicator of general illness severity, along with impairment and fatigue, and that individuals do not reduce their activity level due to illness beliefs. As the Vercoulen et al. model has provided the theoretical rationale for cognitive behavioural and graded exercise treatments for ME/CFS, these failed replication attempts support patient concerns about the lack of efficacy of these treatments, and their doubtful theoretical basis.
The PACE Trial: a Catalogue of Errors
Rarely in the history of clinical medicine have doctors and patients been placed so bitterly at loggerheads. The dispute has been a long time coming. Thirty years ago, a group of psychiatrists offered a hypothesis based on the biopsychosocial model (BPSM) in which ME/CFS is constructed as a psychosocial illness. According to their theory, ME/CFS patients are alleged to have ’unhelpful cognitions’ and ’dysfunctional beliefs’ that their symptoms are caused by an organic disease. The Dysfunctional Belief Theory (DBT) assumes that there is no pathology causing the symptoms; patients are being ’hypervigilant to normal bodily sensations’ (Wessely et al., 1989, 1991). The physical symptoms are presumed to be the result of ’deconditioning’ or ’dysregulation’ caused by sedentary behaviour, accompanied by disrupted sleep cycles and stress. Counteracting deconditioning involves normalizing sleep cycles, reducing anxiety levels and increasing physical exertion. To put it bluntly, the DBT asserts that ME/CFS is ’all in the mind’. Small wonder that patient groups have been expressing anger and resentment in their droves.
The PACE Trial is a textbook example of the top-down research approach with its hierarchical organization of personnel, duties and skill-sets (see Chapter 8). Unless carefully managed, the top-down approach creates a self-fulfilling prophecy with confirmation biases at multiple levels. At the top of the research pyramid sits Professor Sir Simon Wessely, originator of the DBT. The principal investigators (PIs) for the PACE Trial, Professors White, Chalder and Sharpe, are themselves advocates of the DBT, with connections both to the Department of Work and Pensions and to insurance companies.
The objective for the PACE Trial was to demonstrate that two interventions based on the DBT, cognitive behavioural therapy (CBT) and graded exercise therapy (GET), help ME/CFS patients to recover. According to critics, the PACE Trial team were operating within a closed system or groupthink in which they ’know’ their theory is correct and, with every twist and turn, are able to confirm their theory with subjective validation and confirmation bias. Groupthink occurs when a group makes faulty decisions because group pressures lead to a deterioration of ’mental efficiency, reality testing, and moral judgment’ (Janis, 1972).
Given this context, the investigators’ impartiality has been challenged with allegations of many potential conflicts of interest (Lubet, 2017). Furthermore, critical analysis suggests that the PACE investigators involved themselves in manipulating protocols midway through the trial, selecting confirming data, omitting disconfirming data and publishing biased reports of findings, which created a catalogue of errors. The outcome of the PACE Trial has been termed a ’travesty of science’, while sufferers of ME/CFS continue to be offered unhelpful or harmful treatments and are basically told to ’pull themselves together’. One commentator has asserted that the situation for ME patients in the UK is ’The 3 Ts — Travesty of Science; Tragedy for Patients and Tantamount to Fraud’ (Professor Malcolm Hooper, quoted by Williams, 2017: 1).
Critics suggest that the claimed benefits of GET and CBT for patient recovery are spurious. Serious errors in the design, the protocol and procedures of the PACE Trial are evident. The catalogue of errors is summarized in Box 24.3.
Box 24.3 The PACE Trial: A Catalogue of Errors
Critics suggest that the claimed benefits of GET and CBT for patient recovery have been exaggerated. Sir Simon, we respectfully suggest, got this one badly wrong. The explanation lies in a sequence of serious errors in the design, the changed protocol and procedures of the largest trial, known as the PACE Trial. The PACE Trial investigators neglected or bypassed many accepted scientific procedures for a randomized controlled trial, as follows:
Blocking the release of the raw data for five years and preventing independent analysis by external experts was tantamount to a cover-up of the true findings (Geraghty, 2016). ME/CFS patient associations were suspicious of the recovery claims concerning the GET arm of the trial because of their own experiences of intense fatigue after ordinary levels of activity, which were inconsistent with the recovery claims of the PACE Trial reports. For many sufferers even moderate exercise results in long ’wipe-outs’ in which they are almost immobilized by muscle weakness and joint pain. In the USA, post-exertional relapse has been recognized as the defining criterion of the illness by the Centers for Disease Control and Prevention, the National Institutes of Health and the Institute of Medicine. For the PACE investigators, however, the announced recovery results validated their conviction that psychotherapy and exercise provided the key to reversing ME/CFS.
When Alem Matthees, a ME/CFS patient, sought the original data under the Freedom of Information Act and a British Freedom of Information tribunal ordered the PACE team to disclose their raw data, some of the data were re-analysed according to the original protocols. The so-called ’recovery’ under CBT and GET all but disappeared (Wilshire et al., 2017). The recovery rate for CBT fell to 7% and the rate for GET fell to 4%, which were statistically indistinguishable from the 3% rate for the untreated controls.
In light of the re-analyses of the PACE Trial, the DBT appears to be dead in the water. There is an urgent need for new theoretical approaches and scientifically-based treatments for ME/CFS patients. Meanwhile, there is repair work to be done to rebuild patient trust in the medical profession after this misplaced attempt to apply the BPSM to the unexplained syndrome of ME/CFS.
The envelope theory of Jason et al. (1999, 2010, 2016) proposes that people with ME/CFS need to balance their perceived and expended energy levels and provides one way forward, pending further research. Ultimately, we are waiting for an organic account of ME/CFS that is competent to explain the symptoms and to open the door to effective treatments. Patients have a right to nothing less. Recent studies indicate that ME/CFS patients may have systemic abnormalities in cellular energy transduction, particularly when cells are put under mitochondrial stress (Tomas et al., 2017). It seems likely that a physical explanation for ME/CFS will eventually be found, leading to effective treatments.
Caring for a Person with ME/CFS
The lack of effective treatments and training for doctors about ME/CFS makes caring for a person with ME/CFS a challenging task. Fluctuating symptoms, restricted mobility and uncertain prognosis can all take a toll on the family carer. There is a paucity of studies about caring for people with ME/CFS. Experiences of parents with sons or daughters with severe ME/CFS are rarely presented in the scholarly literature. Haig-Ferguson (2014) investigated the impact of having a child with ME/CFS on family relationships from the parents’ perspective. Semi-structured interviews explored 18 parents’ experiences of having a child with ME/CFS and the impact of this on family relationships. Haig-Ferguson identified five main themes: ’Long and difficult journey’, ’Uncertainty’, ’Isolation and restriction’, ’Focus on the unwell person at the expense of family life’ and ’Parental roles’.
The parent(s) felt that life had become a constant struggle and they often felt exhausted with the uncertainty, isolation and restrictions. They felt as if ’family life had become focused on their unwell child, such that some expressed a need to escape from the situation’. They also described how they themselves could often feel marginalized and the illness felt like ’a destructive force’. However, many parents described team working and a feeling that family relationships had benefitted.
In another study, narratives of parent—carers were analysed using interpretative phenomenological analysis (Mihelicova et al., 2015). Results revealed themes of identity change, guilt, feeling like outsiders, uncertainty, changing perceptions of time, coping mechanisms and improvement/symptom management.
Siblings are also likely to experience distress. Velleman et al. (2016) invited 34 siblings to complete questionnaires measuring depression (HADS), anxiety (HADS and Spence Children’s Anxiety Scale: SCAS) and European Quality-of-life-Youth (EQ-5D-Y). The scores were compared with scores from normative samples. The results indicated that siblings had higher levels of anxiety on the SCAS than adolescents of the same age from a normative sample, although depression and quality-of-life were similar. Interviews with nine siblings of children with ME/CFS suggested restrictions on family life, ’not knowing’ and lack of communication as negative impacts on their family, and change of role/focus, emotional reactions and social stigma as negative impacts on themselves. However, they described positive communication, social support and extra activities as protective factors.
1. Further research is needed on developing applications of the biopsychosocial model to long-term conditions that are acceptable to patients.
2. Self-care ability in people living with diabetes and ME/CFS warrants further in-depth research.
3. Jason’s envelope theory provides a possible basis for ME/CFS treatment, pending further research.
4. Further research on stigma prevention in relation to both diabetes and ME/CFS is necessary.
1. Chronic illnesses can strike at any age but more often in middle and older age groups and, while they can be fatal, most people diagnosed with a chronic illness live for many years with the condition.
2. Management of chronic diseases is a principal feature in the lives of 10—15% of the population. This huge amount of informal care occurs almost invisibly to outsiders, with little recognition or financial support from society at large.
3. Chronic illness involves restrictions on activities of daily living and increases in pain and fatigue. ’Juggling’ relationships with health professionals, family and friends requires many adaptations and adjustments. Patients may have views about their care and treatment that differ from those of the professionals.
4. Type 2 diabetes mellitus is the most common form of the disease. People can develop it at any age, even during childhood.
5. Stigmatization can occur with long-term conditions and may be experienced by people with diabetes or ME/CFS. This is an extra burden for patients and informal carers to carry. Sources of stigma include the media, health care professionals, friends, family and colleagues.
6. The practicalities of living with diabetes or ME/CFS can be highly stressful, but people with diabetes or ME/CFS do not need to put their lives on hold.
7. For ME/CFS, pacing one’s activity seems crucial to many patients. ME/CFS can hinder one’s ability to perform activities or cause the person to avoid activities altogether, which may in turn reinforce ME/CFS symptoms. The idea of pacing or balancing activity is supported by Jason’s envelope theory.
8. The Dysfunctional Beliefs Theory of ME/CFS assumes that patients have unhelpful cognitions and dysfunctional beliefs that their symptoms are caused by an organic disease when no pathology is present. The theory is unsupported by empirical evidence, but remains the main basis for treatment in the UK.
9. Parents of children with ME/CFS tend to feel that life is a constant struggle with exhaustion, uncertainty, isolation and restrictions. They may feel as if family life is focused on their unwell child, such that some express a need to escape from the situation.
10. More in-depth training of medical and nursing students concerning ME/CFS and more effective treatments based on scientific evidence are urgently needed.