2.4.1 Psychiatry and Psychology
Both medical and psychiatric literature point towards high levels of co-morbidity between CFS and psychiatric and neuropsychiatric disorders (Lawrie and Pelosi 1995; Rusconi et al. 1994; Hawton and Cowen 1990; Deale and David 1994; Manu, Lane, and Matthews 1993; Shorter 1996), as well as sleeping disorders including narcolepsy (Fossey et al. 2004; MacFarlane et al. 1996; Moldofsky 1995; Schaefer 1995; Ambrogetti and Olson 1994; Manu, Lane, and Matthews 1993; Kempenaers, Bouillon, and Mendlewicz 1994). Psychiatric co-morbidity and high levels
Medical and Psychiatric Literature of psychological distress are common in CFS patients to an extent that cannot be considered coincidental (Kane, Gantz, and DiPino 1997; Michiels and Cluydts 2001; Quillian 1995; Wessely 1993). These co-morbidities do not establish CFS as a psychiatric or neuropsychiatric disorder. This research does, however, demonstrate the need to examine such co-morbidity, both in terms of the degree to which CFS may lead to co-morbidity, and in order to clarify what CFS is on its own terms.
The medical and psychiatric literature has described CFS as neuromyasthenia (Strickland et al. 2001; Stricklin, Sewell, and Austad 1990; 1992), neurasthenia2 (Abbey and Garfinkel 1991), and asthenia3 (Shahar and Lederer 1990). Each of these explanations indirectly implies a relationship between CFS, hysteria and somatisation processes. Substantive collections, such as Post-viral Fatigue Syndrome (Jenkins and Mowbray 1991), and The Clinical and Scientific Basis of CFS (Hyde 1992), strongly challenge these views. Whether this challenge has been observed is questionable.
De-Lange, Kalkman, et al. propose that “CFS may be associated with dysfunctional motor planning” and that motor disturbances may be a crucial component of CFS (2004, p.1948). They do not give reasons for this connection. Research shows that the disturbances to cognitive functioning (Short, McCabe, and Tooley 2002; Capuron et al. 2006) , motor control dysfunction (de-Lange et al. 2004; Siemionow et al. 2004; Starr et al. 2000) working memory deficits (Caseras et al. 2006; Dobbs, Dobbs, and Kiss 2001), neuropsychological functioning (Quillian 1995; Kane, Gantz, and DiPino 1997; Michiels and Cluydts 2001) and the poor attenuation of attention (Ray, Phillips, and Weir 1993) that have been identified in CFS patients cannot be attributed to depression, anxiety or psychological distress (Michiels and Cluydts 2001; Wessely 1993). These studies potentially inform an understanding of what CFS is not and may provide traction on establishing what CFS is.
The studies considered in this section use a variety of diagnostic criteria. Some do not mention the diagnostic criteria used. They tend not to be concerned with assessing CFS on its own terms but rather in the light of more established conditions.
2.4.2 Immunology
Numerous studies suggest that CFS may be caused by viral infection, most commonly the Epstein-Barr virus (Lundell et al. 2006; Okano, Thiele, and Purtilo 1990; Drago et al. 1996; Mitterer et al. 1995), Rickettsiae and Chlamydiae (Bottero 2000), an enterovirus (Colby 2007),
2 Neurasthenia n. A disorder characterized by feelings of fatigue and lassitude, with vague physical symptoms such as headache, muscle pain, and subjective sensory disturbances, originally attributed to weakness or exhaustion of the nerves and later considered a form of neurotic disorder. (Oxford English Dictionary 1989)
3Asthenia n. weakness or loss of strength. Asthenic adj. describing a personality disorder characterised by low energy, susceptibility to physical and emotional stress, and a diminished capacity for pleasure. (1994)
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mononucleosis4 (Buchwald et al. 1990), human herpes virus-6 (Wagner et al. 1996; Kato et al. 1992), Coxiella burnetii infection (Iwakami et al. 2005) or various other viral and immunological dysfunctions (Tirelli et al. 1994; Branco et al. 1994; Calabrese, Davis, and Wilke 1994; Fucikova and Petanova 1993; Cannon et al. 1997; Khan et al. 1993; Dechene 1993). Parvovirus B19 infection has similar symptoms to CFS but there is no conclusive evidence that CFS is a case of parvovirus B19 infection (Heneine et al. 1993; Daly, Thorne, and McIntosh 1997; Matano et al. 2003).
Lindh, Samuelson et al. (1996) found no evidence to support the hypothesis that CFS is caused by enteroviruses. Similarly Heneine, Chapman et al. (1993) found no evidence for the hypothesis that a retrovirus of unusual mode causes CFS. In sum, these findings suggest that although viral infection may be a precursor to CFS, continued viral infection is not present in CFS patients.
Immune dysregulation is commonly found in CFS patients (Gupta et al. 1997; Kashipaz et al. 2003; Kennedy et al. 2006; Kennedy, Spence et al. 2004; Levine et al. 1998; Maher, Klimas, and Fletcher 2005; Marotta, Improta, and Pinto 1994; Sacerdote et al. 1999; Shin et al. 2004; Skowera et al. 2004; Pinto 1995; Plioplys 1997; Dechene 1993; Delahunt et al. 1992; Fletcher, Maher, and Klimas 2002; Gaab et al. 2005; Hickie, Lloyd, and Wakefield 1992; Siegel et al. 2006; Tomoda et al. 2005).
Multiple chemical sensitivities and allergies appear to be common forms of this immune dysregulation (Miller et al. 2000; Lindh and Evengard 1999; Vecchiet et al. 2001; Bell, Baldwin, and Schwartz 1998; Lohmann, Prohl, and Schwarz 1996).
Although there does not appear to be a consistent pattern to the immune dysfunction in CFS, immune dysregulation in CFS appears to be centrally mediated (Natelson et al. 1998; Lyall, Peakman, and Wessely 2003). This research needs to be extended.
Since the immune dysfunction present in CFS may not be purely immunological, further investigation would need to occur from an interdisciplinary perspective. Such research may clarify somatic aspects of the condition not presently well understood and could potentially clarify diagnostic criteria and treatment approaches.
2.4.3 Hormonal studies
Various authors have investigated the role of hormones in CFS and concluded that, other than those hormones involved in the hypothalamo-pituitary-adrenal (HPA) axis, there are no significant hormonal abnormalities (Bennett et al. 1997; Garcia-Borreguero et al. 1998; Garrison
4Mononucleosis: The condition in which the blood contains an abnormally high number of mononuclear leucocytes (monocytes) (1994).
Medical and Psychiatric Literature and Breeding 2003; Grans et al. 2007; Murphy et al. 2004; Sackett-Lundeen et al. 1999; Young et al. 2000). By contrast the HPA axis is found to be dysregulated in CFS patients (Angel et al. 1998; Di-Giorgio et al. 2005; Jerjes et al. 2005; Jerjes, Peters et al. 2006; Jerjes, Taylor et al. 2006; McKenzie et al. 1998; Segal, Hindmarsh, and Viner 2005; Sharpe et al. 1998; Strickland et al. 1998).
CFS patients appear to hyposecrete the stress hormone cortisol. Segal, Hindmarch and Viner (2005) suggest that HPA axis dysregulation in CFS may be due to a reduction in central nervous system (CNS) stimulation of the adrenal glands.
A number of authors have investigated the roles of serotonin and 5-hydroxytryptamine (5- HTP) in the CNS in the dysregulation of the HPA axis in CFS patients. The 5-HTP modulator system in the CNS is clearly consistently disturbed in CFS patients (Berwaerts, Moorkens, and Abs 1998; Cleare et al. 2005; Dinan et al. 1997; Georgiades et al. 2003; Yamamoto et al. 2004). This potentially involves a genetic component which also manifests in immune system dysregulation (Kaushik et al. 2005; Rajeevan et al. 2007).
The pituitary-adrenocortical dysfunction found in short-term shift workers has been compared to CFS (Chattington et al. 1996).
2.4.4 Neurology
A variety of neurological studies have proved inconclusive (Arnold et al. 2002; Lange et al. 1998; Puri 2001, 2006; Badawy et al. 2005; Billiot, Budzynski, and Andrasik 1997; Chaudhuri et al. 2003; Chaudhuri et al. 1997; Greco, Tannock, and Brostoff 1997; Hannestad, Theodorsson, and Evengard 2007). There are, however, a number of areas in which findings have been made.
Neurally-mediated hypotension has been found in CFS patients, particularly in the form of orthostatic intolerance (Barron et al. 1999; Benoit, Wein, and Phaneuf 1999; Calkins 1998; Freeman and Komaroff 1997; Yamamoto, LaManca, and Natelson 2003; Rowe and Calkins 1998; De Becker et al. 1998; Bou et al. 1995). Two studies have shown considerable improvement in the symptoms of CFS by treating neurally mediated hypotension (Thomas and Bell 2000; Bou et al. 1995). These studies have not been repeated.
Numerous neurological studies have observed hypoperfusion and/or low metabolic rate in various regions of the brain in CFS patients (Abu-Judeh et al. 1998; Bested, Saunders, and Logan 2001; Costa, Tannock, and Brostoff 1995; de-Lange et al. 2005; Khan et al. 2003; Natelson et al. 2005; Palaniappan and Sirimanna 2002; Siessmeier et al. 2003; Tirelli et al. 1998; Yoshiuchi, Farkas, and Natelson 2006).
Sherlin, Budzynsky et al. (2007) have investigated characteristic patterns of neural activity in the brains of CFS patients and their healthy identical twins. The authors find significant
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differences in brain activity in the limbic system and the left and right forebrain; “the study corroborates that slowing of the deeper structures of the limbic system is associated with affect. It also supports the neurobiological model that the right forebrain is associated with sympathetic activity and the left forebrain with the effective management of energy” (Sherlin et al. 2007, p.1438).
2.4.5 Comparative Studies
Comparisons between Gulf War Syndrome and CFS are common. Kang, Natelson et al. (2003) have examined the prevalence of CFS in Gulf War Syndrome sufferers. Pollet et al. (1998) report that in a comparison of Gulf War veterans who suffered from Gulf War Syndrome those who concurrently suffered from CFS were less likely to report a sudden onset to their CFS. Chester and Levine (1997) have studied the concurrence of CFS and Sick Building Syndrome. Johnson and Natelson (1993) have shown that information processing deficiencies in Multiple Sclerosis and CFS are similar. Although interesting to some parties, these comparisons do not provide insight into ‘what the experience of CFS is’.
Heijmans and de Ridder (1998) have compared CFS and Addison’s disease as examples of chronic illness. Five dimensions that may differ according to the specific chronic illness are identified: identity, time-line, control/cure, cause and consequences. Conclusions to be drawn from this study are unclear. Whether this research lends any credence to the CFS diagnosis or in any way supports the claim that illness is occurring is debatable.
2.4.6 Other Medical Studies
Herrel, Ashton, et al. (2001) suggest that CFS is moderately heritable but do not identify why this is the case.
Van Rensburg, Potocnik, et al (2001) demonstrate identifiable patterns of trace element concentrations in the blood of CFS patients but do not suggest that this is causative.
Several authors have found evidence of significant oxidative stress in CFS patients (Kennedy et al. 2005; Maes, Mihaylova, and De-Ruyter 2006; Richards, Wang, and Jelinek 2007).
Lindal, Thorlacius, Bergmann, and Stefansson (1996) use grid areas to assess the localisation of pain in CFS sufferers and report that the most common sites for pain were the neck, right buttock, chest, calves and lower back. The significance of these localisations and the source of this pain are not identified.
A study using repeated action research cycles focuses on “contextually sensitive forms of language and models for service delivery suitable for people with CFS in a general practice
Psychosomatic Medicine, Psychology and Psychotherapy setting” (Denz-Penhey and Murdoch 1993). The study is largely inconclusive and has not been repeated.
Medical literature that is specific to women principally measures women’s disability due to CFS by considering reduced functional capacity, for example, reduced cardiovascular fitness and stamina (Riley et al. 1990; Sisto, LaManca, Cordero, Bergen, Ellis et al. 1996; Cordero et al. 1996; Sisto et al. 1995), and women’s information processing efficiency (Barrows 1995; DeLuca, Johnson, and Natelson 1993). Articles that examine women’s experiences in these respects briefly attempt to quantitatively assess self-reported degrees of fatigue and reduced functional capacity (Riley et al. 1990; Barrows 1995). Results vary considerably.
Harlow, Signorello, et al (1998) suggest that women with CFS report more gynaecological complications but a lower level of premenstrual symptomatology than controls. Why this would be the case is not discussed.
Medical and psychiatric research on CFS is informative in a number of ways. This research does suggest that CFS cannot simply be considered a psychiatric or psychological disorder. It does begin to establish the degree to which co-morbidity between CFS and psychiatric and psychological disorders occurs. The medical literature shows some promise in establishing specific somatic aspects of the disorder, particularly that certain brain regions are in some way implicated in the condition. These findings are not accurately reflected in the diagnostic criteria for CFS, have not clarified the etiology of CFS and have not been integrated into management and treatment programs. More research needs to be done to clarify and establish these findings and their implications.
Contributions from medicine and psychiatry are often difficult to compare since they are based on a variety of diagnostic criteria and often tend to address an isolated aspect of the syndrome. These studies are not consistently or comprehensively informed by the phenomenology of CFS.
2.5 Psychosomatic Medicine, Psychology and Psychotherapy
2.5.1 General Literature
The literature on CFS that is not specific to women from the disciplines of psychosomatic medicine, psychology and psychotherapy provides varying accounts of illness phases (Jason, Fennell et al. 2000) and sufferers coping strategies (Findley et al. 1998; Ax, Gregg, and Jones 1998; Nater et al. 2006; Ray, Jefferies, and Weir 1995; Ray, Jefferies, and Weir 1997). These studies do not demonstrate any consistent patterns.
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Research from these disciplines points towards high levels of co-morbidity between CFS and personality disorders (Blakely et al. 1991; Henderson and Tannock 2004; Buckley et al. 1999; Walford, Nelson, and McCluskey 1993; Wood and Wessely 1999), sleep disorders (Stone et al. 1994) mood disorders (Kelly et al. 1999; Stone et al. 1994; Edwards et al. 2001), and an association between CFS and alexithymia (Friedberg and Quick 2007), depression and atypical depression (Buckley et al. 1999; Kirk, Hickie, and Martin 1999; Henderson and Tannock 2005; Moss-Morris and Petrie 2001; Van Hoof, Cluydts, and De Meirleir 2003). Some CFS patients concurrently suffer from seasonal affective disorder or have a disorder which has features of both conditions (Lam 1991). Brunello (1999) suggests that a subgroup of CFS sufferers may represent a variant of dysthymia.
The co-morbidities established by this research do not determine CFS to be a psychosomatic or psychological disorder. Like the co-morbidities established in the medical and psychiatric literature, this research does demonstrate the need to examine such co-morbidity, both in terms of the degree to which CFS may lead to co-morbidity, and in order to clarify what CFS is.
Shorter (1993, 1997) places CFS within the history of somatisation5 disorders in the North American context. Butler (2001) and Howlett and Lindegger (1996) claim to identify the somatisation tendencies of CFS sufferers and suggest that somatisation may be a consequence of attributional style. CFS is attributed by Garralda (1992) to the somatisation of distress in childhood. These studies do not provide clear explanations for what somatisation represents as a process. They do not comment on how or why somatisation may be occurring in the case of CFS. In this literature, somatisation as an explanatory device does not shed light on how CFS might be effectively managed or treated. Or, in broader terms, as it is used here, somatisation does not lead to an understanding of how this form of suffering might be attended.
2.5.2 Literature specific to Girls and Women
A number of studies suggest that CFS is a modern form of neurasthenia6 (Wessely 1997; Abbey and Garfinkel 1991; Saltzstein et al. 1992). Despite increasing challenges (Luthra and Wessely 2004) to this perception these studies continue to carry considerable influence in the research community. They are widely cited and referenced.
5Somatisation disorder is characterised by:
Recurrent, multiple somatic complaints for which medical attention is sought but which have no apparent physical cause are the basis for this disorder. Common complaints include headaches; fatigue; allergies; abdominal, back, and chest pains; genitourinary symptoms; gastrointestinal symptoms; symptoms suggesting a neurological disorder; and heart palpitations (Davison and Neale 1994, p.169).
6“Neurasthenia n. a set of psychological and physical symptoms, including fatigue, irritability, headache, dizziness, anxiety, and intolerance of noise. It can be caused by organic damage, such as head injury, or it can be caused by neurosis (1994)”.
Psychosomatic Medicine, Psychology and Psychotherapy Psychosocial factors in adolescent girls are seen as a contributing factor to CFS in adolescents and CFS is presented as closely related to anorexia nervosa and bulimia nervosa (Pelcovitz et al. 1995). It is not uncommon for women with either anorexia nervosa or bulimia nervosa to be initially misdiagnosed with CFS (Griffiths et al. 1996). These relationships are not explored in depth and these studies do not help to clarify what CFS is.
Tuck and Wallace (2000) use the Derogatis Stress Profile (DSP), Spielberger Trait-Anger Scale, Ways of Coping Survey, Perceived Stress Scale and Profile of Moods States (POMS) Survey to confirm that CFS disrupts women’s quality of life, relations with others, self perception and career. Goodwin (1997) has explored the links between marital relationships and health in women with CFS, reporting on the views of both husbands and wives. Both of these studies identify substantial ways in which CFS disrupts sufferers’ lives. Fischler, Cluydts, et al. (1997) argue that high levels of anxiety in women CFS sufferers may be linked to a longer duration of illness. These ideas are echoed in Walters and Charles’ (1997) claim that the unpredictability that an illness state produces in women’s lives increases anxiety for CFS sufferers. Wheeler (1992) focuses on the specific issue of self-blame in Californian women CFS sufferers. This is the only published study identified in this review that uses a phenomenological approach. The study is not concerned to describe the experience of CFS as a whole. These findings are important and arguably need to be reflected in management or treatment programs for CFS sufferers. In isolation, however, this research does not provide a comprehensive approach to CFS.
Bell, Baldwin, et al. (1998) claim that early life stress and negative paternal relationships can be linked to chemical intolerance and CFS in middle-aged women. Why this would be the case is not explored.
To assess the coping strategies of women with CFS Saltzstein, Wyshak et al. (1998) employ the Beck Depression Inventory, Sickness Impact Profile, Defence Mechanism Rating Scale, a modified Karnofsky scale, and semi-structured interviews. The authors report:
Findings suggest that improved health is associated with a physician who made a relatively early diagnosis and was perceived by her patients as optimistic about prognosis. … Improvement was also associated with subjects feeling that their physician believed that they were ill, offered them multiple treatment options, and allowed for frequent, lengthy medical visits in which they actively participated in their health care. (Saltzstein et al. 1998, p.312) This study highlights the need for patient validation that legitimises illness experience. Such validation appears crucial to the success of the patient-practitioner relationship and recovery from illness.
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Åsbring and Närvänen (2004) examine the strategies that women with CFS employ to gain power and control during health care processes to enable them to manage their illness and influence health care providers. The authors report that in interactions with care-givers women with CFS use exiting, demonstrative distancing, non-compliance, confrontation, making demands and persuasion/insistence techniques to gain the opportunity to have mutual influence over the actions taken in their treatment process. Women perceive gaining knowledge about their illness as a means to exert control over their illness experience.
A personal account of the difficulties involved in convincing others that CFS is a real disease is offered by Phyllis Chesler (1998) who is a psychologist by profession.
Moss-Morris, Petrie et al. (1996) examine the relationship between functioning and illness perceptions. They conclude that illness perceptions have more influence on levels of disability than the coping strategies that participants have learned. The authors observe that participants with a strong illness identity who believed their illness was out of their control, stress induced, and likely to have serious consequences, were the most disabled. In contrast, in qualitatively