People with difficult-to-diagnose illnesses face four coping challenges:
Four coping challenges faced by individuals with difficult-to-diagnose illnesses are suggested. These challenges are situational ambiguity, characterological attributions, blocked information seeking, and limited opportunities for downward social comparisons.
Authors in the health-care literature have often discussed individuals who frequent medical facilities with health concerns that do not have a physiological basis but that are believed to reflect psychological problems.  These individuals are referred to as somaticizers, hypochondriacs, or overutilizers.
However, the literature has virtually ignored another group of individuals who frequent medical facilities and whose medical records may look very much like the group just described. These individuals have physiologically based complaints that are inaccurately attributed to psychological factors because they have rare, difficult-to-diagnose, or typically underdiagnosed illnesses.
In this article, I discuss the problems experienced by these individuals using as an example one group of difficult-to-diagnose patients, those suffering from Interstitial Cystitis. Two models of coping are discussed, and from these models four issues facing the sufferer of a difficult-to-diagnose illness are inferred.
Although the phenomenon of difficult-to-diagnose illness is not addressed in the professional literature, it has been addressed in the popular literature. In Redbook, Castleman  discussed illnesses that physicians often fail to detect or that have symptoms which physicians frequently claim are emotionally based.
Castleman believes these diseases are misdiagnosed because of their insidious onset; that is, they are characterized by vague, nonspecific symptoms that progress slowly. The diseases he described are multiple sclerosis, systemic lupus erythematosus, scleroderma, pet-borne infections, tropical diseases, and diseases resulting from environmental pollutants. Although Castleman did not mention Interstitial Cystitis, it is a disease often misdiagnosed and attributed to emotional problems.
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Interstitial Cystitis is an inflammation of the bladder wall marked by symptoms of urinary frequency and urgency often accompanied by pelvic pain. Urology researcher Messing observes: It is found most frequently in women. Diagnosis is based on the presence of the urinary symptoms described, the absence of other urological or gynecological disease, and the presence of characteristic petechial hemorrhages on a cystoscopic examination. These hemorrhages, or glomerulations, are often seen only when the cystoscopy is done under general anesthesia: that allows for sufficient bladder distention. 
The etiology of Interstitial Cystitis is unknown although researchers have suggested that frequent bacterial infections, antibiotics, allergic reactions, or autoimmune reactions may lead to a breakdown in the coating of the bladder wall, leaving it unprotected against the acidity of urine.  No effective treatment exists for Interstitial Cystitis, although some procedures have produced relief in some patients (e.g., bladder distention, oral and intravesical medications, dietary changes, transcutaneous electrical nerve stimulation, and bladder surgery. 
Interstitial Cystitis is generally underdiagnosed, and until recently, the amount of research conducted on the disease has been extremely limited. Meares described Interstitial Cystitis as a “mysterious disease that is often unrecognized by clinicians and that, when recognized, often responds suboptimally to treatment.” 
It is reported that patients often suffer from the symptoms of Interstitial Cystitis for an average of 7 years before a diagnosis is made. Dr. Vicky Ratner, herself a sufferer of Interstitial Cystitis, stated: 
Eleven months, 10 urologists, and 2 allergists later, the diagnosis of Interstitial Cystitis was confirmed, a diagnosis that was largely attributable to my own medical expertise as well as my determination to find an explanation for the symptoms that had plagued me for so long.
In addition to experiencing unexplained, often debilitating, symptoms for an extended period, these women are often told that their pain is emotionally based.  Harbison, a physician who suffers from Interstitial Cystitis, reported being told at one time to quit medical school and at another time to deal with her problem of “not wanting to be a woman.”  Grannum Sant, a podiatrist and a leading Interstitial Cystitis investigator, admitted that at one point, urologists believed these patients were “crazy” and “neurotic.” 
Because they lacked knowledge of this disease, doctors suggested to their patients that mental health problems were leading to these otherwise unexplainable symptoms.
Seemingly because they lacked knowledge of this disease, medical professionals suggested to their patients that mental health problems were leading to these otherwise unexplainable symptoms.
Although there is clear evidence that Interstitial Cystitis has a physiological basis and no evidence that it is caused by stress,  the illness seems to result in psychosocial distress and impaired functioning. Dr. Vicky Ratner discussed a pilot study conducted by the Interstitial Cystitis Association and the Urban Institute in Washington, DC, which surveyed women with Interstitial Cystitis. Of the women who responded, 40% were not able to work, 28% had experienced the breakup of a relationship, 58% reported being unable to have intercourse, 12% had attempted suicide, and 55% had considered suicide subsequent to the onset of their illness. 
Although the emotional distress caused by Interstitial Cystitis is most likely the result of many factors (e.g., chronic pain and limitations of functioning), it seems that the process of experiencing an undiagnosed illness with symptoms inaccurately attributed to psychological factors is, in itself, stressful. The resources of these individuals are called upon to cope with these debilitating symptoms at the same time as the symptoms are often being attributed, incorrectly, to poor mental health.
Coping has been defined as “the process of managing demands (internal or external) that are appraised as taxing or exceeding the resources of the person.”  Authors in healthcare literature have dealt extensively with the issue of coping with illness, but there is little written that directly applies to individuals coping with a difficult-to-diagnose illness. Models that address coping with negative events in general, however, might suggest what coping challenges these individuals face.
Coping is the process of managing demands (internal or external) that are appraised as taxing or exceeding the resources of the person.
Adjustment involves three cognitive tasks: finding meaning in the experience, regaining a sense of mastery over the event, and enhancing one's self-esteem.
Two models of coping (Lazarus & Folkman, and Taylor) propose that coping is enhanced if an individual understands or clarifies certain aspects of the negative event (e.g., its degree of imminence and threat or its cause). Because individuals with undiagnosed illnesses are unsure what the threatening event actually is, these processes can be hindered. By identifying coping processes that depend on accurate identification of the negative event (i.e., a diagnosis), the coping challenges faced by individuals who do not have such information can be better understood.
Lazarus and Folkman's  description of the coping process considers situational factors, personal factors, and outcome measures:
Taylor  proposed the theory of cognitive adaptation to threatening events. The theory suggests that adjustment to a threatening event involves three cognitive processes: finding meaning in the experience, regaining a sense of mastery over the event itself and over one's life generally, and enhancing one's self-esteem.
Four coping challenges faced by individuals with difficult-to-diagnose illnesses are identified on the basis of the concepts of the two coping models discussed and the experiences reported by women with interstitial cystitis. The challenges are situational ambiguity, characterological attributions, blocked information seeking, and limited opportunities for downward social comparisons.
Lazarus and Folkman  believe the situational factor of ambiguity is an important influence on the coping process. Ambiguity exists when the environmental stimuli are unclear, making it difficult to assess harm and therefore to plan action to combat or avoid the harm. The specific influence of ambiguity on the coping process is unknown, although the resolution of ambiguity is believed to contribute to coping effectiveness. 
Ambiguity drains coping skills: Without a diagnosis, one cannot predict the duration of symptoms or the future effects of illness, nor can one plan.
When resolution is impossible, ambiguity represents a continuing coping challenge. An illness that is not yet accurately diagnosed can be conceptualized as an ambiguous situational factor. Without a definitive diagnosis, one cannot predict the duration of the symptoms and the future harmful effects of the illness, nor can one plan efforts to combat the illness process or manage the symptoms.
When one has a diagnosis of urinary tract infection, for example, one can be reasonably sure that drinking water regularly and taking antibiotics will help the symptoms. If one does not know the basis of the illness, as in the case of undiagnosed interstitial cystitis, one cannot be sure how to deal with the symptoms. The lack of a definitive diagnosis, therefore, would complicate the process of coping with illness because it would act as an ambiguous stimulus, making prediction and control difficult.
Attribution is also an important issue influencing coping with a difficult-to-diagnose illness. Thylor's theory  suggests that the process of attribution, answering the question of why an event has occurred, can be an important aspect of the search for meaning. For example, Taylor's sample had attributed their breast cancer to factors such as stress, a particular carcinogen, heredity, diet, or a blow to the breast. Most of these factors were past events that were not likely to recur or were ongoing controllable events. The cancer was therefore perceived to be manageable.
Wrongly attributing physiologic illness to psychological problems causes low self-esteem and hopelessness. The tactic hinders coping rather than enhancing it.
Many women with interstitial cystitis are told their symptoms are caused by psychological problems. This is an example of a characterological attribution , a belief that a negative event is caused by a personality trait of the individual experiencing the event (e.g., being psychologically troubled) rather than by something specific that the individual does (a behavioral attribution).
Because one then tends to blame oneself for the problem rather than blaming something outside oneself, low self-esteem can result. Because the attribution for the negative event is toward a relatively enduring trait, there is little hope of managing the course of the event or preventing its recurrence. The individual experiences feeling a lack of control.
Thus, attribution of a physiologically based illness to psychological problems is an example of a characterological attribution that can lead to low self-esteem and hopelessness. In this case, the attributional process that Taylor stressed as an important aspect of the search for meaning can lead to beliefs that hinder effective coping rather than enhancing it.
Information seeking plays an important role in Taylor's second theme of cognitive adaptation, gaining a sense of mastery. She quoted the spouse of one of the cancer patients she had studied. “She got books, she got pamphlets, she studied, she talked to cancer patients, she found out everything that was happening to her and she fought it.” 
Information helps patients participate in their care and master the illness. Those with rare diseases are denied this coping strategy: information is not only inaccessible, it is nonexistent.
Obtaining information seemed to increase the cancer patients' sense of involvement in their care and therefore their sense of control over their cancer.
There would be some obvious difficulties involved in gaining information regarding an illness yet to be diagnosed. Books and pamphlets are often written for those with specific diagnoses. Even medical textbooks are usually organized by diagnostic categories. Support groups can provide much information, but these groups are usually formed for individuals suffering from a specific illness.
Also, illnesses often remain undiagnosed because little is known about them due to limited research or clinical investigation. In such cases, information is not only inaccessible, it is nonexistent.
The process of enhancing self-esteem by downward social comparisons might be especially difficult for sufferers of a difficult-to-diagnose illness. When there is no diagnosis, there is no defined group of others with the same illness with which to compare oneself.
Downward social comparison can elevate self regard and one's subjective sense of well-being. Upward social comparion motivates improvement.
Also, although the comparisons made by the breast cancer patients were made on many dimensions (e.g., life situations and extent of illness), often the comparisons were made based on superior coping. One of Taylor's cancer patients said, “Some of these women seem to be devastated, and with really less problems than I encountered, you know, smaller tumors.” 
It would be difficult to conclude that one was coping better than others in relation to the severity of one's symptoms if psychological problems, usually associated with poor coping, were thought to be the cause of the illness. Therefore, not having a diagnosis limits available comparison to others, and attributing the symptoms to mental health problems limits positive comparisons on a dimension frequently used by those suffering from medical illnesses (superior coping).
Attributing illness to psychological factors, with no basis other than apparent overuse of the health-care system and lack of a diagnosis, is patient abandonment.
Systematic identification of difficult-to-diagnose illnesses and factors that might contribute to their being so misunderstood (e.g., varied symptomatology, rarity, and symptoms that mimic a well-known illness) should be determined. Once difficult-to-diagnose illnesses are identified, research on specific illnesses and dissemination of information can follow.
If these diseases were better understood by the health-eare community and the public, diagnostic periods would be shortened, and inaccurate etiological attributions to psychological factors would decrease.
The process of making psychological attributions of illness, with no basis other than the apparent overuse of the health-care system and lack of a definitive diagnosis, needs to be avoided. The influence of emotional factors on somatic symptoms should always be considered as part of a total diagnostic picture but should not serve as definitive etiological explanation because all other known physiological possibilities have been ruled out. The latter too often justifies premature abandonment by the medical community and low self-esteem in those for whom the inaccurate attributions are made.
The experiences of individuals with physiologically based illnesses who continue to be involved in lengthy and frustrating diagnostic processes and who continue to be told their symptoms are emotionally based merit further investigation. The four coping challenges outlined in this article need to be studied empirically. Only by understanding the experiences of these individuals can health-care professionals sensitively provide their care.
|1.||Corbin, L. J., Hanson, R. W., Hopp, S. A., & Whitley, A. (1988). Somatoform disorders: How to reduce overutilization of health care services. Journal of Psychosocial Nursing and Mental Health Services, 26(9), 31-34.|
|2.||Castleman, M. (1988, November). Medical mysteries: Diseases doctors fail to detect. Redbook, pp. 120-121, 180, 182.|
|3.||Messing, E. M. (1987). The diagnosis of interstitial cystitis. Urology, 29(4, Suppl.), 4-7.|
|4.||What is interstitial cystitis and what can be done about it? (1986). American Journal of Nursing, 86, 13-14.|
|5.||Interstitial Cystitis Association. (1987). ICA. New York: Author.|
|6.||Meares, E. M. (1987). Guest editorial: Interstitial cystitis-1987. Urology, 29(4, Suppl.), 46-48.|
|7.||Ratner, V. (1987). Rediscovering a ‘rare’ disease: A patient's perspective on Interstitial Cystitis. Urology, 29(4, Suppl.), 44-45.|
|8.||Lehman, B. A. (1986, September 22). Interstitial Cystitis pain is real. The Boston Globe. p.41.|
|9.||Lazarus, R. S., & Folkman, S. (1984). Coping and adaptation. In W. D. Gentry (Ed.), Handbook of behavioral medicine (pp. 282-325). New York: Guilford Press.|
|10.||Taylor, S. E. (1983). Adjustment to threatening events: A theory of cognitive adaptation. American Psychologist, 38, 1161-1173.|
|11.||Shalit, B. (1977). Structural ambiguity and limits to coping. Journal of Human Stress, 3(12), 32-45.|
|12.||Wortman, C. B. (1983). Coping with victimization: Conclusions and implications for future research. Journal of Social Issues, 39(2), 195-221.|
Health Care for Women International, 12:191-198, 1991