Pain, 49 (1992) 337-347 0 1992 Elsevier Science

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PAIN 02045

Psychological aspects of reflex sympathetic dystrophy: a review of the adult and paediatric literature Mary E. Lynch Pain Management Unit, Ambulatory Care Centre, Victoria General Hospital, Halifax, Nol,a Scotia B3H 2Y9 (Canada) (Received

23 May 1991, revision

received

and accepted

18 December

1991)

In 1864, W. Mitchell and colleagues first described the clinical syndrome which came to be known Summary as ‘causalgia’. Since that time, the concept of sympathetically related pain has evolved. There is general agreement that profound emotional and behavioural changes can follow these types of pain. Opinions have varied widely on the issue of a psychological etiology. It has often been suggested that certain personality traits predispose one to develop sympathetically related pain syndromes. A review of the literature reveals no valid evidence to substantiate this claim. Key words: Causalgia; Reflex sympathetic dystrophy; Etiology, psychological; Behaviour; Emotions

Introduction Mitchell et al. (1864) first presented a graphic clinical description of a group of patients who suffered from intractable burning pain following peripheral nerve injuries. This clinical syndrome was later called ‘causalgia’. Since that time, much has been written about the behavioural and emotional changes observed in patients suffering from sympathetically related pain syndromes. There is general agreement that these changes can be profound. There has been speculation that psychological factors predispose one to the development of sympathetically related pain syndromes (Lidz and Payne 1945; Shaw 1964; Bernstein et al. 1978; Van Houdenhove 1986; Sherry and Weisman 1988; Gybels and Sweet 1989). Most authors (Miller and DeTakats 1942; Echlin et al. 1945; Mayfield and Devine 1945; Spiegel and Milowsky 1945; Cullen 1948; Holden 1948; Sunderland and Kelly 1948; Mayfield 1951; Richards 1967; Bonica 1979; Ecker 1984; Schwartzman and McLellan 1987) have concluded that the behavioural and emotional changes are a result rather than a cause of pain.

Correspondence to: Mary E. Lynch, M.D., FRCPC, Pain Management Unit, Ambulatory Care Centre, Victoria -General Hospital, 1278 Tower Road, Halifax, Nova Scotia B3H 2Y9, Canada.

Despite this, the possibility of a psychological etiology continues to receive some support both in clinical practice and in the medical literature (Van Houdenhove 1986; Egle and Hoffmann 1990; Gybels and Sweet 1989). This, in addition to the fact that patients are sometimes accused of malingering (Arieff et al. 1964; Shaw 1964; Gybels and Sweet 1989), leads to mismanagement and increased suffering (Schwartzman and McLellan 1987). In this article the author has attempted to review all papers containing original psychological information of any type; this usually consists of behavioural descriptions or assessments of the patient’s emotional state. There are a few papers using specific measures, and these will be mentioned when details are reported. There are many papers in which statements are made regarding psychological aspects of the sympathetically related pain syndromes. (For an excellent short review and discussion, see Haddox 1989.) Only those papers containing original clinical data are reviewed here. For the purpose of this review, all types of pain are considered in which the sympathetic nervous system has been thought to play a role in the pathophysiology. This is in accordance with the view of Bonica (1953, 1979). Specifically, major and minor causalgia, reflex sympathetic dystrophy (RSD), Sudeck’s atrophy, algoneurodystrophy, shoulder hand syndrome, reflex neurovascular dystrophy, reflex dystrophy and post-trau-

338

matic dystrophy have been included. In accordance with the recent recommendations contained in the consensus statement of the Special Interest Group on Pain and the Sympathetic Nervous System (Janig et al. 19911, all of these conditions are included in the term, ‘reflex sympathetic dystrophy’. It is suggested that this term be used in a descriptive sense without implication for specific underlying mechanisms.

Psychological aspects of reflex sympathetic dystrophy in adults Description of behaviour and experience of pain

Mitchell et al. (1864) described the pain and behaviour observed in patients suffering from causalgia as follows: “Its intensity varies from the most trivial burning to a state of torture which can hardly be credited but which reacts on the whole economy until the general health is seriously affected.. . . Exposure to the air is avoided by the patient with the care which seems absurd and most of the bad cases keep the hand constantly wet.. . . As the pain increases, the general sympathy becomes more marked. The temper changes and grows irritable, the face becomes anxious and has a look of weariness and suffering. The sleep is restless and the constitutional condition reacting on the wounded limb exasperates the hyperaesthetic state so that the rattling of a newspaper, a breath of air, another’s step across the ward, the vibrations caused by a military band or the shock of the feet in walking, give rise to intense pain. At last the patient grows hysterical, if we may use the only term which covers the fact. He walks carefully, carries the limp tenderly with the sound hand, is tremulous, nervous and has all kinds of expedients for lessening his pain.” Livingston (1947) presents a soldier’s graphic description of the pain: “It is as if a rough bar of iron were thrust to and fro through the knuckles, a red hot iron placed at the junction of the palm and thenar eminence with a heavy weight on it and the skin was being rasped off of my finger ends”. Sunderland and Kelly (1948), in writing of causalgia, describe how, “Harassed by continuous pain and lack of sleep, the patient after a time may become a nervous wreck”. The authors noted that “if seen at this stage by an enthusiastic clinician with a psychosomatic bias, he may have his mind probed in search for psychic trauma, and the practitioner who suspects a psychogenic cause does not often fail to uncover a confirmatory history”.

The above descriptions assist in understanding why there has been such widespread attention to the psychological aspects of the disorder. A review of the literature revealed 29 articles (Table I> where psychological, emotional and behavioural aspects of patients suffering from sympathetically related pain are considered. All agree that this group of disorders can be associated with profound behavioural and emotional change. Many of these papers discussed the contribution of psychological causes. The majority concluded that there is no evidence to support a psychological cause or predisposing constitutional factor. It was said that the behavioural changes were a result of pain. The majority concluded this on the basis of observations made before and after relief of pain. Assessment of premorbid personality was attempted in only one study (Zachariae 1964). These studies are summarized in Table I. Further detail will be provided. Papers containing any information regarding psychological issues will be presented first. In most papers, this consists of general clinical observations on behaviour: in some, there has been an attempt at psychiatric on psychological assessment. This will be mentioned specifically when studies are described. There are several studies containing information obtained through psychometric tests. These will be presented separately. Papers containing psychological information of any type

Mayfield and Devine (194.5) and Mayfield (1951) have described the clinical presentation of approximately 120 soldiers who suffered from causalgia. “The disturbed emotional state of the more severe cases was most striking. Though most of them had been valiant soldiers until the moment of injury and subsequent to relief of pain were regarded as stable emotionally, from the moment of impact, in many cases, they were emotional derelicts. From a stable personality, they became irritable and shut in.. . extremely critical with little interest in family or friends, though many had been overseas for many months” (Mayfield 1951).“Patients kept their hands constantly covered with a wet cloth.. . one kept the hand protected constantly in dry flannel. Most would go to what appeared to be absurd extremes to prevent motion of the part.. . . The facial expression usually reflected intense suffering”. The authors go on to say, “It was impressive to observe the remarkable change in the patient’s attitude and behaviour following relief from pain. Without exception, they became pleasant, cooperative, and happy, and showed interest in their families and friends” (Mayfield and Devine 1945). Fifteen of these patients received a psychiatric examination after pain relief. All were said to have been “classified as a normal individual with a stable person-

339 TABLE I CHRONOLOGICAL

LIST OF PAPERS WHERE PSYCHOLOGICAL

ASPECTS OF RSD ARE DISCUSSED

Psychological measure

Study design

Conclusion regarding psychogenic etiology

case descriptions ‘neuropsychiatric study’ on some case descriptions case descriptions case descriptions case descriptions ‘psychiatric exam’ after pain relief case report by psychiatrist case descriptions ‘psychical assessment’ case descriptions case descriptions psychiatric consult in 11 patients case descriptions none

report of cases

-

report report report report report

_ -

so 24

case descriptions battery of psychological tests

report of cases between groups

Zachariae

47

psychiatric consultation

report of cases

1970 1970

Wirth, Rutherford Pak et al.

32 52

retro review retro review

1971

Omer, Thomas

70

1980

Pollack et al.

60

1981 1981

Wilson Subbarao, Stillwell

12s

1981 1983

Alioto Poplawski, Wiley

2 1.52

1983

DeLeo, Magni

74

1986

Van Houdenhove

32

case descriptions information regarding past history; psychiatric consultation on some patients 45% of patients said to require psychiatric consultation Frieburg personality Spreen anxiety MMPI MMPI, psychiatric consultation on some patients ‘evaluation for stress’ records of psychiatric consultation on some patients Eysenck Personality Inventory psychiatric assessment

1988

Haddox et al.

21 21

1990

Egle, Hoffmann

1990

Grunert

n

Date

Authors

1942

Miller, DeTakats

33

1944 194s 194s 194s 194s

Doupe et al. DeTakats Echlin et al. Spiegel, Milowsky Mayfield, Devine

10 54 30 9 15

194s

Lidz, Payne

1948 1948 1948 1949

Sunderland, Kelly Cullen Holden Shumacker. Abramson

34 24 32 142

1951 195s

Mayfield Langston, Cowan

10s 88

1957 1959

Owens Adler et al.

1964

1

8

20

MPQ DPQ STAI ‘clinical examination and comprehensive psychological testing’ No details presented MMPI

of of of of of

cases cases cases cases cases

case report

+

report of cases report of cases retro review report of cases

_ -

report of cases report of cases

review of cases

between groups

no direct conclusion

_ predisposing personality described _ ‘psychosomatic’ psychological factors important in determining the development mental factors said to be an important detail in the syndrome 37.1% said to have a history of psychiatric problems or emotional disturbance did not address

‘psyche’ important for development _

between groups retro review

did not address

case report report of cases

did not address no direct conclusion

between groups

no direct conclusions

retro review of patients referred for psychiatric assessment between groups

+

did not address

report of cases

+

report of cases

did not address

340

ality”. The authors conclude, “The personality changes always present during painful stages are secondary to pain. There is no evidence in our cases that there is a predisposing constitutional psychic factor in causalgia” (Mayfield and Devine 1945). Cullen (1948) described 24 patients diagnosed with causalgia; again, this was a military population. These patients were subjected to a “psychical” assessment. An opinion was expressed “at a time when the pain was not severe” in hopes of getting a better assessment of the premorbid personality. Two patients were said to be “definitely abnormal”. One was said to be “aggressive and impulsive” and the other was described as having “a mild anxiety state”. The authors had earlier described how “many of the victims were unconscious for a period or were subject to the strain of long evacuation from dangerous areas while in a very poor general state”, After observing these patients before and after relief of pain, the author concluded, “Too much attention has formerly been paid to personality changes. They are the result of pain. Once the pain is relieved, they disappear”. Shumacker and Abramson (1949) described 142 patients with post-traumatic vasomotor disorders. They have excluded major causalgia from their group but include other disorders in which they believe the primary mechanism is a “reflex disturbance.. . involving in the reflex arc the components of the sympathetic nervous system”. Today these patients would be categorized as falling within the spectrum of sympathetic disorders. The authors report, “In 11 individuals, definite evidence of some neuropsychiatric disorder was confirmed by psychiatric consultation”. It is not mentioned whether the rest of the group was assessed by a psychiatrist. It is stated that these individuals were treated primarily by neuropsychiatric means. Further description reveals that 1 individual made “fair progress with active exercise of the affected upper extremity In a second patient, symptoms “cleared so alone”. satisfactorily with ordinary physical activity that no psychiatric treatment was necessary”. In the remaining 9 patients “concerted efforts were made to give the patient insight into his condition and to make him understand that lie had no organic disorder which would prevent him from recovery provided that he continued to push himself to the limit in active use of the affected limb”. In most cases, the treatment also included “suggestive therapy under amytal narcosis and hypnosis”. The authors later state that in 4 patients, results with psychotherapy were good, in 1 fair, and in 4 poor. It is of interest that in 1 of 4 said to have a poor response, the authors state, “Although there was definite evidence of hysteria, no effect was noted from psychiatric treatment. Subsequently, however, considerable though incomplete improvement occurred after sympathectomy”.

The above description raises several issues. What the authors describe as ‘neuropsychiatric treatment’ included strong encouragement to use the affected limb actively. Physical activity alone is known to be an effective treatment for some cases of RSD (Schwartzman and McLellan 1987) and may explain the improvements observed. Amobarbital (Amytal) narcosis was also used. It is important to note that Tasker et al. (1980) found that low doses of intravenous thiopental abolished pain due to deafferentation. Tasker was unable to distinguish ‘psychogenic’ from ‘organic’ pain by using thiopental. Although amobarbital and thiopental have different half-lives, it is possible that amobarbital contributed to the pain relief noted in the patients described by Shumacker and Abramson (1949). One patient said to exhibit “definite evidence of hysteria” who subsequently experienced “considerable though incomplete improvement” with sympathectomy, illustrates the danger of concluding a psychological cause based on behavioural observations made while patients are suffering from pain. Shumacker and Abramson (1949) do not suggest that there is a psychiatric basis for complaints in all patients suffering from post-traumatic vasomotor disorder. In their concluding remarks, they state that one should utilize “psychotherapy in encouraging continued and gradually increasing active exercise of the affected limb and to employ more specific psychotherapeutic measures in those instances in which psychiatric factors are a definite accompaniment, if not the primary basis for disability”. Miller and DeTakats (1942) presented case descriptions of 33 patients diagnosed with Sudek’s atrophy. Most patients were said to have been “subjected to a neuropsychiatric study”. One patient was said to have been “a very unstable individual”, 1 “to have been repeatedly committed to an institution because of a severe psychoneurosis”, and 2 others to have “exhibited other instances of autonomic’ instability such as hypermotility of the intestinal tract and neurocirculatory asthenia”. In reviewing their entire series. the authors state “There has been, however, no single pattern or type of personality which was found to be characteristic of this syndrome.. . the nervousness and excitability of the late cases may just as well be the result of many months of suffering and not the cause of the syndrome, which most neurologists still maintain”. Doupe et al. (1944) presented case descriptions of 12 patients, 7 diagnosed with causalgia, 3 with “dystrophic pain”, 1 with “psychogenic pain” and 1 exhibiting osteoporosis and hyperhydrosis without pain. There is no formal psychological evaluation. The patient said to be suffering from psychogenic pain was diagnosed on the basis of a placebo response and inability to flex toes voluntarily when a well marked flexor response was obtained to plantar stimulation. The authors note

341

that causalgic pain “tends to lead to profound changes in the emotional state of the patient” and that “reclusive traits disappeared immediately” when the pain had been treated by sympathectomy. In an attempt to explain the great disparity in severity of the pain complained of by different patients, one theory presented is that of “psychogenic exaggeration of the pain in the most severe cases”. DeTakats (1945) reported 54 cases of causalgia. There is no mention of systematic psychological evaluation. One patient was said to “cry easily and had fainting spells”. She was said to have “a suggestible, psychoneurotic background”. Overall, he observed, “In this group of 54 cases, there was no evidence that any definite type of personality was responsible for this unusual reaction to a sensory stimulus”. Echlin et al. (1945) presented a descriptive study of 30 patients. No psychological measure or psychiatric examination is mentioned. The authors state, “In the patients in whom pain was relieved by sympathectomy there was a remarkably rapid return of the emotional state to normal levels if there had been emotional disturbances prior to the operation”. Spiegel and Milowsky (1945) described 9 cases of causalgia. There was no formal psychological or psychiatric examination; however, detailed descriptions of behaviour were presented. The authors state, “There wa$ no evidence in any of the cases of a personality disorder. . . . Personality disorders and hysteria are the result rather than the cause of causalgia”. Sunderland and Kelly (1948) described 34 cases of causalgia; behavioural descriptions were provided. Sunderland argues very strongly against conceptualizing behavioural or emotional factors as evidence for a psychological etiology. He states, “Some physicians and surgeons, finding it difficult to appreciate the fact that these patients suffer intensely, are ever on the alert for evidence which may suggest that they are dramatizing or exaggerating their symptoms. To adopt such an attitude is in fact to commit a grievous error; apart from the serious mistake in diagnosis, a great injustice is being done to the patient in attempting to lay upon him both the blame for being ill and the responsibility for curing himself ‘. Holden (1948) presented a retrospective review of 32 patients diagnosed with sympathetic dystrophy. No details are provided regarding behavioural observations and no mention is made as to whether patients were examined psychologically. It is stated, “In some of these patients, emotional disturbances have occurred with such severity and for so long a time that psychiatric therapy is necessary before and after treatment of their sympathetic dystrophy. There is sufficient evidence at the present time to maintain that the emotional instability is for the most part a result of the months of pain and discouragement these patients

endure and not the cause of the changes observed in the extremity”. Owens (1957) presented 50 cases of causalgia. There was no psychological evaluation and no details were provided regarding behaviour, emotion or psychological factors. The author states, “Frequently a psychogenic etiology is suggested, and many patients have been unjustly considered to be malingerers or hysterics. No constitutional psychogenic factor can be found”. Wirth and Rutherford (1970) presented a retrospective report of case histories of 32 patients diagnosed with causalgia. The authors state, “In reviewing the medical records of our patients, it was not unusual to encounter such impressions (malingerer, psychoneurotic, or emotionally unstable) or more diplomatic comments”. They later state, “Furthermore, many of these cases were not only initially misdiagnosed or maligned, but they were frequently mistreated.. . . Twelve of our 32 patients were considered to have an underlying psychiatric disturbance and yet only two of these had persisting emotional problems after their pain was relieved”. No further detail regarding psychological issues is presented. Alioto (1981) described 2 cases of RSD in which “behavioural treatment” was used. Patients were said to receive “an evaluation for stress” and rated “general feeling of well being” on a scale of O-10. Relaxation strategies, autogenic feedback training, education and cognitive approaches were used when patients did not respond adequately to corrective surgery and drug therapy or when there was difficulty in obtaining further stellate ganglion blocks. Both patients were said to have responded with 100% pain relief. The author approach to pain states, “The didactic-experiential control illustrates the importance of involving the patient in his or her own treatment by using techniques that place responsibility in the patient’s hands”. The author does not imply a psychological etiology. Poplawski et al. (1983) reviewed the hospital records of a series of 126 patients diagnosed with post-traumatic dystrophy. They added another 26 patients seen later upon referral. The authors examined 62 of the entire group and reported, “The patients’ general health and psychological state were assessed. For a number of patients, records of psychiatric assessments were available”. Nineteen of 62 patients they examined and 14 of 40 patients with long-term symptoms showed “overt psychiatric abnormalities, and there were two patients whose psychological makeup left them unable to cope well with even the ordinary stresses of life”. They made the unsubstantiated claim that “individuals with psychiatric illness are more prone to the development of dystrophy” but later quote and agree with Miller and DeTakats’ (1942) claim that no single pattern or type of personality is characteristic of this syndrome. They state, “We should not try to separate

342

post-traumatic dystrophy from a disturbed state of mind, but rather recognize the importance of the influence of state of mind on the severity and progression of this syndrome”. Indeed, many authors have suggested that psychological factors are important in the progression of RSD (Miller and DeTakats 1942; Poplawski et al. 1983; Schott 1986; Schwartzman and McLellan 1987). However, no study has examined this question to date. Studies containing data form psychometric tests

There are 3 reports in the literature which contain information regarding results on the Minnesota Multiphasic Personality Inventory (MMPI). Subbarao and Stillwell (1981), in a retrospective review, presented the records of 125 patients diagnosed with RSD. They extracted MMPI results and information regarding psychiatric consultations when available. A follow-up questionnaire was sent to all patients who were still living. Seventy-seven responses were received. Fortyfive patients (36%) had completed the MMPI. Analysis showed the Hysteria, Depression and Hypochondriasis scales to be abnormal in 14 patients. Thirty-two patients had received psychiatric evaluation. Some of these patients had not completed the MMPI. No further information regarding the content or conclusions of psychiatric interviews was presented. Outcome measures evaluated symptoms, medication intake, daily activities and vocational activities. It was concluded that “the outcome did not correlate with statistical significance to abnormalities on the MMPI, the need for psychiatric care or medicolegal involvement”. The question of a psychological etiology was not addressed. Wilson et al. (1981) administered the MMPI to a consecutive group of dystrophy patients as well as to patients with physical injuries that were not complicated by severe pain. None of the patients with pain demonstrated any pathological deviations on the MMPI. It was concluded that “patients with an abnormal personality are not predisposed to develop sympathetic dystrophy. However, when severe pain occurs, an individual’s ability to handle the situation can be complicated by any personality deficiencies”. This study is notable in that there was a control group. Unfortunately, not enough information was presented to support the author’s conclusions. Grunert et al. (1990) administered the MMPI to 20 patients diagnosed with RSD. The authors found 18 of these patients had elevations on the Hypochondriasis and Hysteria subscales. There was no control group. All patients had been referred for psychological evaluation and management. Patients were exposed to a combination of thermal biofeedback, relaxation and supportive psychotherapy. Results indicated that patients were able to increase their hand temperatures and reduce subjective pain ratings.

One must use caution in interpreting MMPI results in patients suffering from chronic pain. The simple fact that a patient suffers from pain will lead to elevations on several scales. This information is often misinterpreted. For further discussion, see Merskey (1987) and Haddox (1989). Haddox et al. (1989) administered three psychometric tests to a group of 21 patients with RSD. These results were compared to scores of 21 patients diagnosed with radiculopathy. The tests administered included the McGill Pain Questionnaire (MPQ), the Dartmouth Pain Questionnaire (DPQ), and the Trait subscale of the State Trait Anxiety Inventory (STAI). Statistical analysis revealed no differences between the two groups, despite the finding of more treatment successes in the radiculopathy group. Evidence for psychogenesis?

Ten papers are cited as evidence for a psychological etiology. Pak et al. (1970) presented a retrospective review of case records of 140 patients diagnosed with RSD. Fifty-two (37.1%) of these patients are said to have had a history of psychiatric problems or emotional disturbance prior to the onset of symptoms of RSD. Twenty-five were said to have had formal psychiatric consultations. The 3.5 patients not referred for formal psychiatric evaluation during their present illness were thought by their internists to have ‘psychiatric’ problems. No diagnostic criteria were offered; symptoms were not described. There is no further information regarding the history of “psychiatric problems or emotional disturbance”. It is simply stated that there is this history. Clearly, one cannot conclude a psychogenic etiology on this basis. Van Houdenhove (1986) presented a retrospective review of 32 patients “referred for psychiatric assessment because persistent pain complaints were thought to be discrepant with demonstrable organic etiology or aggravated by psychological factors”. The author examines this population, with no control group, and concludes that psychological factors, particularly painful affective loss, are important in the etiology of neuroalgodystrophy. He does state that the findings should be interpreted cautiously because of the retrospective character of the study, the psychiatric bias, and the lack of a control group. Certainly, these issues severely compromise any inferences one can make regarding causality. Adler et al. (19591 compared a group of 12 patients with RSD to a control group of 12 patients who did not have RSD but who were said to have “the same, or almost the same, antecedentia”. A battery of tests was administered. These included Raven’s matrices, subtests of the Wechsler Bellevue Scale, the Bender Gestalt and the Zulliger Plates. The authors state that results revealed differences in intelligence, productiv-

343

ity, utilization of intellectual power, affect, aggressiveness, social adaptation and ego strength. From these results, the authors conclude that psychological factors were important in determining the development of RSD. They propose that this is a “psychosomatic disorder”. The design of this study does not allow the authors to rule out the possibility that the differences found on these tests may have been the result, rather than a contributing cause of RSD. Other concerns include the use of projective tests to make inferences regarding personality and psychological functioning, the possibility that other differences noted may have been a result of the group difference in intelligence, and the fact that aggressiveness and social adaptation were not statistically significant. Pollack et al. (1980) administered psychometric tests to 2 groups of patients. A group of 40 patients who had fractured the radius and developed Sudeck’s dystrophy and aVcontrol group of 20 patients who had fractured the radius but had not developed Sudeck’s. Freiburgs Personality Inventory and the questionnaire for anxiety after Spreen were administered to all patients. Two psychological types were identified in the test group. The authors defined these as “Sudeck A” - characterized by increased anxiety, emotional lability, a tendency toward depression and the presence of psychosomatic symptoms - and “Sudeck B” - characterized by increases in self-confidence, a tendency to lie and extraversion. The Sudeck A group was said to differ significantly from the control group. In the discussion, the authors conclude that the data support the importance of a factor of the ‘psyche’ in the development of Sudeck’s dystrophy. They state that patients of the Sudeck A type are at risk of developing Sudeck’s dystrophy. The design of this study does not allow one to rule out the alternate hypothesis that these findings of increased anxiety, emotional lability, depression and presence of psychosomatic symptoms are a result rather than a cause of RSD. All patients in the test group were already suffering from Sudeck’s dystrophy upon entry into the study. DeLeo and Magni (1983) administered Eysenck’s Personality Inventory to a group of 39 patients who had developed Sudeck’s dystrophy after trauma and compared the scores to those of a control group of 35 patients who suffered a similar traumatic fracture but who had not developed Sudeck’s. Results indicated that the Sudeck’s patients were more introverted, but there was no difference between groups in their tendency to develop neurosis or psychosis. The authors do not suggest that this supports a psychogenic etiology. They state that the strongly introverted personality of these patients leads to isolation and insufficient management of the traumatic event. This paper has been quoted as having confirmed Pollack’s definition of type A (Egle and Hoffmann 1990).

Langston and Cowan (1955) presented a review of 88 patients who were followed at 5 years by interview or questionnaire following surgery for Dupuyten’s contracture. In their discussion, the authors stated, “In our personal experience, the type of patient who gives the most trouble post-operatively is the high-pressure businessman, often overweight, with a flabby palm, poor muscle tone and a tense nervous system. In such a patient, sympathetic dystrophy almost invariably develops, prolonging the time needed for recovery”. No detail is provided to substantiate this claim. There is no indication that a psychological evaluation was done. There is no mention of the percentage or number of patients who developed RSD. There is no detailed information showing an association between the development of RSD and the psychological or physical aspects described above. This study is quoted as showing “a clearly defined predisposed personality whose most pronounced characteristics are hypertension and a tense nervous system” (Egle and Hoffmann 1990). Egle and Hoffmann (1990) presented 8 patients with RSD who were described to have undergone “extensive clinical examination and comprehensive psychological testing”. It was stated that the personal history “showed that at the time of trauma or operation and the subsequent onset of RSD, all patients were going through what was described as an extraordinarily difficult period in their lives”. Five of 8 patients were said to display character traits attributed to type A by Pollack, and 3 out of 8 type B. Four of 8 patients had spent the first years of life in an orphanage or with foster parents. The remaining 4 had experienced a difficult childhood according to criteria established by Engel in his pain-proneness study (Engle 1959). Details regarding the psychological testing are not presented. The author states, “The factor causing the disease (RSD) and determining its clinical course is, therefore, not the intensity of objective trauma, but a narcissistic reaction and subsequent depressive withdrawal marked by inertia and self-indulgence or unconsciously accusing/appealing behaviour”. There is no mention as to how this group of 8 patients was selected and there was no control group - issues of importance when attempting to make inferences regarding causation. An interesting study by Zachariae (1964) described the incidence and course of post-traumatic dystrophy following an operation for Dupuytren’s contracture. This study is the only attempt at a prospective design to date. Pre-operatively, 47 of the patients were assessed by a psychiatrist “who assessed their character and gave a statement containing a presumption regarding the postoperative course”. The surgeon was blind to the psychiatrist’s assessment. Patients were followed post-operatively and then the predicted course was compared to the actual post-operative course. According to the psychiatrist, 32 patients were predicted to go

on to an uncomplicated postoperative course. In 10 patients “the psychiatrist did find traits which might be imagined to give rise to post-operative complications, but these patients were not believed to possess sufficient strength of character to maintain this state for any length of time”. In 8 of these, the development of oedema, fibrosis and stiffness was said to be “fairly prolonged” but restored in approximately 3 months. Two experienced an uneventful postoperative course. In 5 cases it was stated that the psychiatrist expected a serious risk of protracted postoperative complications, possibly with permanent sequelae. Closer reading reveals that 1 of these 5 was not expected to develop postoperative complications but did develop a protracted course of oedema, fibrosis and stiffness for 4 months. Two of 5 patients did not develop complications and, in fact, were described as “remarkably energetic” in training the hand and as having a “remarkably short postoperative course”. One patient, described as “aggressive, aggrieved and self-opinionated” did develop a “full-blown state of dystrophy and 4 months after the operation the hand was not fully restored”. A fifth patient was described as a “bitter, dissatisfied, self-centered person with hysteriform traits, so he was expected to develop dystrophy”. It was stated that “his postoperative complications were accurately localized to the operated finger which was stiff and swollen.. . . it was 4 months before he had recovered”. There is no mention of dystrophy in this patient. There is no description as to whether an attempt was made to measure psychological variables systematically. There arc no statistical tests of significance as to the accuracy of predictions made by the psychiatrist. The statement that there was “close conformity between the postoperative course expected by the psychiatrist and the actual course” remains unsubstantiated. The author concluded, that “Although mental factors are probably not the sole explanation, they no doubt constitute an important detail in the syndrome”. This study is quoted as showing that “potential RSD patients are apparently characterized by aggressive inhibition, lack of self-assurance, self-absorption, self-pity and hysterical personality traits” and supports a “psychogenic aspect” (Egle and Hoffmann 1990). Omer and Thomas (1971) make the unsubstantiated claim that the mood and general affect of causalgia patients is most often manic. Lidz and Payne (1945) present a case report in a soldier diagnosed with causalgia who was said to respond to psychotherapy. Closer reading reveals that treatment also included encouragement to increase the use of the hand and sodium pentothal narcosis. This paper raises the same concerns discussed previously.

Summary

of the adult literature

In summary, there are 29 reports in the adult literature containing original data in which there is some discussion of the psychological aspects of sympathetically related pain syndromes. The majority agree that this type of pain can be associated with profound emotional and behavioural consequences. When trying to draw conclusions beyond this, there are many problems. The populations in the studies reviewed here are heterogenous. The symptom complex is identified by many different names. When diagnostic criteria are presented, they are not consistent between studies. In looking at psychological issues, there are further problems. Behavioural criteria are not clearly defined. Often, psychiatric and psychological terminology are used incorrectly. Most of these studies had as their main focus something other than psychological aspects. There are few objective measures of psychological constructs. Control groups and statistical tests of significance are often lacking. As discussed by Haddox (I 989), “Unsupported statements by established authorities have occurred more frequently than have studies designed to address these issues”. Overall, this is inadequate data when trying to make inferences regarding causality. There are, however, large series of case descriptions written by clinicians with a wealth of experience. In every case, these authors were impressed by the profound suffering when patients were in pain and the equally profound behavioural change when there was relief from pain. It was the impression of the majority of clinicians that the emotional and behavioural changes were a result of pain and that there is no evidence to support a predisposing personality leading to RSD. The papers supporting a psychological etiology (Litz and Payne 1945; Langston and Cowan 1955; Adler et al. 1959; Zachariae 1964; Pollack et al. 1980; Van Houdenhove 1986; Egle and Hoffmann 1990) were limited methodologically for reasons discussed. Many interesting questions have been raised in these papers, Are there psychological factors that are important in influencing the progression and severity of the disease as suggested by Miller and DeTakats (1942) and others (Poplawski et al. 1983; Schott 1986; Schwartzman and McLellan 1987)? Schott (19861, in a review, described the importance of psychological interactions in causalgia. He stated, “just as causalgia can be worsened by psychological and other central factors, so alleviation can occur by psychological means”. This has not been studied in a controlled way. Is there a factor other than personality that predis-

345

poses one to develop RSD? Several authors have proposed an autonomic reactivity as a predisposing factor (Miller and DeTakats 1942; Owens 1957; Poplawski et al. 1983). One could speculate that psychological factors are important when considering response to treatment. These questions and others await proper scientific study.

Psychological in children

aspects

of reflex sympathetic

dystrophy

There is a small body of literature describing RSD in children. Those papers containing information regarding psychosocial aspects will be reviewed below. Matles (1971) reported a case of RSD in a child. The management plan included sympathetic blocks and referral for psychiatric assessment. It was noted that 1 day prior to psychiatric consultation and 2 days prior to the blocks, the patient’s symptoms subsided. The psychiatric diagnosis was said to be “psychosomatic reaction to psychic stress”. It was later concluded that “delay in implicating the psychiatric component in this patient’s condition contributed to her length of disability”. No evidence is presented to substantiate the author’s conclusion. Fermaglich (1977) described 2 cases of RSD in children. No details regarding the psychosocial aspects of the history are presented. The author states that in certain individuals “psychotherapy may be indicated as an adjunct form of therapy”. This article is mentioned as it has been cited by other authors when discussing the “psychogenic aspects” of RSD in children (Ruggeri et al. 1982). Forster and Fu (1985) presented a case report of RSD in a lo-year-old girl. The only comment made regarding the social history was that “social history was unremarkable with the exception that the patient had been maintained from a very early age on a strict vegetarian diet that excluded all poultry, fish, and meat”. The authors later state, “children have also been found to respond much more rapidly to physical therapy and psychological counselling” but present no evidence of their own to substantiate this statement. There is nothing in the case presented to suggest that psychological counselling was a part of the patient’s management. Ruggeri et al. (1982) described 6 cases of RSD in children. The authors note that psychological evaluation in 1 child revealed an unstable home environment. Another child was said to be concerned with her body image. The authors later state, “the psychogenic aspects of this syndrome in children are important” but do not present any evidence to substantiate this claim. Indeed, the articles they cite as supporting this statement (Casten and Betcher 19.55; Matles 1971; Fer-

maglich 1977; Bernstein et al. 1978) either have their own methodological problems (Matles 1971; Fermaglich 1977; Bernstein et al. 1978) or imply the opposite (Casten and Betcher 1955). Bernstein et al. (1978) presented a series of 23 children diagnosed with reflex neurovascular dystrophy. Psychological consultation was obtained in 17 of these children. No attempt was made to impose a fixed protocol or interview schedule. It was said that review of the psychological consultation records indicated certain trends in their backgrounds. The authors described a history of overt parental conflict in 10 out of 12 families. Data was said to be insufficient for the remaining 5 of these 17 children, Eleven of 13 were said to have a “marked tendency” to accept responsibility beyond their years. Difficulty in expressing anger or being assertive on their own behalf was evident in 9 of 10 children. The authors later conclude that “certain personality traits may have been significant predisposing or contributing factors”. They speculate that “illness offered the children a safe means of frustrating their Parents’ demands for performance without having to take responsibility for their behaviour”. These observations are valuable and interesting, but caution is needed when making inferences regarding causality. It is not explained why only 17 children received psychological evaluation. The authors make statements regarding 10 of 12 children in this group of 17 (see above) when, in reality, it is 10 of 23 children in the entire group that are being considered; this is confusing. Other problems include the subjective nature of ‘personality traits’ with no fixed protocol or interview schedule, the lack of blinding of the psychological evaluators, and the lack of a control group. This article has been cited by others as evidence for a psychological etiology in RSD in children (Ruggeri et al. 1981). Sherry and Weisman (1988) reported on 21 families with children who have been diagnosed with reflex neurovascular dystrophy. All patients and parents were interviewed by a psychologist. Other measures included the Family Environment Scale, the Wechsler Intelligence Scale for Children-Revised, the Child Behaviour Checklist, and the Wide-Range Achievement Test-Revised. Seven patients completed the Brief Symptom Inventory, and 15 patients and their mothers identified the patients’ overall global state of health using a visual analog scale. The authors summarized their results as follows: two distinct family patterns were identified based on information from the Family Environment Scale. Fifteen families were said to exhibit high cohesion, expression and organization with low-to-average conflict. Six families were said to have low levels of cohesion, marked conflict and below average expressiveness and organization. All families were said to exhibit parental enmeshment. Other prominent fea-

tures included marital discord in 12 families, significant school problems in 13 children, a history of sexual abuse in 4 children, and possible role models with similar symptoms in 10 children. All patients experienced multiple stressful events within several months prior to the onset of reflex neurovascular dystrophy. The authors later conclude, “reflex neurovascular dystrophy in children is almost always associated with significant underlying psychologic stress”. They state, “these data support the concept that childhood reflex neurovascular dystrophy is frequently a stress-related disease”. As with the previous studies described, one must be cautious in interpreting these results. There is no control group. It is possible that the family patterns described may be present in many children with chronic pain of any type or, for that matter, any type of chronic illness. The same may be said about school problems. Indeed, school problems may be the result of chronic pain or illness. Similarly, the presence of role models exhibiting pain behaviour and a history of sexual abuse are not specific to reflex neurovascular dystrophy. The conclusion that this disorder is a stress-related disease appears unfounded.

Summary

of paediatric

literature

The concerns raised when reviewing the adult literature also hold when considering the paediatric population. The information available is useful and interesting; however, the data are insufficient for inferences regarding causality. There are no control groups; often, psychological and behavioural criteria are not clearly defined or systematically applied; there is no blinding of psychological evaluators, and there are no statistical tests of significance. Confusing statements are made, conclusions regarding a psychogenic etiology are unfounded, and misinformation is perpetuated through uncritical citation of others’ work.

Overall summary

and conclusions

In summary, there is general agreement that the behavioural and emotional issues in patients suffering from RSD are important and can be profound. However, a complete review of the literature from the late 1800s to the present reveals no worthwhile evidence to substantiate the claim that psychological factors or certain personality traits predispose one to develop RSD. Several papers have discussed the importance of psychological factors in increasing suffering related to causalgia (Miller and DeTakats 1942; Poplawski et al. 1983; Schott 1986; Schwartzman and McLellan 1990)

and have raised other interesting questions regarding psychological issues in RSD. Further study incorporating the suggestions for research recently elaborated by the Special Interest Group on Pain and the Sympathetic Nervous System is needed.

Acknowledgements

The author would like to thank Dr. W.O. McCormick, Dr. I. Purkis, and Dr. H. Merskey for advice in preparation of the manuscript; Dr. C. Hault for her kind assistance in translating the German papers; Mrs. Shelagh Parker for typing; and Ms. Beverly Quigley for her assistance in obtaining papers from foreign centres.

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