CRPS also attempt to explain the development of CRPS-dystonia ..... cal factors in the development of reflex sympathetic dystrophy: a ... Swift DW, Walker SE.
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Journal of Pain and Symptom Management
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Original Article
The Symptom Checklist-90 Revised Questionnaire: No Psychological Profiles in Complex Regional Pain Syndrome–Dystonia Lijckle van der Laan, MD, PhD, Karel van Spaendonck, PhD, Martin W.I.M. Horstink, MD, PhD, and R. Jan A. Goris, MD, PhD Departments of Surgery (L.v.L., R.J.A.G.), Medical Psychology (K.v.S.), and Neurology (M.W.I.M.H.), University Hospital Nijmegen, Nijmegen, The Netherlands
Abstract Complex regional pain syndrome (CRPS) is a syndrome usually localized in the extremities, mostly occurring after a preceding trauma or operation. Dystonia is present in a minority of CRPS patients, but, when present, leads to severe disability. Various pathological factors have been postulated to present in CRPS-dystonia, such as involvement of the sympathetic system, reorganization of the central nervous system, and psychological distress. In the present study, we investigated the involvement of psychological distress in CRPS-dystonia with the aid of the Symptom Checklist-90 Revised (SCL-90R) questionnaire. The SCL-90R is a multidimensional self-report inventory covering various dimensions of psychological distress. In a population of 1006 CRPS patients, we analyzed the SCL-90R scores of 27 patients with CRPS-dystonia (23 female and 4 male) and compared the scores to sample scores of a control female (n 5 577) and a control rehabilitation population (n 5 56). Insomnia scored significantly higher in the female CRPS-dystonia population, as compared to the control female population (P , 0.001), and in the total CRPS-dystonia population, as compared to the rehabilitation population (P ,0.01). Remarkable was the significantly higher score of somatization in the rehabilitation population, as compared to the CRPS-dystonia population (P 5 0.006). For the other dimensions of psychological distress of the SCL-90R, the scores of the CRPS-dystonia and control populations were similar. With regard to the SCL-90R scores, we conclude that specific psychological profiles are not present in CRPS-dystonia. J Pain Symptom Manage 1999;17:357–362. © U.S. Cancer Pain Relief Committee, 1999. Key Words Reflex sympathetic dystrophy, dystonia, psychopathology, Symptom Checklist-90 Revised, psychology, complex regional pain syndrome
Introduction
Address reprint requests to: Lijckle van der Laan, MD, PhD, Department of Surgery, University Hospital Nijmegen, PO Box 9101, 6500 HB Nijmegen, The Netherlands. Accepted for publication: August 3, 1998. © U.S. Cancer Pain Relief Committee, 1999 Published by Elsevier, New York, New York
Complex regional pain syndrome (CRPS) is a syndrome most often localized in the extremities. The major causes of CRPS are a preceding trauma or operation of the affected extremity.1 CRPS is characterized by alterations in skin color and skin temperature, edema, impaired active range of motion, and increased 0885-3924/99/$–see front matter PII S0885-3924(99)00009-3
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pain sensations. The disease is frequently accompanied by various neurological complaints or signs, such as hypesthesia, hyperalgesia, paresis, tremors, incoordination, and muscle spasms.1,2 In a minority of CRPS patients, the syndrome is accompanied by abnormal movements and postures, such as dystonia and myoclonus.3–6 These complications of CRPS are mostly resistant to any therapy and may lead to severe disability of the patient. Up to now, there has been no consensus about the pathogenesis either of the CRPS or the dystonia-like postural abnormalities (CRPSdystonia). The most widely accepted hypotheses involve a disturbed hyper- or hypoactive reaction of the sympathetic system,7–10 and an exaggerated (neuro)inflammatory response1,11,12 in the injured extremity. Although some authors argue that a psychological predisposition is involved in CRPS,13,14 there is no direct evidence in support of this hypothesis.15–17 Some hypotheses applied to the pathogenesis of CRPS also attempt to explain the development of CRPS-dystonia, namely interaction of the neuropeptide substance P in the sympathetic system, reorganization of the central nervous system, and psychological distress of the patient.5,6 To our knowledge, the possible involvement of psychological distress in the presentation of CRPS-dystonia has never been investigated. The aim of the present study is to evaluate whether psychological distress is involved in CRPS patients who developed CRPS-dystonia. For this purpose, we studied a subgroup of CRPS-dystonia patients belonging to our welldocumented population of 1006 CRPS patients.
Methods At the outpatient clinic of the Department of Surgery, University Hospital Nijmegen, CRPS was diagnosed in 1006 patients, according to the criteria of Veldman et al.,1 during the period of January 1, 1984 to January 31, 1996. The majority (90%) of the CRPS patients visited our department only once for a second opinion or for therapeutic advice. Within this population, CRPS-dystonia was present in 41 CRPS patients (4% of the 1006 CRPS patients). CRPS-dystonia was defined as a fixed abnormal posture of the affected extremity, such as hy-
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perflexion of the wrist and fingers, hyperextension of the fingers, or inversion and plantar flexion or extension of the foot.18 A questionnaire was mailed to all 41 CRPSdystonia patients, with a response from 31 patients. One respondent was excluded from analysis because of missing data. At the time of answering the questionnaire, three patients were cured from CRPS and the CRPS-dystonia. Consequently, we analyzed the data of 27 patients with active CRPS-dystonia. The age at the onset of the disease, gender, initiating cause of CRPS (e.g., trauma, surgery, spontaneous, others), presence of CRPS in another extremity (multiple CRPS) localization (upper or lower extremity), and psychological state of the CRPS-dystonia population (n 5 27) were compared to a population of CRPS patients without severe complications of the affected extremity (n 5 932), including recurrent infections, chronic edema, ulcers, and myoclonus.
Symptom Checklist-90 Revised (SCL-90R) In this study, the Dutch adapted version of the Symptom Checklist-90 Revised (SCL-90R) was used,19 which is very closely related to the original SCL-90 questionnaire of Derogatis.20 The SCL-90R is a multi-dimensional selfreport inventory, consisting of 90 items covering 8 dimensions of psychological distress: phobic anxiety, anxiety, depression, somatization, obsessive-compulsitivity, distrust and interpersonal sensitivity, hostility, and insomnia. The overall total score of the SCL-90R is used to represent psychoneuroticism. Each item describes a physical or psychological symptom that is rated on a five-point scale ranging from 1 (5 not at all) to 5 (5 extremely). The patients were asked to answer the items to indicate the extent to which the symptoms of the SCL-90R were manifest during the week preceding the day of answering. Because of the low number (n 5 4) of male CRPS-dystonia patients, a comparison with the normal male population was not performed. The SCL-90R scores of the female CRPS-dystonia patients were compared to the normative data from a normal female population. This group consisted of persons older than 18 years, randomly selected, and representing the Dutch population with regard to education, work, religion, and age.19 The SCL-90R data for the fe-
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male control group were obtained by mailed questionnaires, performed and analyzed according to Arrindell and Ettema.19 The SCL-90R scores of the total population of CRPS-dystonia patients were compared to data obtained in a population of patients suffering from a somatic disorder requiring rehabilitation treatment at the outpatient clinic of the Department of Rehabilitation of the University Hospital Groningen.19
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itself after a trauma of the affected extremity. In two patients, CRPS developed subsequent to an operation for carpal tunnel syndrome. In 10 patients, CRPS developed in more than one extremity. When a patient had more than one extremity with CRPS-dystonia, the first affected extremity (upper or lower extremity) was used for the localization. In four patients, besides CRPS-dystonia, myoclonus of the CRPS extremity was also present. The age at the onset of the disease, gender, localization and the etiology of the population of CRPS-dystonia was not significantly different, as compared to the CRPS without severe complications (Table 1). Only multiple CRPS was more present in the CRPS-dystonia population, as compared to the CRPS without severe complications (P , 0.01). Of the 10 CRPS patients, one male and nine female, who failed to return the mailed questionnaire, nine had a dystonia of the leg and one had dystonia in an upper extremity. The CRPS developed in six patients after trauma, in three patients after an operation, and in one patient no precipating factor was present. In this population of nonresponders, the age at the onset of the CRPS varied from 18 to 53 years, with a median of 26 years. The SCL-90R scores of the female CRPSdystonia population (n 5 23) were similar to those of the female control group (n 5 577) (Table 2). Only the score for insomnia was significantly higher in the female CRPS-dystonia population, as compared to the female control group (P , 0.001).
Statistical Analysis
The data are expressed as mean 6 standard deviation. Statistical analysis of the SCL-90 questionnaire data was performed by Student’s t -test, two-tailed. The Fisher exact test, two-sided, was performed to compare the CRPS-dystonia population to CRPS without severe complication population. For the age range only, the KruskalWallis test, two-sided, was performed. The level of significance was set at P , 0.05.
Results The patients with CRPS-dystonia included 23 women and 4 men (Table 1). None of these patients had a family history of dystonia or psychiatric problems in the past. The age at the onset of the CRPS varied from 17 to 63 years, with a median of 38 years; age at the time of the study varied from 24 to 70 years, with a median of 46 years. The CRPS-dystonia had been present for 5.1 6 3.9 years. In 55% of the CRPS-dystonia patients, the CRPS manifested
Table 1 Characteristics of CRPS-Dystonia Population versus the CRPS-Population without Complications CRPS-dystonia population %
n
%
P-value
27 17–63 (38) 23 4 12 15 10
85 15 44 56 37
932 10–84 (44) 699 233 578 354 120
75 25 62 38 13
ns ns ns ns ns ,0.01
15 10 1 1
55 37 4 4
621 182 94 35
66 20 10 4
ns ns ns ns
n Total Age at the onset of the CRPS (median) Female Male Upper extremity Lower extremity Multiple CRPS Etiology (%) Trauma Surgery Spontaneous Others ns 5 not significant.
CRPS without severe complications population
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Table 2 SCL-90R Scoresa from a Female Normal Population versus a Female CRPS-Dystonia Population Normal population (n 5 577) Age SCL-90R score Phobic anxiety Anxiety Depression Somatization Obsessive-compulsive Sensitivity Hostility Insomnia Psychoneuroticism aValues
CRPS-dystonia (n 5 23)
43.3 6 14.2
42.3 6 11.9
8.7 6 3.4 14.6 6 5.7 23.8 6 8.6 18.7 6 7.1 14.1 6 5.1 26.3 6 8.8 7.6 6 2.4 5.2 6 2.8 128.9 6 36.4
8.5 6 2.7 12.9 6 4.3 23.2 6 8.4 21.6 6 7.2 15.4 6 5.5 23.8 6 6.3 7.1 6 1.8 7.7 6 4.4 131.1 6 34.1
P value
,0.001
are mean 6 SD.
In the total population of CRPS-dystonia (n 5 27), the insomnia score was also significantly higher as compared to the rehabilitation population (n 5 56) (P , 0.01) (Table 3). A significantly higher score for somatization was found in the rehabilitation population, as compared to the population of CRPS-dystonia (P 5 0.006). For the other dimensions of psychological distress of the SCL-90R, the scores of the two populations were similar.
Discussion The main finding of the present study is that the SCL-90R questionnaire did not yield evidence for a psychogenic factor involved in the presentation of CRPS-dystonia. In fact, the somatization score in the control rehabilitation
patient group was significantly higher than in the CRPS-dystonia population. All other SCL90R scores were statistically similar. It is generally argued that a psychogenic cause of dystonia has to be suspected when the clinical characteristics over time are incongruous or inconsistent with established forms of dystonia. Fahn and Williams,21 and Lang22 enumerate a number of clinical features that help to distinguish psychogenic dystonia from established causes of dystonia. For example, precipitating factors (e.g., injury), onset with resting dystonia, and the presence of fixed spasms all suggest psychogenic dystonia, whereas spontaneous appearance, action dystonia, and mobile spasms are more typically seen in idiopathic dystonia. Furthermore, a rapid progression and spread to other parts of the body suggests
Table 3 SCL-90R Scoresa from Outpatient Clinic Rehabilitation Patients, and CRPS-dystonia Patients
Age Female (%) Male (%) SCL-90R score Phobic anxiety Anxiety Depression Somatization Obsessive-compulsive Sensitivity Hostility Insomnia Psychoneuroticism aValues
are mean 6 SD.
Rehabilitation (n 5 56)
CRPS-dystonia (n 5 27)
33.9 6 11.9 59 41
44.1 6 11.9 85 15
9.5 6 4.4 15.6 6 7.2 26.1 6 11.7 26.5 6 7.4 16.8 6 6.2 25.7 6 9.2 8.0 6 3.0 5.8 6 2.7 141.5 6 44.7
8.9 6 3.3 16.5 6 4.4 23.6 6 8.3 21.7 6 6.8 16.2 6 5.6 23.9 6 6.1 7.1 6 1.7 7.8 6 4.2 133.6 6 33.3
P value
50.006
,0.01
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a psychogenic involvement. Counter pressure on attempted movement is typically seen in idiopathic dystonia. Pain, especially pain appearing in the extremity before dystonia, or pain at onset, is thought to favor a psychogenic cause. According to these rules, it is readily apparent that the movement disorder in patients with CRPS is clearly incongruous with the classical idiopathic dystonia of central nervous system origin.3,23 Most of the distinguishing features discussed above, however, are insufficient on their own to justify a psychogenic profile in CRPS-dystonia. Features present in CRPS patients are also present in so-called dystonia following peripheral injury.24,25 Both CRPS patients and patients with peripherally induced movement disorders have a preceding peripheral injury or an apparently spontaneous causalgia in common. In most of the patients with peripheral injury, the spasms are present at rest and are typically sustained, producing a fixed abnormal posture. The disorder also shows a rapid progression. Thus, although the incongruities with classical dystonia indicate that the movement disorder in CRPS differs from classical dystonia, this does not mean that the cause must be psychological. The studies mentioned above strongly favor the idea that abnormal postures and movements may occur after peripheral injury and in patients with peripherally induced pain syndromes. It is much more likely that CRPS is related to these peripherally induced syndromes with which they are congruous and consistent over time, rather than to classical dystonia of central nervous system origin. Bhatia et al. concluded from their analysis of a population of 18 CRPS-dystonia patients (16 female) that two pathophysiological mechanisms might be involved in dystonia6: (1) a functional neurological illness or (2) psychological distress. This psychological distress hypothesis is mainly supported by the finding that, in various studies, women are more prone to develop CRPS-dystonia.3,6,26 In the present study, the majority of the population of CRPSdystonia patients also consisted of women. From this finding, however, it cannot be concluded that a psychogenic origin plays a role in the pathogenesis of dystonia, as in the population of 1006 RSD patients, 76% of the patients are also female.1 Another weak argument for
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the involvement of psychogenic profile is derived from the fact that in some CRPS-dystonia patients, the abnormal posture of the hand resembles the clenched fist syndrome,3 which is sometimes thought to be a conversion disorder.27,28 We examined our patients with CRPS-dystonia with the SCL-90R, which is a generally accepted questionnaire for the detection of various psychological abnormalities, such as anxiety, depression, somatization, or psychoneuroticism.17,19,20 Comparison of the SCL-90R score of the CRPS-dystonia population with control groups detected no difference in psychological distress between these groups. Remarkably, the rehabilitation population had significantly more somatization than the CRPSdystonia population. Patients with CRPS-dystonia suffered significantly more from insomnia, which may be explained by the presence of severe pain accompanying CRPS. In the present population of patients suffering from CRPS-dystonia, psychological distress could not be established by the SCL-90R questionnaire. Our findings are consistent with several prior reports, which failed to find direct evidence in favor of a psychogenic cause in patients with comparable syndromes. For instance, none of the patients of Bhatia et al.6 exhibited conversion symptoms or overt psychological disorders, and treatment with psychotherapy failed to improve the patients symptoms.6,22,26 These studies, together with our study, suggest that CRPS patients may develop painful and abnormal postures without the presence of specific psychological profiles. They further suggest that the movement disorders may result from peripherally induced abnormal sensorimotor control.
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