Pain, 55 (1993) 393-396 0 1993 Elsevier Science Publishers B.V. All rights reserved 0304-3959/93/$06.00

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PAIN 02443

Reflex sympathetic dystrophy treated by electroconvulsive therapy: intractable pain, depression, and bilateral electrode ECT

John H. King * and Shelley Nuss

Department of Behavioral Medicine and Psychiatry, West Virginia University School of Medicine, Morgantown, WV26506 (USA)

(Received 12 May 1993, revision received and accepted 11 August 1993)

Summary An adult female patient without previous history of pain or psychiatric disorder developed reflex sympathetic dystrophy (RSD) in her left hand and arm after 2 separate injuries, and subsequent arthroscopy and arthroscopic surgery. Traditional management with systemic medications, nerve blocks, and behavioral pain management were unsuccessful. With subsequent development of depression and suicide attempt, electroconvulsive therapy (ECT) was administered using bilateral lead placement. Total resolution of her pain occurred for several hours after the first treatment. Over the course of the full ECT series the pain, vasculature changes of RSD, and depression resolved completely. Prominent psychiatric disturbances were complications arising after ECT and the resolution of the RSD, and during long-term follow-up. A review of the literature from the 1940s revealed numerous cases of chronic, intractable pain treated successfully by ECT, although none of the case series were controlled studies. More recent literature questions if ‘modified’ ECT, using unilateral lead placement, is less effective than bilateral lead placement, in treating pain syndromes. Resolution of the vascular changes of RSD after ECT raises questions of possible cerebral contributions to the pathophysiology of RSD.

Key words: Reflex sympathetic dystrophy; Electroconvulsive therapy; Pain, intractable; Affective disorder; Therapy; Case report; Review of reported cases

Case report

A 32-year-old single Caucasian female presented for her first psychiatric hospital admission, referred by the Anesthesia Pain Clinic for evaluation and treatment of depression associated with chronic left arm pain and disability from reflex sympathetic dystrophy (RSD).

Her problems began after she suffered a fall on ice in December of 1991 and sustained a fracture of her left wrist which was casted for a short time. In June of 1992, she re-injured her left hand by accidentally slamming it in a car door, and again, it was casted. After the cast was removed, she continued to have pain and partial loss of function of her left arm. She was examined by an orthopedic surgeon in October of 1992 and an arthrogram revealed torn cartilage and inflamed ligaments. Arthroscopic repair was performed and there- after her arm began to show earlier signs of RSD. She was referred to the anesthesia pain clinic where stellate ganglion blocks as well as amitriptyline were tried without relief. The left arm pain progressed to extreme levels and was accompanied by weakness, piloerection,

* Corresponding author: Dr. Ring, Department of Behavioral Medicine and Psychiatry, West Virginia University School of Medicine, 930 Chestnut Ridge Road, Morgantown, WV 26506, USA. Tel.: (l-304) 293-2411; FAX: (l-304) 293-5555.

swelling, decreased range of motion and function, discoloration and hyperesthesia. She became more depressed and disabled from per- forming her most important activities, such as participating in com- petitive sports and working at a local department store.

Her past medical history was remarkable only for hypertension, and she had no past psychiatric history and no history of depression. History of substance abuse was negative, including nicotine and caffeine.

Upon administration to the psychiatric hospital, she had a some- what masculine appearance with a large frame, mild hirsuitism and moderate, generalized thinning of her hair. She was noted to be very downcast with extreme sadness. She felt her life was hopeless and worthless. Her sleep and appetite were disturbed with significant weight loss. She was guarding and holding her left arm to her side, flexed at 90” at the elbow, and refused to allow it to be touched. The arm was swollen and the skin was dry and scaly with marked piloerection, and a pink to purple discoloration. The examiner gained permission to lightly touch the arm, which was found to be cool and hyperesthetic. Motor function and deep tendon reflexes were impos- sible to accurately evaluate, but mild atrophy was noted.

Initial multidisciplinary management included behavioral pain management, antidepressants, and numerous nerve block proce- dures. All of these interventions were without significant effect and her symptoms worsened. Several weeks into the hospitalization, she attempted suicide by suffocation and was transferred to the locked

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psychiatric intensive care unit. She remained actively suicidal. The pain continued to intensify, such that a bed sheet or a draft of air crossing it was painful. Electroconvulsive therapy (ECT) was consid- ered for depression unresponsive to medications.

The patient received ECI’ with bilateral, bitemporal electrode lead placement, which contradicts the American Psychiatric Associa- tion Tusk Force Report on ECT, but is consonant with an authorita- tive report in the literature (Abrams and Taylor 1976). The results of her first ECT were quite remarkable. Upon awakening from anesthe- sia, during the period of post-ictal confusion and for the next 12 h, her arm was totally free of pain with normal function, and the vascular discoloration subsided significantly. No limitations of use or movement could be elicited. This improvement faded during the second 12 h. Over the course of 7 subsequent ECT treatments, the improvement in pain and vascular features persisted for longer and longer periods, until both resolved completely. Depressive symptoms resolved simultaneously with pain symptoms and vascular changes. By the end of the course of treatment, there was no complaint of pain whatsoever, and the skin of her arm had returned to normal temperature and appearance. No piloerection, blanching, erythema or edema was observed. She demonstrated unrestricted range of motion and use of the arm. Follow-up 6 months later indicated that the patient was no longer in treatment for her RSD. She had noted episodic return of mild symptoms of arm pain, but she was “much better” and had no limitation of movement or function whatsoever. However, she remained in treatment for depression and she de- scribed multiple somatic complaints, such as visual acuity problems, seeing double, and feeling dizzy. She also described a number of severe phobias, and she had been unable to return to work. She spent most of her time in the back room of her parents’ retail store, answering the telephone and generally staying out of sight of other people.

Discussion

Review of the available English literature for the past 43 years revealed no previous cases of response of RSD to ECT. However, the terminology of ‘electrocon- vulsive therapy’ and ‘reflex sympathetic dystrophy’ were not accepted terms at the onset of this period.

The term ‘reflex sympathetic dystrophy’ or RSD has been generally accepted rather recently. For many years the syndrome was grouped with others related to physical trauma, under the names of ‘Sudeck’s atrophy’, ‘causalgia’, and ‘degenerative osteoporosis’. Similarly, terminology descriptive of ECT in the 1950s included ‘shock therapy’, ‘electric shock therapy’, ‘electronarco- sis’, and ‘electroconvulsive therapy’. These changes presented difficulties in tracing the literature for ECT used in RSD.

The literature indicated that ECT was used effec- tively during the 1940s and 1950s for treatment of numerous syndromes of intractable pain (Pisetsky 1945; Boyd 1956; Annotations 1959; Bradley 1963). However, several factors nearly abolished the practice. ECT per- formed in that day, unmodified by muscular paralysis, anesthesia and unilateral placement of electrodes, pro- duced unacceptably high levels of morbidity and mor- tality. The use of psychotropic medications flourished after 1955, and medications were much safer to use in

the treatment of severe psychiatric disturbances. Modi- fied ECT was in its infancy at the time, but the ground swell of psychopharmacological treatment nearly over- whelmed the use of ECT to the point of extinction.

Modified ECT became the standard, when used at all, but while the use of unilateral electrode placement over the non-dominant frontal lobe reduced confusion, it may have resulted in lower response rates of in- tractable pain to ECT and subsequent abandonment of the procedure for pain syndromes (Mandell 1975; Salmon et al. 1988).

During the heyday of ECT of the 1940s and 1950s many case reports described ECT as effective in treat- ing intractable pain. We were able to find one case that bore a striking resemblance to our own. Von Hagen (1957) described a patient who was treated for a lacer- ated finger. The wound was ultimately explored, and a lacerated tendon was discovered and repaired. ‘Causalgia’ developed, which was described as “red- ness and coldness of the finger, wasting and weakness of muscles of the hand, and pain which at times radi- ated up to the shoulder.” Stellate block and a sympa- thectomy were initially successful; however, the pa- tient’s pain recurred, she became addicted to narcotics, and depression and anxiety became prominent. Twelve ECT treatments over 4 weeks resulted in a dramatic reduction of pain. Electrode lead placement was not described, but in that day bilateral, bitemporal place- ment was standard. A recurrence of the pain with agitation and depression was managed with 5 addi- tional outpatient shock treatments that produced com- plete recovery.

Von Hagen’s case seemed to meet the criteria for RSD and was very similar to our own. Depression was prominent in our case, as it was in pain patients who responded to ECT. Depression and RSD resolved si- multaneously with treatment by ECT.

Some dissimilarities in our case deserve mention. At various times before the ECT treatment and later, prior to discharge, the patient was noted to demon- strate astasia abasia, an ataxia usually associated as a conversion or hysterical reaction. This cleared tem- porarily during the course of ECT, but recurred, until the day of discharge, when she was confronted for malingering by the staff who had seen her walking normally when she was unaware of being observed.

After the ECT series and the resolution of her RSD, an amytal interview was performed. The patient demonstrated a dense, partial amnesia for her injuries with great difficulty remembering the events. She also described a violent rape at knife point, for which she had also been partially amnestic, as well as an exagger- ated fear at the time of her injuries that amputation of her arm would be necessary. The loss of an arm was important because that would greatly restrict her pas- sion for sports, including “...tackle football! I like to

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see the surprised expressions on the guys’ faces when I knock them on their ass!” Subsequently psychotherapy revealed intense issues of dependency and conflict within her family. We have described the longer term psychiatric sequelae above.

While we acknowledge the element of malingering that accompanied this case, we feel assured that the RSD was present as a physical syndrome, documented numerous times by those experienced with manage- ment of these patients. We are unaware of a method to reproduce the syndrome of RSD factitiously.

As psychiatrists, we are interested in the etiology and pathophysiology of RSD in patients where the vascular changes present along with prominent psychi- atric presentations. Anesthesiologists and neurologists have traditionally assumed that pathological processes in RSD must reside somewhere between the peripheral site of the nerve injury, along the course of the nerve, to the level of the spinal cord. These mechanism(s) are thought to include the sensory and autonomic motor reflex pathway loops. In our case, and in the literature from the 1940s and 1950s higher central nervous sys- tem modulations are suggested in the pathophysiology of RSD and intractable pain. We summarize the fol- lowing information as supporting such possibilities.

(1) The older literature, cited here, documents that ECT, frontal lobotomies, cingulotomies, and leuco- tomies were indicated for treatment of chronic pain, as well as psychiatric disorders (Pisetsky 1945; Freeman and Watts 1946; Grantham and Spurling 1953; Walker 1955). Thalamic lesions are contemporary neurosurgi- cal interventions.

(2) Studies using unilateral ECT for the treatment of chronic pain did not produce robust results (Mandell 1975; Salmon et al. 19881, but older reports described success when using bilateral treatments. Bilateral ECT has been proposed to be more effective for depression because of the passage of the electrical impulse through the diencephalon, including the thalamus and hypotha- lamus (Abrams and Taylor 1976; Salmon et al. 1988). These structures are pathways for pain sensations and perception.

(3) Direct local effects of the electrical current from the ECT on cortical tissue (Croft 1952) are acknowl- edged. A case has been described, where temporary blindness was produced after ECT was administered with electrodes experimentally placed over the visual cortex of the occipital poles (personal communication, M. Fink, 1990). These effects suggest an acute effect of ECT on cerebral cortex.

(4) Massive quantities of neurotransmitters are re- leased into the bloodstream during the convulsion pro- duced by the electrical current. These neurotransmit- ters circulate throughout the vascular system during the ‘catecholamine phase’ of ECT (Allen 1974; Hola- day 1986; Salmon et al. 1988). Acute changes of auto-

nomic function are characteristic of this phase as well. The effects of such a pulse of neurotransmitters, re- peated over the course of a series of ECT treatments, could modulate neural function at numerous sites, throughout the nervous system generally, and within the autonomic nervous system and smooth muscle of the vasculature specifically. Modulation of opioid activ- ity is also possible, as suggested in the references.

(5) Surgical or pharmacological sympathectomy has been ineffective in treating RSD, where ECT has been effective (see case reports above).

(6) The sympathetic nervous system is highly modu- lated by the limbic cortex, thalamus, and hypothala- mus. This phenomenon is best characterized by the effects of the ‘fight or flight’ response. Such emotional states produce neurohumoral and sympathetic stimula- tion as demonstrated by increases in serum cortisol and catecholamines, and changes in heart rate, blood pres- sure, perspiration and piloerection. Examples such as this demonstrate that the sympathetic system receives modulation from the cortex and the limbic system during emotional arousal.

We are unable to find any literature which provides experimental information specific to these questions. Elucidation of these issues must derive from experi- mental data yet to come. We feel more confident that the important value of this case report resides in the recognition that bilateral ECT should be considered as a treatment option for suicidal, depressed patients with RSD and other intractable pain symdromes. In the case presented, hope had been abandoned by the pa- tient and her treators, and the response to ECT was perceived as life-saving.

References

Abrams, R. and Taylor, M.A., Diencephalic stimulation and the effects of ECT in endogenous depression, Br. J. Psychiat., 129 (1976) 482-485.

Allen, J.P. et al., Corticotrophin release during ECT in man, Am. J. Psychiat., 131 (1974) 1225-1228.

Annotations of the Editors, Shocks for pain, Br. Med. J., 5115 (1959) 161.

Boyd, D.A., Jr., Electroshock therapy in atypical pain syndromes, Lance& 76 (1956) 22-25.

Bradley, J.J., Severe localized pain associated with the depressive syndrome, Br. J. Psychiat., 109 (1963) 741-745.

Croft, P.H.,The effect of electrical stimulation of the brain on the perception of pain, J. Ment. Sci., 98 (1952) 421-426.

Fink, M., Personal communication, Course on the Technique of Electroconuulsiue Therapy, University of Chicago, Chicago, IL, 1990.

Freeman, W. and Watts J.W., Pain of organic disease relieved by prefrontal lobotomy, Lancet, 29 June (1946) 953.

Grantham, E.G. and Spurling, R.G., Selective lobotomy in the treat- ment of intractable pain, Ann. Surg., 137 (1953) 602-608.

Holaday, J.W. et al., Endogenous opioids and their receptors -

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