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Chapter VI PAIN AND RELATED PHENOMENA, INCLUDING CAUSALGIA James C. White and Bertram Selverstone A. INTRODUCTION In the belief that painful phenomena are of intrinsic importance, a separate study was made of their frequency and association with other characteristics, notably those of treatment and other aspects of follow-up status. True causalgia is rare in this series but of sufficient clinical interest to warrant some discussion and the presentation of individual cases. The rest of the chapter is concerned with disagreeable phenomena of lesser importance and greater frequency, e. g., complaints of unpleasant sensations and pain on use or pressure. Causalgia, a term coined by S. Weir Mitchell, should include only cases of intense pain, burning in character, radiating diffusely up the injured extremity, and often brought on by extremes of temperature and psy- chological stimuli, as well as by the lightest touch or even breath of air over the injured part. Unfortunately, many modern writers have not conformed to this concept. The authors of this chapter have agreed to restrict their use of the term causalgia to the sense originally proposed by Mitchell and not to allow it to become a catch-all for many poorly understood varieties of burning pain. When such a limitation is applied, it is found that nearly all the wounded suffering from this condition were relieved by sympathectomy. The number of patients with true causalgia found in the present study was too few to warrant statistical analysis and will therefore be discussed separately below. B. CAUSALGIA 1. Description of Causalgia Syndrome Clinical Picture. In 1864 Mitchell, Morehouse, and Keen (55) gave their classical description of this syndrome. Its name we apparently owe to Silas Weir Mitchell (54), as he wrote: "Perhaps nothing can better il- lustrate the extent to which these statements may be true than the cases of burning pain, or as I prefer to term it, causalgia, the most terrible of all the tortures which a nerve wound may inflict." These three physicians, while in charge of nerve injuries at a military hospital near Philadelphia during the War Between the States, emphasized that the peculiar burning, 311

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often agonizingly severe and persistent pain is referred in a diffuse fashion throughout the distal portion of the injured extremity, but never to the chest or abdomen. As they observed, it usually results from penetrating wounds with partial injury to a nerve, most often the median or sciatic. Thirty years later J. K. Mitchell, who had reexamined some of his father's veterans of the Civil War, found (53) that the causalgic syndrome was still present in a few, and that severe burning pain, not related to psychologi- cal stimuli but to thermal changes and use of the part, was often a cause of long-continued incapacity and suffering. Weir Mitchell and his colleagues stressed the peculiar emotional factors which characterize the causalgic state, citing the increase in pain which is manifested by the victims with a multitude of psychic stimuli: the stirring music of a military band, jarring noises in the war, even the rattling of a newspaper. In World War II White, Heroy, and Goodman (84) listed an even greater variety of emotional stimuli that aroused intense suffering. These included: Loud or unexpected noises; annoying radio programs. Children crying; arguments with other patients. Jarring of the bed. Exciting movies; stirring music; watching a baseball game; going too fast in an automobile. Prospect of a hypodermic injecrion. Laughing. Other homeostatic factors of interest comprised: Exposure to cold, damp, or very hot weather. Cold air on the painful extremity. Physical exertion. Defecation or urination. Drinking anything cold. Many of the psychological irritants, such as present-day jarring radio programs, exciting movies, and the clatter of aeroplane motors flying too close to the hospital roof, have added to the patient's suffering since Mitchell's day. Boyd (7) has summed up these factors nicely by his state- ment that causalgia is characteristically aggravated by "disturbing the patient's environment." Foerster (23), who coined the term hyperpathia, intended that it should be used to designate the peculiar features of pain seen in the causalgic state. His definition of hyperpathia, given below in full (p. 336), emphasizes the accompanying defects in sensory conduction which make it difficult for the subject to determine the nature or type of painful stimulation, as well as the delayed and explosive nature of the pain, and the prolonged after- discharge. Unhappily recent writers have used the term causalgia to describe any type of persistent hyperalgesia whose cause is difficult to understand, much as dermatologists tend to classify many forms of resistant chronic dermatitis as eczema. As an extreme example, Macfarlane (47) has included such 312

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unrelated conditions as spinal injuries, postamputation neuralgias, painful osteoporosis, and meralgia paraesthetica. No wonder that he has been surprised that so few of these conditions react favorably to sympathectomy! Such use of the term as a catchall for all forms of poorly understood pains in the extremities is to be deplored. We believe that causalgia is a distinct clinical entity, provided the use of the term be restricted to penetrating injuries of nerves which give rise to intense burning pain, influenced by environmental and psychological factors as originally observed by Mitchell. The characteristic pain is likely to start within a few hours after infliction of the wound. Painful states of this sort fall into a consistent group and are strikingly influenced by the sympathetic discharge from the hypothalamus. In addition to the victim's desire to retire into a quiet room where he can escape the emotional stimuli of the open ward, there are other characteristic features. The sufferer from severe causalgia is more comfortable if he pro- tects his hand by wrapping it in a damp cloth or by soaking it in cool water. He is very susceptible to either extreme heat or cold. Trophic changes are frequently seen, and the subject may be unable to wash the painful part or cut his nails. With total disuse because of pain, the trophic changes soon set in. There is a glossy texture to the skin, and the fingers acquire a taper- ing shape. Contractures and osteoporosis then appear, resulting in irrevers- ible changes if pain is not relieved in time. In addition, if unrelieved, the patient rapidly will lose morale and is likely to become a narcotic addict. Because of the striking psychological changes and aggravation of the burning pain by emotional stimuli, physicians who have had no experience with causalgia have often made a diagnosis of psychoneurosis or malingering. Mitchell, et al. (55) pointed this out, stating that "surgeons, who have hap- pened to encounter a single one of the worst of them, have been so surprised at the character of the suffering as to suspect that such an extreme hyper- aesthesia must be due, at least in some measure, to a desire on the part of the patient to magnify his pains." Nerves Involved. Injuries to certain nerves are far more likely to result in causalgia than others. In the 64 cases we have been able to study in detail there has been the following numerical incidence: Median 33 Ulnar 2 Sciatic 17 Posterior tibial 10 Peroneal 2 It is possible that the incidence of causalgia following ulnar and peroneal nerve injuries is somewhat higher than the figures given above, as there are 11 combined median and ulnar lesions in this series which have all been included under the median and 2 combined posterior tibial and peroneal lesions listed under the tibial nerve. No other nerves appear to be responsible for this type of pain, although they often give rise to severe dysaesthesia following injury and partial recovery. 403930—57 22 313

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Diagnosis. To summarize and simplify matters a bit, we agree with Boyd (7), who has written one of the best recent British descriptions of causalgia, that the diagnosis depends on the following factors: a. The appropriate injury of a peripheral nerve. b. Diffuse burning pain (hyperpathia), paroxysmal in character and aggravated "by disturbing the patient's environment." c. Temporary relief by blocking the regional sympathetic ganglia with procaine and permanent benefit following adequate sympathectomy. 2. Historical Considerations At a lecture delivered at the Peter Bent Brigham Hospital, Boston, on October 20, 1953, Sir James Paterson Ross of St. Bartholomew's Hospital, London, pointed out that, although Mitchell deserves the credit for naming causalgia and the general recognition of the syndrome, the condition had been described by a number of others dating back to the 18th century. Alexander Denmark cited one of Wellington's troopers who suffered the characteristic agonizing burning pain following a wound at the Battle of Badajoz in 1812. After the arm was amputed for pain, its dissection showed the median nerve involved in a neuroma. Other examples were recorded by John Abernethy, who described a case following venisection and men- tioned an earlier description of the pain by Percival Pott, which followed partial division of a nerve. Mr. Pott was surgeon to St. Bartholomew's Hospital in the middle of the 18th century. In 1838 John Hamilton of Dublin also reported a peculiar train of symptoms following partial injury of nerves. Other early cases were mentioned by Charles Bell in 1812 and by Antonio Scarpa in 1832. In 1864 James Paget gave an even better descrip- tion in which he said that "glossy fingers appear to be a sign of peculiarly impaired nutrition and circulation due to injury of nerves . . . and are always associated, I think, with distressing and hardly manageable pain and disability." These cases, however, were not the result of pentrating wounds and no mention was made by Paget of aggravation by environmental factors. The references to these early accounts will be found in a previous paper by Ross (64). The epochal papers of Mitchell and his associates (54, 55) have been mentioned above. Most of the graphic descriptions in the first account published with Morehouse and Keen must have been written by Mitchell, as he repeats many of them in his second book. Following the War Be- tween the States little interest was shown in this condition for many years. In and after World War I Leriche (39) began to use periarterial sym- pathectomy and stellectomy in the treatment of severe brachial neuralgias. Spurling (74) and Kwan (37) seem to have been the first to use present forms of upper thoracic sympathetic ganglionectomy. Spurling's boot- legger and Kwan's Chinese soldier had both suffered gunshot wounds with partial injury to the brachial plexus. Spurling's patient was relieved for a number of hours after the chill that followed intravenous injection of typhoid vaccine had subsided* just as was observed in World War II after a malarial 314

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chill. It is of interest to point out that during the stage of defervescence sympathetic activity is decreased, whereas during the actual period of the chill, when vasoconstrictor and pilomotor activity is increased, the pain has been greatly intensified. Neither of these men derived any benefit from extensive neurolysis of the brachial plexus or from resection of a portion of the axillary artery, but both responded in a most gratifying manner to upper thoracic sympathectomy. In World War II Mayfield found an incidence of causalgia of somewhat over 5 percent in nerve injuries treated at the Percy Jones Army Hospital. Others, as shown in table 192, have found it as low as 2 percent. Sym- pathectomy, following favorable results of preliminary diagnostic block with procaine, soon manifested its value in giving immediate relief. Im- pressive evidence in favor of this form of therapy is given in section 4 below and is summarized in Mayfield's monograph on causalgia (49), in Shu- macker's review (72), and in White, Smithwick, and Simeone's volume on the autonomic nervous system (85). Table 192.—Published Cases of Causalgia in World War II and Its Incidence After Wounds of Nerves Authors Cases of causalgia Total number of wounds involv- ing nerves Percent- age of wounds with causalgia Result of sympa- thectomy: Percent- age distribution Total Sym- pathec- tomized Ex- cel- lent Fair Fail- ure Doupe, Cullen, and Chance (22) 7 15 75 9 5 12 100 100 95.7 100 Mayfield and Devine (50) . Ulmer and Mayfield (80) . . Speigel and Milowsky (73) . Rasmussen and Freedman (62) 737 1,477 275 2 5 3.3 70 7 100 40 30 62.5 10 27.5 7 Allbritten and Mai thy (1) . Kirldin, Chenoweth, and Murphey (36) 67 93 52 48 69 29 17.5 2 1.8 Shumacker (72) 57 80.7 White, Heroy, and Good- man (84) . 13 400 3.3 100 3. Treatment The statistics in the following section demonstrate the permanent ef- fectiveness of present therapy. When a patient with a lacerated or penetrat- ing wound develops the classical causalgic syndrome, surgical intervention 315

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on the sympathetic nervous system should be undertaken at a relatively early date, before the reaction to persistent agonizing pain can produce serious contractures from inability to move the painful part, narcotic ad- diction, or psychological deterioration. In the early years of World War II these unfortunate individuals were often branded as malingerers or psychoneurotics, but it was soon found that psychotherapy had little to offer and appropriate interruption of sympathetic pathways gave immediate and dramatic relief. Neurosurgeons working in the Army and Navy hospitals soon came to realize that neurolyses and other local procedures on the injured nerves were useless. According to Mayfield (49), resection of the neuroma with suture of the nerve was often successful. In his paper he cites 28 successful cases and relatively few failures. Our findings have not confirmed this. Furthermore, early resection is rarely an advisable procedure, as the injury to the nerve is usually a minor one, and far better return of sensory-motor function is likely to be obtained if the neuroma is left intact. The conclusion arrived at and thoroughly justified by ex- perience was that demoralizing pain should be relieved by sympathectomy first and injuries to the nerves and adjacent structures dealt with later. The first logical procedure is chemical blocking of the regional sympa- thetic ganglia with a local anesthetic agent. The cervicothoracic or stellate ganglion should be infiltrated when the arm is involved, the lumbar chain for causalgia in the lower extremity. When the pain follows a wound of the sciatic nerve high up in the thigh or buttock care must be taken to carry the infiltration upwards to include the first lumbar and lowest thoracic ganglia. The surgeon who is inexperienced in these techniques may refer to the description in White, Smithwick, and Simeone's textbook (85). If he has any uncertainty concerning the position of his needle he will find it helpful to check its position by a lateral film of the spine, as first suggested by White and Gentry (83). Relief of causalgic pain generally coincides with ganglionic block and may last for many hours. When relief is clear-cut, but brief, it is best to proceed with sym- pathectomy. On the other hand, when relief persists for a longer time, it is advisable to try a series of blocks in the hope that the early mild case of causalgia will improve progressively. This occurred, with the result that no sympathectomy was necessary, in 40 18 of 344 cases reported by Mayfield (49) and in 2 of the 64 cases summarized below. If sympathectomy is necessary it is important that it be carried out at a level sufficiently high to denervate the actual area of injury of the periph- eral mixed nerve. In the arm denervation will be complete after any of the standard upper thoracic operations. This is best accomplished by Smithwick's "preganglionic" sympathectomy because this operation leaves intact the nerve supply to the pupil and upper eyelid, thereby sparing the individual the minor disfigurement of a Homer's sign. While minor u Many of these were early cases with injections carried out in overseas hospitals soon after the onset of pain. 316

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degrees of vasoconstrictor and sudomotor activity can often be demon- strated within a year, there has been no tendency for causalgic pain to recur. To relieve causalgia in the lower extremity it is only necessary to remove the second and third lumbar ganglia if the injury has involved only the ankle or foot but, as Ulmer and Mayfield (80) pointed out, this will not suffice for wounds at higher levels. It is therefore advisable to include the first lumbar ganglion for injuries below the midthigh and to resect the chain up through the diaphragm in case of wounds to the buttock. The technique of these operations also is described in White, Smithwick, and Simeone's monograph (85) and in many other current publications. 4. Statistics From Present Study Difficulties were encountered in attempting to extract cases of true causalgia from the code. Owing to confusion in terminology it has been necessary to review the individual records of patients who were listed under the following headings: Sympathectomy for pain before separation from service. Sympathectomy for pain after separation from service. Good data on course of causalgia. We have also included a number of the senior author's personal cases at the U. S. Naval Hospital at St. Albans, New York, some of which are not included in the population studied in this chapter. Working in this somewhat unsatisfactory fashion, we have been able to obtain records of only 64 veterans for study, 35 who have had some form of Sympathectomy for causalgia in the arm, 29 in the leg.19 We could find no instances in which crippling causalgia can still be said to exist in any veteran examined after an appropriate form of Sympathectomy. Residual complaints have been restricted to sensibility to heat or cold, paraesthesiae, cutaneous hyperalgesia, and moderate discomfort on use or pressure, but never severe enough to prevent use of the extremity. The early intense hyperpathia with accompanying psychological phenomena is no longer present. We have no late observations on individuals with inadequate operations who were still suffering severely when separated from the service. The major statistical reports of early results published during and shortly after World War II are summarized in table 192. It is unfortunate that all of these men have not been followed and doubly so that many of them were not examined by the centers, so that only meager information could be obtained from Veterans Administration examinations. Late results are given in table 193 for the upper and lower extremities of 64 " It is obvious that some of the patients originally diagnosed causalgia and treated by Sympathectomy have not been traced. Mayfield (49) in his questionnaire to neuro- surgeons serving with the Army Medical Corps in the war received reports of 350 cases. This represents the incidence amongst a much larger number of peripheral nerve inju- ries, and it is questionable whether all of these men had typical causalgia. 317

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individuals treated by operations on the paravertebral sympathetic chains and a small group by other procedures. Table 193.—Summary of Results in Causalgia 35 cases upper extrem- ity 29 cases lower extrem- ity Result Satisfactory relief following sympathectomy maintained at 2 to 8 years 20 10 Good early result, but less than 2-year follow-up 1 5 Moderate degree of overresponse or hypersensitivity to heat or cold. . Lasting relief following repeated blocking of sympathetic ganglia 6 1 9 2 Relief following excision of neuroma and suture 2 0 Failures: Inadequate sympathectomy 1 1 No sympathetic block, or test preceding ganglionectomy failed to give significant relief 1 t 2 0 Excision of neuroma and suture ... 0) 3 (>) 3 Cause unknown 0 1 1 One of these patients was later relieved by sympathectomy, another failed to benefit by a subsequent periarterial sympathectomy. These are also listed above under the appropriate headings. 3 Sympathectomies subsequently performed in these 3 cases resulted in 2 successes and 1 failure in an individual who did not respond to preliminary paravertebral test block with procainc. These are also included above under the appropriate headings. These statistics show that, when appropriate sympathectomy has been performed in properly selected cases, there has been only a single failure to obtain immediate and enduring relief. On reexamination after intervals of 10 months to 8 years, 56 of these veterans (including 3 treated by re- peated chemical blocking) have made no mention of serious residual dis- comfort; 15 of these have had some variety of annoying complaint such as mild spontaneous pain, often related to cold or damp weather, or dis- comfort on use of the hand or pressure on the foot, but this has never been incapacitating. In table 193 the latter appear on the third line and their present disabilities are summarized in table 194. The majority of residual complaints may be classified as "overresponse," such as occurs so frequently after any damage to a nerve with incomplete sensory reinnervation. A few have mentioned transitory mild burning discomfort on direct exposure to heat and in case 23 this is still brought on by emotional excitement. (See histories of cases 17, 20, and 23 in table 195.) The intense burning pain with emotional correlation has never returned. 318

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: *j a be . «j .a TJ -d J3 T) .3 Poor selection. Diagnosis of causalgia is questionable and pain not well relieved by procaine block. I 3 -al 1 1 t8§|« 1 1 I ag I £ !!§s& | 1 111 I i -a * ! S 1 1 < si 1§ * IITDal i §e I|L £ i Msl1 i* ! S S. x'5 o-^ ° '° ^ .5 § S d. 1- 115 a 13; , 1 I illl}! 1 ill iliill Si^i tliif! i j 8 j= illl^l l-Sil J i S.S s § 8, >=.*> fSJs?-a 0^-30 ^^5SJi-§ S : csiuie6£>m>< w i S S §. „ £ ON (H P CO t^1 C OO I JO « jjj C§ n n > K ^ u 8e •v >. >* 1 OO 00 Elbow : : : : : : d : : : d : : !5I§ ltl§l§l 11 1 1 S S aSS 13 V C If 2 8 ! S 321

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motor impairment, but a slight although definite hypalgesia over the distribution of the median nerve. Case 18. Maj., USMC, 25 years: This officer was struck by shell frag- ments at Okinawa on May 4, 1945, from which he sustained a compound fracture of the surgical neck of the left humerus and other injuries of lesser importance. The missile, which entered his axilla, partially divided the axillary artery and ulnar nerve, and severed the median. Severe causalgic pain developed immediately throughout his entire hand. On admission to St. Albans he was in poor general condition and still had an unhealed wound in his left axilla. This soon began to extrude clots of blood so that it was necessary to ligate the axillary artery on September 15, 1945 to prevent serious hemorrhage. This operation permitted evaluation of the nerve injury, but median suture in the presence of sepsis was out of the question. It was of interest that this patient, in addition to noticing an increase in his burning pain on cold and psychic stimuli, complained bitterly of the throbbing pain in his hand during any slight argument or whenever his children cried. Following diagnostic block, thoracic sympa- thectomy was performed on September 27,1945, and produced an excellent result. His median nerve was sutured on March 6, 1946, a month after final healing of his wound, together with lysis of the swollen ulnar nerve. In this patient, as in others personally observed in the St. Albans series, there was no vasodilatation in the cutaneous area of the median nerve, where the postganglionic sympathetic fibers had degenerated from the injury to the nerve trunk (skin temperature of first 3 fingers averaging 72° F. but 90° in fifth finger). He remained free of pain at the end of a year. Five years later he had changed his former occupation of civil engineer to salesman. He had had no treatment since discharge. His complaints were limited to poor sensation, some loss of muscle strength, a feeling of coldness at all times, fatiguability on long use, and stiff joints. He con- sidered that he was only slightly handicapped and that the arm had been "repaired as well as possible under the circumstances." He said he was "clumsy with the left hand, tending to drop objects quite frequently." Motor examination showed perceptible movement only in the flexor profundus indicis and opponens with good recovery of flexor carpi ulnaris, but none in distal ulnar muscles. There was also no rcco%-ery of radial and musculocutaneous nerves. Case 20. Pfc., USMC, 19 years: received multiple wounds in the right upper and lower arm at Okinawa on May 10, 1945, with partial median and complete ulnar paralysis. His burning pain began 5 hours after he was wounded. Next to case 17 this patient had the most severe causalgia of the St. Albans series. When he was admitted in July the burning pain involved the entire hand. This became much worse in the cold and quite unbearable on any psychological disturbance, so that he lay in a quiet, darkened room with his arm immobile on the bed and usually protected by moist towels. He complained particularly of the aggravating effect of 330

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loud noises, jarring of the bed, exciting movies, of cold draughts of air over his hand, or cold, rainy weather. The patient stated that each night when he got quieted down and relaxed his pain largely disappeared and he was able to sleep well, but it would appear again soon after he awakened. At the time of his admission he unfortunately had a complicating infectious hepatitis, so that we did not dare submit him to general anesthesia and operation until his jaundice cleared. During this period of waiting his causalgia was relieved three times by paravertebral procaine infiltration of the upper thoracic ganglia, only to recur within a few hours on each occasion. Finally sympathectomy was performed on September 19, 1945, and his causalgia disappeared from this date. A week later the nerves in his arm were widely exposed and a long gap in the ulnar repaired by transplantation and suture. There was a complete injury to the ulnar nerve and surrounding adhesions of the median, which undoubtedly gave rise to the causalgic syndrome. In the forearm there were only fine adhesions to each trunk. It is again of interest to note that, whereas vasodilatation of the median area was complete following pre- ganglionic sympathectomy (skin temperature of the first 3 fingers in a room at 67° F. being between 86.5° and 90°), the temperature in the hemianesthetic ring finger measured 90° on the median and 86° on the ulnar side, and in the completely denervated little finger, where there was complete degeneration of the postganglionic sympathetic fibers, was reduced to 74°. He was under observation on the service for 1 year and remained free of his former burning pain. In August 1953, this veteran gave a carefully considered answer to a routine questionnaire. He states that "the terrible burning pain I had before the sympathectomy was performed has remained dormant with a few exceptions. I have this same severe burning sensation in my first three fingertips while exposed to hot sun such as experienced at the beach. I also experience this sensation on hot, humid days and while exercising excessively. The cold weather has little effect other than the usual stiffness in my fingers." He also experiences a moderately annoying degree of gustatory sweating in the sympathectomized side of his face when he eats spicy foods. There has apparently been little recovery following the extensive neurolysis, transposition, and suture of his ulnar nerve, although there has been useful recovery of the median following the neurolysis and resection of the lateral neuroma. He works as a salesman with some handicap in typing or handling heavy suitcases, and says that he is handi- capped in most sports because he cannot grip well. Case 21. Pfc., USMC, 19 years: On February 28, 1945, at Iwo Jima this Marine was wounded by gunshot. The bullet, which traversed the lower third of his right arm, caused a partial injury of the median and ulnar nerves, nicking the brachial artery as well. Eleven days later the resulting aneurysm was operated upon at Pearl Harbor. Immediately afterward he began to complain of burning pain deep in the hand, made worse by cold, touching the extremity, or by excitement such as that caused by watching 331

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a baseball game or by a close call in an automobile. On arrival at St. Albans he had a cold hand without any radial pulse, the characteristic signs of median and ulnar paralysis, and severe ischemic fibrosis of the flexor muscles in his forearm. On August 21, 1945, a week after his admis- sion, following temporary relief obtained by paravertebral procaine block, his moderately severe causalgia was relieved by preganglionic sympathec- tomy. Although the relief of his burning pain and hyperesthesia was complete and the hand became totally dry, there was little increase in temperature in the anesthetic median and ulnar areas. After this operation he was hospitalized for a period of 6 months for plastic procedures to release scar tissue contractures in his forearm and elbow. No operation on the injured nerves was necessary because of their spontaneous regenera- tion. It was of interest that with early return of sensation he developed the usual mild cutaneous sensitivity that often accompanies nerve regenera- tion, but without any trace of his previous burning pain. This has been a common finding in other cases. This patient, after a letter of inquiry, sums up his status 8 years after sympathectomy as follows: "I am feeling well with hardly any pain at all, except for a burning sensation in my lower arm and hand when I am exerting myself or in rainy weather and cold weather. It is not an extreme pain, but more of a burning and throbbing sensation. In rainy and cold weather my hand turns dark purplish blue, from lack of circulation, I imagine, but in nice weather my hand is its natural color . . . The results of the operation have been very effective . . . Under emotional stress I notice no pain . . . When I am exerting myself, such as working or par- ticipating in sports, I perspire freely on the left side of my body only. When I eat spicy, highly seasoned foods such as pickles, peppers, spaghetti, etc., I perspire freely on my right side from the top of my head down to my waist.20 At the present time, and since my discharge, I am working for the Company in the Shipping Department. I am handi- capped insofar as I am limited in the type of job I can do, as I cannot do any heavy lifting and cannot rotate my arm from the elbow down or cannot clench my fist tightly; also I cannot move my fingers individually." Case 23. Pvt., USMC, 22 years: This man was injured by fragments of mortar shell explosion in the upper arm on 9/16/44 at Pelelieu. There was complete paralysis of the musculocutaneous and partial of the median nerve. Severe causalgic pain appeared in the area of its palmar distri- bution the day after he was wounded. He described this pain as intense burning and throbbing in the thenar side of his hand, which was increased by any light touch or rub, or by any sudden noise, mental upset, fright, anger, or excitement. Instead of being aggravated by cold, this patient complained particularly of hot weather, when he had to protect his hand by ice or cold, wet packs. The most striking feature of all was the relation of his pain to any attempt to swallow cold liquids. This began a month 20 This form of gustatory hyperhidrosis has been described by Haxton (32). 332

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after his wounding and forced him to limit his fluid intake to sips of warm milk. Brachial plexus block with procaine at another hospital had failed to relieve his pain, but the result from paravertebral infiltration of the sympathetic ganglia in our hands was excellent. For the first time he was able to drink a tumbler of cold water. Preganglionic thoracic sympa- thectomy, performed on 11/14/45, gave a most satisfactory result. At the end of 13 months he had no further complaints and was very happy to be able to drink cold liquids, especially beer. In spite of residual biceps paralysis, he had good use of his arm, which had recovered normal sensa- tion in the hand and a fair degree of elbow flexion through compensatory movement by the brachioradialis muscle. In answer to a follow-up letter in July 1953, he reported: "To begin with, I can honestly say that the result of the operation has stood up. The pain I have at present is negligible when compared to the pain I experienced prior to the operation. When the skin is pressed or rubbed, pain is brought about. All the things you mentioned such as emotional excitement, changes of the weather, fatigue bring on pain. Of these, hot humid weather and fatigue cause the more intense burning sensation . . . I have overcome enough of the paralysis to follow a vocation chosen prior to entering service in World War II. I am now employed as a com- mercial artist . . . Today I can hardly believe that I once begged a field doctor at one of the hospitals in the Pacific to amputate my arm because the burning pain was so intense ... I had 15 months of hell's fire in my extremities and you quenched it. The pain I now have on occasion is as nothing to what I had then." Case 24. Pfc., USMC, 23 years: A machine-gun bullet passed through the left forearm on 1/5/45, causing partial paralysis of the median and radial nerves. Five hours later, on recovering from anesthesia for d6bride- ment of the wound, he began to suffer from causalgic pain through his hand. Neurolysis and partial suture of both partially paralyzed nerves had been performed 4 months prior to his admission to St. Albans, and this had resulted in some improvement in his burning pain, but he still complained of severe stabbing, shooting pain in his wrist and hand. This was made definitely worse by cold weather, but was not related to emotion. We regarded this as a somewhat atypical case of causalgia, but we obtained satisfactory relief by diagnostic procaine block. Preganglionic sympathec- tomy, performed on 9/5/45, was followed by a complete remission of all his complaints. There was only partial vasodilatation in the residual hypesthetic territory of the median nerve. In answer to a recent follow-up letter the patient has given us the follow- ing report of his status nearly 8 years after operation: "Relief following sympathectomy has stood up. No pain during hot or cold weather. No pain due to fatigue, emotional excitement, pressure or rubbing of abnormal areas. Skin tone: Excellent. Tautness and blue coloring of fingers com- pletely vanished. Sensitivity: No sensation in areas affected by ulnar and radial nerves; however, if fingers touch an extremely hot object (hot 333

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water, hot dish, door handle exposed to bright sun, etc.) feeling is intensi- fied, almost to the point of touching a hot stove ... In general, the operation you performed has made me much more comfortable, and has enabled me to lead a perfectly normal life within the limitations of my left arm disability . . . Everything that was to have been rectified, has been rectified ... I swim well, and can swing a baseball bat or golf club effectively, making allowance for lack of mobility in left elbow and wrist." Case 27. Pfc., USMC, 22 years: On 3/11/45 a shell fragment caused a partial injury of the right ulnar nerve at the internal condyle. The patient began to notice causalgic pain as soon as he recovered from an accompanying cerebral concussion. When he was admitted to St. Albans he suffered from moderately severe burning pain and hyperalgesia in the ulnar area of his hand, aggravated by cold and the usual psychological factors. The first attempt to block his upper thoracic sympathetic ganglia resulted in vasodilatation, but failed to produce satisfactory drying of the skin or any Horner's sign; furthermore, his pain was not relieved. Follow- ing a second, effective diagnostic block and throughout the 5 months he was under observation following surgical preganglionic denervation he remained free of pain. In July 1953, he wrote a good description of his present state and then came to Boston for examination. These are his statements: "I have come a long way since the operation you performed on me in 1946. Although a major part of the pain is gone, I still get a burning pain in my hand. I usually get this pain when the weather changes, very hot and very cold weather, fatigue and when the little finger is pressed or rubbed. Strenuous exertion may cause the burning to become really disagreeable, otherwise I can forget it. My elbow is still very sensitive and at times I get a very sharp pain in my back where the sympathectomy was performed. Because of this I am unable to lift anything that is too heavy or carry anything for more than a couple of blocks. I am at present a traveling auditor for a freight forwarding company. I do a tremendous amount of writing and typing which causes my arm and hand at times considerable pain. It has not, thank God, prevented me from doing my work. The only time I'm handicapped is traveling. I have three pieces of luggage to worry about." On neurological examination he was found to have good motor recovery. He could feel a fine von Frey hair as well in the autonomous zone of the little finger as in normal areas, but a 2-gram pinprick caused distinct dysesthesia. In summary, this man at 8 years shows the reaction of overresponse with mild burning at times, but this is never related to emotional stimuli nor is it incapacitating. 6. Summary a. In patients who have true causalgia with burning pain which is aggravated by psychological factors and exposure to heat or cold, appro- priate sympathectomy is nearly certain to give relief. Mild degrees of 334

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overresponse may remain, and a few individuals have persistent discom- fort on exposure to heat. b. It is advisable to do a preliminary procaine block, particularly in any atypical case. A few cases of causalgia respond so favorably to the first block that the pain can be relieved without operation by a series of injections with local anesthetic solution. c. In the severe case of causalgia sympathetic surgery should be under- taken at an early date, before immobility of the painful part can lead to irreparable orthopedic deformities, or the individual can develop serious loss of morale or narcotic addiction. d. Periarterial sympathectomy and neurolysis are both useless. e. Excision of neuromata and suture, although occasionally effective, are inadvisable. Delay for a period long enough to make certain that useful regeneration will not take place spontaneously is unjustifiable and a favorable response is at best uncertain. C. OTHER PAINFUL PHENOMENA 1. Definitions Since examiners in the various centers were found to have interpreted rather differently the meanings of terms used to describe painful and other unpleasant phenomena, exact definitions have been formulated. Where substantial semantic conflict has been found, additional exclusions have been made, under statistical control. Complaints of actual pain have been studied in three categories: a. Spontaneous pain is considered to be constant or intermittent pain in the injured limb, present even at rest. It is often particularly annoying in damp or cold weather. It may represent overresponse to unrecognized stimuli, but this is by no means certain. Spontaneous pain may occur when there are fibrotic changes in joints, tendons, or other tissues, in the absence of nerve injury. Old soldiers have for generations complained of their wounds with changes in weather. b. Hyperesthesia (or more strictly, hyperalgesia) should refer only to pain evoked by stimuli which, when applied to the normal subject, would be below threshold. This is a rare phenomenon; in the great majority of nerve injuries elevated thresholds can be demonstrated with graded hairs and pins. Since most examiners took "hyperesthesia" to mean an exaggerated, disagreeable response to tactile or painful stimuli, whatever the threshold, we have ecceptcd this definition although re- luctantly. c. Pain on use or pressure is self-explanatory. This chapter includes also certain sensory complaints other than pain. These phenomena may be a source of considerable annoyance in some "sensitive" individuals. They have been classified as follows: (1) Parcsthesia includes peculiar sensations such as "tingling" or "crawling" which are not painful. (2) Gross sensory loss implies a feeling of numbness or "wooden" sensation. Ordi- narily, this complaint is associated with a demonstrable reduction in sensation to algesiometer, von Frey hairs, or both. (3) Feeling of coldness may in some cases be a major complaint. (4) Bizarre sensory pattern. (5) JUlceration. 335

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In the course of examination for threshold of pain sensation in the autono- mous zone of the injured nerve, examiners were asked to indicate the presence of "hyperpathia" or "dysesthesia." Considerable confusion has arisen in connection with these terms: (1) Dyseslhesia. According to strict etymology* the Greek prefix "dys" has severa meanings: painful, abnormal* or impaired. It is not surprising, therefore, that dysesthesia was construed at some examining centers to be synonymous with pares- thesia and at others as a disagreeable quality to any stimulus. We have grouped individuals who appeared to be in actual pain on local (often minimal) stimulation under this heading and have eliminated those considered to exhibit dysesthesia when other definitions have been used. (2) Hypetpathia. The term hyperpathia was coined by Foerster, who proposed this designation "for certain characteristics of the so-to-speak pure pain sensation which is experienced under pathologic conditions following a nerve suture when pain only is appreciated and all other modalities of sensation are absent. These characteristics are: absence or impairment of correlation between intensity of stimulus and intensity of sensation with a relatively high threshold; a considerable latent period between stimulus and response; or explosive appearance of pain at stimuli above threshold; an abnormal exceedingly unpleasant character to the pain leading to vigorous defensive movements and reactions in the vasomotor and vege- tative spheres; a long persistence of pain after cessation of the stimulus in which pain-free intervals may alternate several times with painful periods; a defective appreciation of the extent and location of the area stimulated; poor discrimination as to the application of two or more stimuli simultaneously, with irradiation of the pain; lack of features which enable the subject to determine the nature or type of painful stimulus." Most of the centers used this term to connote intensely disagree- able sensations and evidence of profound discomfort on stimulation of the injured extremity. The word, as used by Foerster, would include the most severe forms of overresponse as well as cases of causalgia. The categories of "hyperpathia" and "dysesthesia" have been studied together, for statistical reasons. The raw data concerning the unpleasant sensory and related sequelae of nerve injury may be summarized as follows among the 2,962 nerve injuries with some follow-up: Number Nature of complaint of nerves A. No complaints 228 B. Complaint of spontaneous pain 902 C. Painful overresponse: 1. Manifested by complaint of pain on use or pressure 1* 035 2. Manifested by complaint of hyperesthesia 529 3. Manifested by "dysesthesia" or "hyperpathia" on examination for pain threshold 667 D. Causalgia (64)» E. Complaints of unpleasant phenomena other than pain: 1. Paresthesia 1, 080 2. Coldness 1, 000 3. Gross sensory loss 1, 241 4. Bizarre sensory pattern 48 5. Ulceration 150 11 The sixty-four cases of causalgia in World War II, reviewed in section B, were not all drawn from this population. Since causalgia often results from nerve injuries so minor that neither neurolysis nor nerve suture is done, it is impossible from our data to find the true incidence of the condition. 336

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In all there were 5,985 complaints and 667 instances of evoked over- response on examination. In 973 instances there were 3 or more com- plaints. It will be noted that in only 8.3 percent of the cases was there no complaint. An attempt has been made to determine which, if any, of the charac- teristics of the injury or of its treatment may be responsible for the per- sistence of pain or of other unpleasant sensory phenomena. The effects of the agent and site of injury, associated damage to arteries or bones, time of operation, operative findings and technique, and of other obvious fea- tures have been sought by means of statistical analysis, independent of the clinical impressions of the authors. The relation of unpleasant sensory phenomena to motor, sensory, and autonomic recovery has been sought and an attempt has been made to determine whether they have a bearing on occupational disability of the veteran. No significant difference has been found between patients with single and those with multiple nerve lesions in the incidence of painful and related phenomena. These two groups have therefore been combined. However, center variation in the observations on painful phenomena has required that all analyses be made separately for each center. 2. Relation to Characteristics of Injury In a group of median and ulnar nerve injuries, unselected except by center, only 49 of 453 wounds had been caused by cutting instruments. Spontaneous pain occurred in only 18 percent of the cutting injuries as compared with 34 percent of the missile wounds (table 196). This dif- ference, which depends upon only 49 instances of cutting injury, has a probability of about .04 and agrees with clinical experience with civilian injuries. Overresponse, as judged either by complaint or by examination (the latter is not shown in table 196) or paresthesia, appears to be no more common after missile wounds than after wounds produced by cutting instruments. Perhaps because the series is small, especially after sub- division by center, extensive soft tissue damage, requiring plastic repair, is not reliably associated with persistent pain. An associated injury to bones or joints appears to be of little or no importance in determining whether persistent pain will follow a nerve injury, except in the case of the median nerve, where such complaints are somewhat more common in men with complex wounds of this type. Even when infection severe enough to delay nerve repair has been present, no consistent increase is noted in the incidence of painful or other unpleasant phenomena. As might be expected, associated arterial injury is not associated with an increased incidence of spontaneous pain or hyperesthesia. Pain on ordinary use is also found no more frequently in patients with arterial injury, but observations were not made on the possible effect of prolonged exertion in the presence of arterial injury. Painful overresponse to examination for pain threshold after median and ulnar injuries is somewhat more common in patients with associated arterial damage, but the evidence is suggestive 337

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only. Persistent paresthesia seems clearly related, however, to arterial injury complicating wounds of the median nerve (table 197). Some sugges- tion of such an effect is found for the radial nerve, and a rather more questionable effect for the ulnar. It is possible, therefore, that impaired blood supply, in the case of the median nerve at least, may be associated with persistent paresthesia. The possible effect of the level of injury on the incidence of persistent pain has been studied in median, ulnar, and sciatic nerves with negative results. Table 196.—Prevalence of Painful Phenomena and Agent of Injury, Median an I Ulnar Sutures l Complaints Agent Gunshot Cutting in- struments Total Percent 34. 4 Percent 18.4 Per.rnt 32.7 Spontaneous pain Paresthesia 44. 1 51.0 44. 8 Hyperesthesia 19.3 16.3 19.0 Pain on use or pressure 33. 9 36.7 38.6 Number of lesions studied .... 404 49 453 1 Median and ulnar sutures studied in New York center, and ulnar sutures in Philadelphia. Table 197.—Prevalence of Paresthesia and Associated Arterial Injury, by Nerve, All Centers, Complete Sutures Only Arterial injury Nerve Present Absent Total Number of lesions Percent pares- thesia Number of lesions Percent pares- thesia Number of lesions Percent pares- thesia Median 128 30 136 54.7 43.3 47.1 237 250 448 35.4 32.4 38.8 365 280 584 42.2 33.6 40.8 Radial Ulnar 3. Relation to Treatment of Injury Persistence of pain is apparently unrelated to the interval between injury and operation or to the necessity for multiple operations. Only one ques- 338

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tionable exception to this rule was found: over-response to examination for pain threshold occurred in 47 percent of men whose median nerve had been repaired more than once and in only 34 percent of men with a single definitive neurorrhaphy. In most cases, the operator evaluated the amount of scarring of nerve ends and measured the surgical defect just prior to suture. Neither factor exerted a discernible effect upon the persistence of painful phenomena at follow-up examination. Curiously enough, patients who had undergone preliminary bulb suture actually were less likely to have pain or related complaints than were patients who had had only a definitive nerve suture. In attempting to explain this unexpected finding, it was thought that poorer sensory recovery would probably be found in such cases, and might be correlated with a decreased incidence of pain. Actually, as shown in chapter V, sensory recovery was apparently not adversely affected by bulb suture. Extensive mobilization or transposition of the nerve had no sig- nificant association with the prevalence of pain. Tantalum, silk, and plasma glue as suture materials were equally likely to be associated with pain at follow-up examination, and the use or removal of tantalum cuffs similarly provided no useful correlation. The data do not give a clear picture of the effect of sympathectomy in the treatment of pain following nerve injuries. Sympathectomy is known to be of value chiefly in the treatment of causalgia, which, in the coding system used for this study, is not clearly separable from other painful phenomena. Residual spontaneous pain caused by changes in the joints, for example, will still be coded in a patient whose causalgia has been relieved by sympathectomy. Then, too, causalgia is rare in patients whose nerve injury, as in the majority of this group, has been treated by nerve suture. For these reasons causalgia and its treatment are discussed in a separate section of this chapter, on the basis of a review of case records. 4. Relation to Other Follow-up Characteristics A correlation clearly exists between sensory complaints and complaints of impairment of motor function. Only 3 percent of men with sensory complaints were free of motor complaints, while as many as 21 percent of the small group without sensory complaints had no motor complaints (table 198). When a relationship is sought, however, between the various subjective indices of motor impairment (loss of coordination or power, fatigability on long use, easy fatigability, cramps, stiff joints) and spon- taneous pain or overresponse, results are not striking. Only "easy fatiga- bility" shows a reliable correlation. Of patients complaining of spon- taneous pain or overresponse, 51 percent had this motor complaint. In the absence of sensory complaints, only 23 percent complained of easy fatigability. Suprisingly little correlation can be found between motor recovery (as judged by the British Summary) and persistent pain. In the upper limb there is no clear relationship. In the sciatic nerve and its branches, spon- 339

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taneous pain was somewhat less common in men with good motor function than in those whose motor power remained grossly impaired. Neither overresponse nor paresthesia shows any similar relationship to motor recovery. Table 198.—Relationship Between Motor and Sensory Complaints Following Com- plete Suture, All Centers and All Nerves Combined Number of lesions Percentage with sensory complaint Motor complaint None 97 62 9 Anv 1,877 92 7 Total 1,974 91.2 Complaints of pain and related abnormalities bear no discernible rela- tionship to return of sensation as judged by the British Summary. It was necessary to seek further for correlation between overresponse and sensory recovery, since absence of overresponse is a criterion of good sensation in the British Summary. Thresholds of pain and of touch were therefore studied in relation to the history or finding of overresponse. No relation can be found between pain threshold and painful overresponse. Touch threshold, however, seems to be pertinent. After neurorrhaphy, patients whose touch thresholds remain poor or fair are less likely to exhibit over- response on examination than those with good touch recovery. After neurolysis, on the other hand, it is the group with poor return of touch thresholds who are likely to show painful overresponse. Apparently over- response is more closely related to recovery of touch threshold than pain threshold, but the relationship is complex and not explained in terms of these data. Complaints of pain bear no apparent relation to those of "adverse reaction to heat or cold" or to excessive sweating. Curiously, there is no clear evidence that change in occupation or even the presence of an occu- pational handicap is associated with either complaints of pain or evidence of overresponse to examination for pain threshold. 340