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Causalgia a Meta-Analysis of the Literature

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Causalgia a Meta-Analysis of the Literature ORIGINAL ARTICLE Causalgia A Meta-analysis of the Literature S. Ahmad Hassantash, MD; Maryam Afrakhteh, MD; Ronald V. Maier, MD Background: Causalgia is not familiar to most physi- nerve alone or in combination with other nerves (56%) cians whose training and experience are limited to civil- and sciatic trunk injury (60%) were the most common ian practice. nerves involved. In 92%, the nerve injury was incom- plete. The most prominent clinical manifestations in- Hypothesis: Through a thorough review of the litera- cluded burning pain in 86%, increased sweating in 73%, ture, we attempted to determine the boundaries of cau- relief with application of cold in 62%, warmth in 50%, salgia and separate it from other sympathetically related paresthesias in 96%, absence of anesthesia in 81%, and disorders. sensitivity to stimuli in 98%. Response to sympathetic blocks was observed in 88%. Finally, a total of 94% of Data Sources: Database search for English-language ar- the patients undergoing sympathectomy were cured. ticles in MEDLINE and Index Medicus up to the year 2000 as both keyword and subject under causalgia. Conclusions: Cases of causalgia are easy to recognize and treat, with excellent results. Causalgia always fol- Study Selection: References that described any new lows a somatic nerve injury, usually partial, and is asso- cases referred to as “causalgia” by their authors were in- ciated with near-constant, very severe pain distal to the cluded in a meta-analysis. injury in the extremity, varied in nature but character- istically with a predominantly burning quality. An effec- Data Synthesis: One hundred ten articles contained a tive anesthetic block of the appropriate part of the sym- total of 1528 cases of causalgia. High-velocity missiles pathetic chain frequently immediately relieves the pain. caused at least 77% of the injuries. In 72% and 90% of Most cases are cured by surgical sympathectomy. the cases reported, the time from injury to onset of pain was within 1 week and 1 month, respectively. Median Arch Surg. 2003;138:1226-1231 AUSALGIA COMES from the ogy. Many of the articles discuss the cause, Greek words meaning pathophysiology, clinical presentation, and heat (␬␣␷␴␱␵) and pain treatment of these syndromes together as (␣␭␥␱␵), ie, burning pain. a group.3-5 Therefore, we performed a meta- Causalgia, also known as analysis of the literature to evaluate all cur- Ctrue major causalgia, needs to be differen- rently reported cases. Our aim was to bet- tiated from minor causalgia and many ter define the condition of causalgia as a other posttraumatic pain syndromes, such specific syndrome that could be differen- as reflex sympathetic dystrophy, Sudeck tiated from other, less well-defined post- atrophy, sympathalgia, and posttrau- traumatic painful syndromes.6 matic edema. These syndromes are pre- Although patients with different sumed to result from a reflex disturbance manifestations of sympathetically initi- initiated by the injury or inflammation in ated pain have been treated medically which effective therapy necessitates cer- with varying results,3,7-9 the only success- tain measures not ordinarily required to ful treatment for major causalgia is com- From the Department of manage the inciting cause.1 plete interruption of all sympathetic Surgery, Beheshti University of While reviewing the literature in the pathways to the affected limb, usually Medical Sciences, Tehran, Iran process of reporting our cases of causal- achieved by surgical sympathectomy.2,10,11 (Drs Hassantash and 2 Afrakhteh); and Department of gia in an earlier report, we often noted that Patients with other posttraumatic pain Surgery, University of patients with the same clinical manifesta- syndromes usually respond less well to Washington, Seattle tions were labeled with different condi- this invasive mode of therapy. This (Dr Maier). tions according to the above terminol- emphasizes the importance of a more (REPRINTED) ARCH SURG/ VOL 138, NOV 2003 WWW.ARCHSURG.COM 1226 ©2003 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/29/2021 Civil World World Korean Vietnam Lebanon Iran-Iraq War War I War II Conflict Conflict Civil War War 400 367 350 330 300 249 242 250 200 No. of Cases 150 110 100 61 48 42 34 50 33 7 5 00 0 1862-1864 1916-1920 1921-1940 1941-1945 1946-1950 1951-1955 1965-1970 1971-1975 1976-1980 1981-1985 1986-1990 1991-1995 Years Incidence of causalgia in relation to wartime. descriptive definition of the clinical manifestations of Eight groups of patients were described twice.19,26-34 Three causalgia. series of reports, with 4 reports in each group (total of 12 re- Sympathetic-derived pain syndromes present a wide ports), contained more patients in the second of the series (and third in one series) and were then duplicated up to 4 times in spectrum of manifestations and have been defined by vary- 11,15,18,35-44 ing nomenclature, producing significant confusion.1-5 By the literature. In 2 groups of reports, consisting of 2 articles each, there were patients common in each one from the using a meta-analysis approach, we believe causalgia can other group.29,30,45,46 In one series of reports,47,48 the group of be described distinctively enough to be identified as a patients in the first report was included in a larger group of pa- unique entity among this mixture of syndromes. Cau- tients in the second group. If individual duplicated patients in salgia is rarely seen except during wartime (Figure) and consecutive reports from the same institution could be iden- is consequently unfamiliar to most physicians whose train- tified, then these duplicated patients and not the report as a ing and experience are limited to civilian practice. As a whole were excluded. If some data not reported in the se- result, the diagnosis is frequently delayed or missed, caus- lected article were available in the excluded “duplicated” ar- ing protracted suffering and increased disability.2,11 ticle, then these data were included without duplicating pa- Through this review of the literature, we have at- tients. The weighted average of reported means in every article tempted to define the true clinical manifestations and cor- was calculated as the sum (number of patients multiplied by the mean of those data for each individual article) divided by rect treatment of this chronic debilitating disease. the total number of patients in all articles reporting informa- METHODS tion on that specific subject. Thus, we were able to identify 1528 patients labeled as hav- All efforts were made to include all reported cases of causalgia ing causalgia by the authors of the articles. The following in- in this study. A database search was performed for English- formation was extracted and analyzed for all patients de- language articles in MEDLINE (1966-2000, as both keyword scribed: and subject) under causalgia. This was also done with Index General Medicus (up to 1965) under both causalgia and neuralgia, since Mechanism of injury the latter term was sometimes used for this condition during Nerve and limb involved this earlier period. All references presenting new cases were in- Time from injury to onset of pain cluded in the meta-analysis if they represented “causalgia” ac- Clinical manifestations cording to their authors. Additional references to cases re- Type and distribution of pain ported in other articles that were not included in MEDLINE Sweating or Index Medicus were also included in our study. A total of Limb warmness 378 articles were identified and reviewed in relation to the topic. Color changes They were classified as meta-analysis, descriptive, experimen- Sensitivity to cold tal, animal study, or case reports. Articles focusing on animal Treatment studies or review of the literature without presentation of new Response to sympathetic block cases were excluded. A group of 109 articles describing cases Response to sympathectomy of causalgia, as well as our recent report (total, 110 articles), form the basis of this analysis. The articles contained case se- ries of causalgia,2,12-23 patients with causalgia along with other RESULTS sympathetic-based pain syndromes,11 single case reports, and case series of specific complications of treatment modalities, The first description of the condition, while not re- such as causalgia as a complication of first rib resection for tho- ferred to as causalgia, is believed to be by Denmark in racic outlet syndrome24 or thoracoscopic cervical sympathec- 1813.49 Our analysis includes all articles published from tomy,25 or after venipuncture.5 1867, including the first case of causalgia, which was la- (REPRINTED) ARCH SURG/ VOL 138, NOV 2003 WWW.ARCHSURG.COM 1227 ©2003 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/29/2021 Table 1. Mechanism of Injury in All Patients With Causalgia Table 4. Nerves Involved in Total Causalgia Group Cause No. (%) of Patients No. of Patients (% of Total) Nerve Injured [% of Total With Nerve Specified] War related 877 (57) Bullet 13 (1) Brachial plexus 104 (6.8) [12.7] Gunshot wound 109 (7) Median 195 (12.7) [23.8] Shell 37 (2) Ulnar 49 (3.2) [6.0] Nerve trauma 92 (6) Radial 41 (2.6) [5.0] Iatrogenic 61 (4) Median + ulnar 49 (3.2) [6.0] Vascular 25 (2) Median + ulnar + radial 17 (1.1) [2.0] Nonpenetrating 72 (5) Antebrachial 7 (0.5) [0.8] Fracture 13 (1) Lumbosacral 7 (0.5) [0.8] Other 51 (3) Sciatic 144 (9.4) [17.5] Not reported 178 (12) Posterior tibial 40 (2.6) [5.0] Total 1528 (100) Anterior tibial 17 (1.1) [2.0] Anterior + posterior tibial 4 (0.3) [0.5] Peroneal 19 (1.2) [2.3] Saphenous 10 (0.6) [1.2] Intrathecal 6 (0.4) [0.7] Table 2.
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