Bilateral Forearm Compartment Syndromes Resulting From Neuroleptic Malignant Syndrome James M. Schneider, MD, Douglas J. Roger, MD, Richard L. Uhl, MD, Albany, NY
Neuroleptic malignant syndrome (NMS) is an idiosyncratic reaction to the administration of neuroleptic drugs. The primary manifestations of this syndrome are hyperthermia, rhabdomyolysis, muscle rigidity, autonomic dysfunction, and altered level of consciousness. Because rhabdomyolysis and muscle rigidity can lead to swelling within a closed compartment, an unusual complication of NMS is compartment syndrome. 1 We report a case of a patient with neuromuscular malignant syndrome who developed bilateral forearm compartment syndromes.
Case Report A 48-year-old man with known coronary artery disease underwent percutaneous transluminal coronary angioplasty under local anesthesia. Early the next morning the patient was given thorazine for complaints of intractable hiccups. The patient became anxious, and started bleeding from the angioplasty site after climbing out of bed. He was given haloperidol intravenously for further sedation. Despite this, the patient became progressively more agitated. On the following day, he developed an oral temperature of 40.8~ muscle rigidity, further mental status changes, leukocytosis, and oliguria. Serum
From the Section of Hand Surgery, Division of Orthopaedics, Albany Medical College, Albany, NY. Received for publication Sept. 30, 1994; accepted in revised form March 7, 1995. No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Reprint requests: Richard L. Uhl, MD, Division of Orthopaedics, A61OR, Albany Medical College, Albany, NY 12209.
creatinine phosphokinase and creatinine levels became elevated, and the patient developed myoglobinuria. He was intubated and admitted to the medical intensive care unit with a presumptive diagnosis of neuroleptic malignant syndrome. Dantrolene sodium and bromocriptine mesylate were given. The patient remained normotensive throughout this, with blood pressure measurements between 110/70 mmHg to 130/86 mmHg. On the third day, the patient's temperature was near normal but he complained that his forearms were "uncomfortable." These complaints continued over the next several days, and were felt by the supervising medical staff to represent residual muscular strain and stiffness. In the evening of the sixth day after admission, the patient's condition worsened. He developed rigidity of both the anterior and dorsal compartments of both forearms and paralysis of his hands and wrists. He also developed myoglobinuria. An orthopaedic surgery consultation was obtained for the worsening forearm discomfort. He had severe pain with passive flexion and extension of his fingers and wrist, and decreased sensation to light touch in all fingers. The dorsal and anterior compartment pressures of both forearms were measured using a calibrated gauge (Stryker Co., Kalamazoo, MI). Pressures in all compartments were elevated (fight dorsal, 45 mmHg; right palmar, 55 mmHg; left dorsal, 43 mmHg; left palmar, 53 mmHg). The patient was taken to the operating room, where bilateral anterior and dorsal fasciotomies were performed (Fig. 1) with bilateral carpal tunnel releases. Muscle ischemia and necrosis were evident in all compartments. Areas of muscular necrosis were noted on microscopic examination of the The Journal of Hand Surgery 287
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Figure 1. Anterior fasciotomy of the left forearm. Note the pale, relatively avascular areas of the flexor mass secondary to compartment syndrome.
tissue. Following release of the forearms, the patient's laboratory values returned to normal over the next 2 days. Functionally, the patient's initial therapy evaluation demonstrated marked loss of wrist motion, thumb motion, and grasp. The patient continued with therapy daily and used hand and wrist continuous passive motion machines 10 hours daily to maintain the motion gains made with therapy. At 25-month follow-up evaluation, he had returned to his previous work and leisure activities (Table 1).
Table 1. Postoperative Results at 25-month Follow-Up Evaluation
Grip Strength Pinch Strength Sensation--2pt Thumb Little Wrist ROM Extension Flexion Hand ROM
Right
Left
60 lbs 17 lbs
80 lbs 17 lbs
7 mm 10 mm
7 mm 7 mm
50~ 45~ full fist
40~ 70~ full fist
Discussion Neuroleptic drugs, which include the phenothiazines, butyrophenones, and thioxanthenes, are extensively prescribed in the treatment of psychiatric illnesses. They are also frequently used as antiemetics and for the control of agitation in the elderly.2 Neuroleptic malignant syndrome is a severe and potentially lethal complication of neuroleptic drug use, with a reported incidence of 0.05% to 1%. 2 It is a potentially fatal idiosyncratic reaction characterized by "lead pipe" muscular rigidity, rhabdomyolysis, fever, autonomic dysfunction, and altered consciousness. Early studies reported a 20% to 30% mortality rate, usually as a result of respiratory arrest, myoglobinuria with acute renal failure, or cardiovascular collapse. 3-5 In addition to neuroleptic drugs, neuromuscular malignant syndrome is also associated with the use of thymoleptics (antidepressants), metoclopramide (antiemetic), benzodiazepines, phenelzine, and amphetamines. 6 Some authors have noted that most cases of NMS have appeared in patients who have received haloperidol. 7 Furthermore, debilitated patients may be at higher risk for NMS because of dehydration, exhaustion, malnutrition, or the use of restraints. 7 Although the pathophysiology of NMS is not well understood, theories
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include central dopaminergic blockade-induced muscular rigidity and a direct toxic effect on skeletal muscle.8 Once swelling within a compartment exceeds the tissue perfusion pressure, compartment syndrome develops. Conservative measures at this point are rarely successful in decreasing the pressure, and the patient should undergo surgical decompression.9 An increased awareness of an association between neuroleptic malignant syndrome and compartment syndrome might decrease the likelihood of delay and prevent catastrophic complications.
References 1. Hashimoto F, Sherman CB, Jeffrey WH. Neuroleptic malignant syndrome and dopaminergic blockade. Arch Intern Med 1984;144:629-30.
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2. Rosenberg M, Green M. Neuroleptic malignant syndrome: review of response to therapy. Arch Intern Med 1989;1149: 927-1931. 3. Caroff SN. The neuroleptic malignant syndrome. J Clin Psychiatry 1980;41:79-83. 4. Eiser AR, Neff MS, Slifkin RE Acute myoglobinuric renal failure: a consequence of the neuroleptic malignant syndrome. Arch Intern Med 1982;142:601-3. 5. Pope H, Keck P, McElroy S. Frequency and presentation of neuroleptic syndrome in a large psychiatric hospital. Am J Psychiatry 1966;14:1227-33. 6. Ebadi M, Pfeiffer RF, Muffin LC. Pathogenesis and treatment of neuroleptic malignant syndrome. Gen Pharmacol 1990;21:367-86. 7. Levinson JL. Neuroleptic malignant syndrome. Am J Psychiatry 1985;142:1137-45. 8. Kurlan R, Hamill R, Shoulsen I. Neuroleptic malignant syndrome. Clin Neuropharmacol 1984;7:109-20. 9. Blick SS, Brumback RJ, Poka A, Burgess AR, Ebraheim NA. Compartment syndrome in open tibia fractures. J Bone Joint Surg 1986;68A:1348-53.