Fatal Rhabdomyolysis with Bilateral Gluteal, Thigh, and Leg Compartment Syndrome After the Army Physical Fitness Test
Timothy R. Kuklo
A Case Report
Acute exertional rhabdomyolysis is a clinically variable syndrome resulting from the lysis of skeletal muscle cells and the release of myoglobin and other cellular components into the circulation. The classic description by Knochel is that of a patient who has confusion, pallor, and hyperthermia, followed by renal failure, hyperkalemia, and disseminated intravascular coagulation. Relatively recent reports suggest that this entity may be more common than previously appreciated, and that it is not always accompanied by hyperthermia and renal failure. In addition, rhabdomyolysis may be associated with severe metabolic disturbances and compartment syndrome. Multiple causes of rhabdomyolysis have been identified, including crush injury and direct compression.
Compartment syndrome of the thigh after exercise is considered an extremely rare condition. The first case of exercise-induced acute compartment syndrome of the thigh was reported by Kahan et al. in 1994. Gluteal compartment syndrome is even less common, and to our knowledge, exercise-induced gluteal compartment syndrome has not been previously described in the literature. This report illustrates a fatal case of acute exertional rhabdomyolysis complicated by multisystem organ failure, and bilateral leg, thigh, and gluteal compartment syndromes.
A 33-year-old mildly obese black man (weight, 102 kg; height, 173 cm) collapsed near the finish of a timed 2-mile run during a semiannual U.S. Army Physical Fitness Test. He was immediately taken to a local community hospital, where he was noted to be agitated and confused, and was intubated for airway protection. Before intubation, he complained of mild left-calf and thigh pain, and an initial examination revealed mild bilateral thigh swelling but no lower leg swelling or peripheral neurologic deficits. No gluteal compartment examination was documented. The patient’s temperature on admission was 101.1 [degrees] F, he was hypotensive and tachycardic (heart rate, 180 beats per minute) despite aggressive fluid resuscitation, and he had numerous electrolyte abnormalities (Table 1). A computed tomography scan of his head was normal. Because of the elevated creatine kinase, he was transferred to the cardiac care unit at our institution.
TABLE 1 Laboratory Test Results for the Patient in this Study Upon Admission to the Hospital(a)
CPK Hb PT PTT
Date (U/l) (g/dl) (sec) (sec)
4/16 341 16.6 12.3 24.2
4/17 134,240 16 51.1 56.6
Ca K [HCO.sub.3] BUN
Date (mg/dl) (mEq/l) (mEq/1) (mg/dl)
4/16 10.2 4.8 5 9
4/17 5.9 3.2 19 22
Creatinine SGOT SGPT
Date (mg/dl) (U/l) (U/l)
4/16 2.3 293 68
(a) CPK, creatine phosphokinase (creatine kinase); Hb, hemoglobin; PT, prothrombin time; PTT, partial thromboplastin time; Ca, calcium; K, potassium; [HCO.sub.3], bicarbonate; BUN, blood urea nitrogen; SGOT, serum glutamic-oxaloacetic transaminase; SGPT, serum glutamic-pyruvic transaminase.
On admission to the cardiac care unit, the patient was noted to have gross myoglobinuria, metabolic acidosis, acute renal insufficiency, and elevated creatine kinase, all consistent with acute exertional rhabdomyolysis. His spouse confirmed his history of a 17-pound weight loss over the previous 2 weeks, accompanied by crash dieting and the excessive use of “natural diuretics” and other over-the-counter nutritional supplements, including creatine and ephedrine. The patient had no history of exercise or heat intolerance, sickle cell trait, or alcohol or drug abuse. Aggressive volume repletion and urine alkalinization were continued in the critical care unit, and his mental status improved slowly.
The patient was started on hemodialysis the following evening because of persistently decreasing renal function and acidosis and he was successfully extubated, but reintubation was required for acute respiratory failure. His course that evening was complicated by the development of disseminated intravascular coagulation, acute liver failure manifested by hypoalbuminemia, hyperbilirubinemia, and hypoglycemia and a systemic inflammatory response syndrome. In addition, the patient’s thighs and legs now appeared progressively tense and edematous. While his mental status had temporarily improved, he complained of left-calf and thigh pain, and the staff of the intensive care unit noted decreased plantar flexion of his left foot compared with the right. Serum creatine kinase had risen to 615,535 U/I (normal, 50 to 200 U/I) (Fig. 1), and the orthopaedic surgery service was consulted the next morning because the patient was suspected to have compartment syndrome of both legs.
[Figure 1 ILLUSTRATION OMITTED]
On physical examination by the orthopaedic consultant, the patient was intubated and sedated. Both legs and thighs were tense throughout, especially over the anterolateral aspect of his thighs and left calf. Compartment pressures were measured with a handheld intracompartmental pressure monitoring system (Stryker Corp., Kalamazoo, Michigan) and were found to be elevated in the peroneal, anterior, and deep posterior compartments of the left leg and the superficial posterior compartment of the right leg (Table 2). Serum creatine kinase levels were now 765,910 U/I. Despite the patient’s coagulopathy, he was immediately taken to the operating room for bilateral lower extremity fasciotomies.
TABLE 2 Compartment Pressures (in millimeters of mercury) in the Patient in this Study.
Date Side Peroneal Anterior Deep Superficial
4/19 Right 35 36 46 57
Left 60 55 70 39
Date Side Anterior Posterior
(thigh) (thigh) Gluteal
4/19 Right 65
4/19(a) Right 17 14
Left 54 25
4/20 Right 110
(a) After fasciotomy.
Thigh pressures were monitored intraoperatively and were found to be elevated in both the anterior and posterior compartments of both legs. Fasciotomies were performed bilaterally in all four leg compartments and all three thigh compartments; each thigh was accessed through a two-incision technique, as described by Tarlow et al. In the left leg, the superficial layers of the anterior and peroneal compartments and the deep posterior compartment appeared to be necrotic. The right anterior compartment also had areas of dusky-colored muscle, but no necrosis. When the thighs were incised laterally, the vastus lateralis muscles immediately bulged through the fascial incisions and appeared quite edematous. The vastus intermedius and rectus femoris muscles appeared dusky throughout. The medial l;high compartments were also released because of the appearance of the patient’s other lower extremity compartments, but no edema or necrosis was discovered. All compartments were packed with sterile saline-soaked gauze, and the patient was urgently transferred back to the surgical intensive care unit for hemodialysis to control his rising and uncontrollable serum potassium and cardiac arrhythmia.
Later that evening, the patient’s serum creatine kinase decreased to 352,400 U/I; however, his coagulopathy was difficult to control and h.e required multiple blood and plasma transfusions. He was also resuscitated three times for cardiac arrest thought to be secondary to his decreased core body temperature resulting from multiple transfusions. An unexplained increase in his serum creatine kinase the following morning accompanied by continued hyperkalemia prompted the taking of gluteal compartment measurements, noted at 110 mm Hg on the right side and 60 mm Hg on the left side. He was returned to the operating room shortly thereafter, and both gluteal compartments were released through extension of the lateral compartment fasciotomy incisions. The gluteus maximus muscle was edematous and several areas of superficial necrosis were noted. The thigh and leg compartments were reexamined, and the left rectus femoris muscle was acontractile and necrotic throughout its extent and was excised. A significant amount of the left vastus lateralis muscle was also necrotic and was debrided. The remainder of the wounds were debrided, irrigated, and repacked with saline-soaked gauze.
The patient required multiple blood, plasma, and platelet transfusions over the next 24 hours. He also required almost continuous hemodialysis for fluid and electrolyte management. Despite aggressive medical management, he developed multisystem failure and expired the following afternoon. The family declined an autopsy.
This case represents an extremely unusual instance of fatal rhabdomyolysis complicated by bilateral lower extremity compartment syndromes involving the gluteal, thigh, and leg compartments. Further, the presentation alerts us to the potential deleterious effects of nutritional sports supplements and binge dieting.
Exertional rhabdomyolysis is a well-recognized disorder that follows symptoms of acute overexertion. The patient typically has pain and swelling over the affected muscles, along with the laboratory findings discussed later. Line and Rust have emphasized the need to screen for occupational, recreational, environmental, and medical risk factors in patients who have signs and symptoms similar to those in our patient. In this case, there were no known medical risk factors, and the fitness test was conducted on a spring morning with a mild breeze and an average temperature of 68 [degrees] F.
Screening for acute rhabdomyolysis includes a urine dipstick positive for blood (orthotoluidine positive) without microscopic hematuria (red blood cell count less than five per high-power field). In suspected cases, an elevated serum creatine kinase level should confirm the diagnosis (normal, 50 to 200 U/I). Treatment requires prompt recognition and includes aggressive intravenous fluid replacement to promote a brisk diuresis and urinary dilution and correction of electrolyte abnormalities. Some authors have emphasized hypovolemia as an added nephrotoxic factor and have reported fluid requirements of up to 10 liters within 12 to 24 hours. This patient exhibited much the same, except that fluid depletion was estimated at 14 liters. Acidosis, an additional nephrotoxic factor, was also present. The serum creatine kinase initially decreased after the compartment releases, but then increased a second time. This second rise was most likely secondary to the gluteal compartment syndromes, which were not recognized at the time of the initial fasciotomies. This case is also remarkable for the level of serum creatine kinase, 765,910 U/I. This was the highest reported level we had seen and was most likely due to the massive muscle involvement in this case.
Another unusual aspect of this case was the persistence of hypokalemia and hyperphosphatemia for the first 2 days after admission to our hospital. He initially had had normal serum potassium (3.9 mEq/l; normal, 3.5 to 5.0 mEq/l), normal serum calcium (9.0 mg/dl; normal 9 to 10.5 mg/dl), and hypophosphatemia (1.7 mg/dl; normal, 2.3 to 4.7 mg/dl) but later developed hypokalemia (3.2 mEq/l), hypocalcemia (5.1 mg/dl), and hyperphosphatemia (9 rog/ dl). Hyperkalemia, hyperphosphatemia, and hypercalcemia are usually found in patients with rhabdomyolysis. We speculate that our findings were secondary to the localization of calcium and phosphorous in the lower extremities until cell death, when large quantities of the electrolytes were released.
Several nephrotoxic cofactors were present in this case, including diuretics and hypovolemia, that undoubtedly led to the onset of acute renal failure. Multisystem organ failure and disseminated intravascular coagulation rapidly followed and ultimately led to death.
Acute, exercise-induced compartment syndrome is rare, and there have been only two cases of exercise-induced thigh compartment syndrome associated with rhabdomyolysis reported in the literature.[13,38] [Editor’s Note: We have published two papers on acute compartment syndrome of the thigh associated with exercise in this issue because we believed they both deserve notice and the fatal outcome in one patient took this case report beyond the usual. REL] Exercise-induced compartment syndrome may not develop until 24 to 48 hours after the precipitating event, and may not always be recognized immediately. In the case reported here, the patient was first treated for hypovolemia and possible myocardial infarction and did not develop notable extremity swelling, pain, or weakness until 48 hours after admission to the hospital. Thus, it appears that acute rhabdomyolysis preceded the onset of multiple compartment syndromes in the exercised extremities. It is possible that this patient developed an acute gluteal or thigh compartment syndrome that was initially unrecognized, and that this caused the rhabdomyolysis; however, this would not explain the delayed onset of bilateral leg and thigh symptoms. It is more likely that strenuous exercise in this dehydrated, undernourished patient caused an acute rhabdomyolysis and excessive swelling in the exercised extremities. The swelling was aggravated by aggressive fluid resuscitation and, combined with the hypotension, caused multiple, acute compartment syndromes. The multiple compartment syndromes caused massive muscle necrosis, which worsened the rhabdomyolysis and perpetuated a vicious circle.
Many patients who are at risk for developing exercise-induced compartment syndrome may seek medical care in a delayed fashion. Therefore, immediate consultation of the orthopaedic surgery service, close follow-up, and repeated examinations are critical for prompt recognition and treatment. This case report also illustrates the importance of closely monitoring the patient after compartment release for resolution of symptoms and involvement of other compartments.
To our knowledge, exercise-induced gluteal compartment syndrome has not been documented. Gluteal compartment syndrome, however, has been observed after drug abuse accompanied by altered mental status and prolonged stasis, alcohol abuse, intraoperative positioning complicated by prolonged compression, superior gluteal or medial femoral circumflex artery rupture, epidural analgesic infusion, and trauma[2,3,9,12,18,24,25,31,32]
Bleicher et al. found only 27 previous cases of gluteal compartment syndrome, and only 3 of these cases were bilateral. Since these authors’ report on bilateral gluteal compartment syndrome, Klockgether et al. reported a case of gluteal compartment syndrome with sciatic nerve palsy after intravenous injection of heroin. The case described here is the 38th reported case of gluteal compartment syndrome and the 5th bilateral case (Table 3).
TABLE 3 Reports of Gluteal Compartment Syndrome in the Literature
Study Year Cause of syndrome
Kaufman and Choi 1972 Unknown, no trauma
Klock et al. 1973 Substance abuse, unconsciousness
Evanski and Waugh 1977 Substance abuse, unconsciousness
Owen et al. 1978 Fall
Substance abuse, unconsciousness
Vukanovic et al. 1980 Substance abuse, unconsciousness
Goldberg et al. 1980 Surgical positioning
Dahlberg and Howard 1982 Surgical positioning
Anonymous 1985 Intramuscular drug injection
Leventhal et al. 1985 Surgical positioning
Rommel et al. 1986 Surgical positioning
Petrik et al. 1988 Struck by automobile
Smith et al. 1989 Surgical positioning
Brumback 1990 Traumatic superior gluteal
Lachiewicz and Latimer 1991 Surgical positioning
Scmalzried and Eckardt 1992 Substance abuse/fall,
Scmalzried et al. 1992 Ehlers-Danlos, bleeding from
inferior gluteal artery
Barnes et al. 1992 Substance abuse, local pressure
Yoshioka 1992 Trauma
Prynn et al. 1994 Substance abuse, unconsciousness
Hynes and Jackson 1994 Substance abuse, unconsciousness
Jupiter et al. 1995 Surgical positioning
Pai 1996 Iatrogenic medial circumflex
Bleicher et al. 1997 Substance abuse, unconsciousness
Hill and Bianchi 1997 Substance abuse
Kontrobarsky and Love 1997 Epidural infusion
Klockgether et al. 1997 Intramuscular injection
The gluteal compartment has been described as three separate compartments encompassing the gluteus maximus muscle, the gluteus medius-minimus muscle, and the tensor fascia lata. Schmalzried and Eckardt further emphasized the importance of recognizing the gluteal compartment syndrome, the three-compartment nature of this compartment, and the location of the neurovascular structures. Henrys described a “question-mark shaped” extensile incision for gluteal compartment exposure. In our case, this approach was not used because the patient had previously undergone bilateral thigh compartment releases through an extensile lateral approach, and these incisions were extended proximally in a curvilinear posterior direction.
In all compartment syndromes, successful outcome is centered around rapid diagnosis and treatment. In this example, intensive, multidisciplinary support was readily available, and compartment releases could be performed in the ideal environment of the operating room. If this support is not available, fasciotomies may be performed in the intensive care unit at the bedside. Treatment should not be delayed while more ideal conditions are arranged.
Sports Nutrition and Dieting
Numerous factors may cause or aggravate rhabdomyolysis, such as high ambient temperature and humidity, dehydration, fatigue, recent viral illness, use of medications, use of cocaine or alcohol, sickle cell trait, and renal insufficiency. Rhabdomyolysis has been reported following the Army Physical Fitness Test, in military recruits, in well-trained high-performance athletes, and in police- and fire-academy candidates.[6,10,21,22,30,36] The patient in this report was not a recruit. He had served in the United States Army for more than 11 years and had previously completed numerous physical fitness tests without difficulty. Therefore, he should have been physically conditioned and not at high risk for the development of rhabdomyolysis. Only conjecture is possible in determining the potential role played by dieting, rapid weight loss, and dehydration secondary to the excessive dieting. This patient started the Army Physical Fitness Test approximately 17 pounds below his usual weight. This weight loss occurred in the 2 weeks preceding the test. Without screening laboratory evaluation, it is difficult to determine his preexercise kidney function or electrolyte levels; however, he had no medical history of renal insufficiency. Ephedrine may have contributed by causing increased body temperature and decreased peripheral perfusion. Creatine has been reported to cause renal dysfunction. It is likely that these factors, combined with diuretic use, possible creatine use, and overexertion, led to the development of rhabdomyolysis followed by compartment syndrome, renal failure, refractory hyperkalemia, acidosis, disseminated intravascular coagulation, and multiorgan failure.
Funding for this study was supported by the Department of Clinical Investigation, Walter Reed Army Medical Center, Washington, DC.
[1.] Barnes MR, Harper WM, Tomson CR, et al: Gluteal compartment syndrome following drug overdose. Injury 23: 274-275, 1992
[2.] Bleicher R J, Sherman HF, Latenser BA: Bilateral gluteal compartment syndrome. J Trauma 42:118-122, 1997
[3.] Brumback RJ: Traumatic rupture of the superior gluteal artery, without fracture of the pelvis, causing compartment syndrome of the buttock. A case report. J Bone Joint Surg 72A: 134-137, 1990
[4.] Dahlberg P J, Howard RS: Rhabdomyolysis: An unusual postoperative complication. Urology 127: 520-521, 1982
[5.] Evanski PM, Waugh TR: Gluteal compartment syndrome: Case report. J Trauma 17: 323-324, 1977
[6.] Exertional rhabdomyolysis, acute renal impairment: New York City and Massachusetts, 1988. MMWR Morb Mortal Wkly Rep 39: 751-756, 1990
[7.] Goldberg M, Stecker JF Jr, Scarff JE Jr, et al: Rhabdomyolysis associated with urethral stricture repair: Report of a case. J Urol 124: 730-731, 1980
[8.] Henry AK: Extensile Exposures. New York, Churchill Livingstone, 1957, pp 183-189
[9.] Hill SL, Bianchi J: The gluteal compartment syndrome. Am Surg 63: 823-826, 1997
[10.] Hurley JK: Severe rhabdomyolysis in well conditioned athletes. Mil Med 154: 23-25, 1989
[11.] Hynes JE, Jackson A: Atraumatic gluteal compartment syndrome. Postgrad Med J 70: 210-212, 1994
[12.] Jupiter JB, Ring D, Rosen H: The complications and difficulties of management of nonunion in the severely obese. J Orthop Trauma 9: 363-370, 1995
[13.] Kahan JSG, McClellan RT, Burton DS: Acute bilateral compartment syndrome of the thigh induced by exercise. A case report. J Bone Joint Surg 76A: 1068-1071, 1994
[14.] Kaufman G, Choi B: Ischemic necrosis of muscles of the buttock [case report]. J Bone Joint Surg 54A: 1079-1082, 1972
[15.] Klock JC, Sexton MJ: Rhabdomyolysis and acute myoglobinuric renal failure following heroine use. Calif Med 119: 5-8, 1973
[16.] Klockgether T, Weller M, Haarmeier T, et al: Gluteal compartment syndrome due to rhabdomyolysis after heroin abuse. Neurology 48: 275-276, 1997
[17.] Knochel JP: Catastrophic medical events with exhaustive exercise: “White collar rhabdomyolysis.” Kidney Int 38:709-719, 1990
[18.] Kontrobarsky Y, Love J: Gluteal compartment syndrome following epidural analgesic infusion with motor blockage. Anaesth Intensive Care 25: 696-698, 1997
[19.] Lachiewicz PF, Latimer HA: Rhabdomyolysis following total hip arthroplasty. J Bone Joint Surg 73B: 576-579, 1991
[20.] Leventhal I, Schiff H, Wulfsohn M: Rhabdomyolysis and acute renal failure as a complication of urethral surgery. Urology 26: 59-61, 1985
[21.] Line RL, Rust GS: Acute exertional rhabdomyolysis. Am Faro Physician 52: 502-506, 1995
[22.] Olerud JE, Homer LD, Carroll HW: Incidence of acute exertional rhabdomyolysis. Serum myoglobin and enzyme levels as indicators of muscle injury. Arch Intern Med 136: 692-697, 1976
[23.] Owen CA, Woody PR, Mubarak SJ, et al: Gluteal compartment syndromes. A report of three cases and management utilizing the wick catheter. Clin Orthop 132: 57-60, 1978
[24.] Pal VS: Compartment syndrome of the buttock following a total hip arthroplasty [case report]. J Arthroplasty 11:609-610, 1996
[25.] Petrik ME, Stambough JL, Rothman RH: Posttraumatic gluteal compartment syndrome. A case report. Clin Orthop 231: 127-129, 1988
[26.] Poels PJE, Gabreels FJM: Rhabdomyolysis: A review of the literature. Clin Neurol Neurosurg 95:175-192, 1993
[27.] Pritchard NR, Kaita PA: Renal dysfunction accompanying oral creatine supplements. Lancet 351: 1252-1253, 1998
[28.] Prynn WL, Kates DE, Pollack CV Jr: Gluteal compartment syndrome. Ann Emerg Med 24:1180-1183, 1994
[29.] Rommel FM, Kabler RL, Mowad JJ: The crush syndrome, a complication of urological surgery. J Urol 135:809-811, 1986
[30.] Schiff HB, MacSearraigh ETM, Kallmeyer JC: Myoglobinuria, rhabdomyolysis and marathon running. Q J Med 47: 463-472, 1978
[31.] Schmalzried TP, Eckardt JJ: Spontaneous gluteal artery rupture resulting in compartment syndrome and sciatic neuropathy. Report of a case in Ehlers-Danlos syndrome. Clin Orthop 275: 253-257, 1992
[32.] Schmalzried TP, Neal WC, Eckardt JJ: Gluteal compartment and crush syndromes: Report of three cases and review of the literature. C/in Orthop 277: 161-165, 1992
[33.] Sinert R, Kohl L, Rainone T, et al: Exercise-induced rhabdomyolysis. Ann Emerg Med 23: 1301-1306, 1994
[34.] Smith JW, Pellicci PM, Sharrock N, et al: Complications after total hip replacement. The contralateral limb. J Bone Joint Surg 71A: 528-535, 1989
[35.] Tarlow SD, Achterman CA, Hayhurst J, et al: Acute compartment syndrome in the thigh complicating fracture of the femur. A report of three cases. J Bone Joint Surg 68A: 1439-1443, 1986
[36.] Teitjen DP, Guzzi LM: Exertional rhabdomyolysis and acute renal failure following the Army Physical Fitness Test. Mil Med 154: 244-245, 1989
[37.] Vukanovic S, Hauser H, Wettstein P: CT localization of myonecrosis for surgical decompression. A JR Am J Roentgenol 135: 1298-1299, 1980
[38.] Wise JJ, Fortin PT: Bilateral, exercise-induced thigh compartment syndrome diagnosed as exertional rhabdomyolysis. A case report and review of the literature. Am J Sports Med 25: 126-129, 1997
[39.] Yoshioka H: Gluteal compartment syndrome: A report of 4 cases. Acta Orthop Scand 63: 347-349, 1992
Timothy R. Kuklo,(*) LTC, MC, USA, John E. Tis,(*) ([dagger]) CPT(P), MC, USA, Lisa K. Moores,([double dagger]) MA J, MC, USA, and Richard A. Schaefer,* LTC, MC, USA
From the (*) Orthopaedic Surgery Service and the ([double dagger])Critical Care Medicine Service, Walter Reed Army Medical Center, Washington, DC
([dagger]) Address correspondence and reprint requests to John E. Tis, MD, 4216 Gelding Lane, Olney, MD 20832.
The opinions or assertions contained herein are the private views of the authors and are not to be construed as official or as reflecting the views of the Department of the Army or the Department of Defense. The research was approved by the Walter Reed Army Medical Center Clinical Investigation Committee.
No author or related institution has received any financial benefit from research in this study. See “Acknowledgments” for funding information.
COPYRIGHT 2000 American Orthopaedic Society for Sports Medicine
COPYRIGHT 2001 Gale Group