A forty-three-year-old man presented to the emergency department (ED) with severe bilateral lower-extremity (BLE) pain, approximately twenty-four hours after a two-mile run. Midway through the run, he had experienced “shooting pains” along the lateral calves that radiated up the thighs to both buttocks, with resulting tightness, warmth, swelling, and erythema of both lower extremities. He denied any trauma, twisting, or falling. All of the symptoms were worse on the left side, but both legs were symptomatic.
The treating ED physicians found that the patient had no pain with passive range of motion of either lower extremity at the foot, ankle, knee, and hip; had minimal paresthesias on the dorsum of the left foot; and had a positive bilateral straight leg raise test. The patient was diagnosed with sciatica and was discharged.
The patient returned to the ED twenty-four hours later with worsening pain, increased bilateral foot swelling, and inability to walk. On examination, the blood pressure was measured at 148/78. Both lower legs were swollen, shiny, and erythematous over the lateral surface (Figs. 1-A through 1-D). These areas were moderately tense and tender to palpation. The anterior compartments were not nearly as swollen or painful as the lateral compartments, and the posterior compartments and thighs were completely soft and compressible with palpation. Bilateral posterior tibial and dorsalis pedis pulses were palpable and symmetric. In the right leg, strength was normal, sensation was grossly intact, and there was no pain with passive range of motion, including flexion and extension of the toes and ankle. In the left leg, strength was 5/5 with plantar flexion, 4/5 with dorsiflexion, 3/5 with extensor hallucis longus, 4/5 with eversion, and 4/5 with inversion. Sensation was grossly diminished to diffuse light touch, and there was minimal pain with passive dorsiflexion, eversion, inversion, and plantar flexion of the foot at the ankle. Additionally, the patient had difficulty rising from the bed to standing because of pain.
BLE compartment pressures (Table I) were checked with the handheld Intra-Compartmental Pressure Monitor (Stryker, Mahwah, New Jersey). The patient was taken immediately to the operating room for anterior and lateral compartment fasciotomies and muscle debridement of the left leg. Details of this procedure and all subsequent surgical procedures are noted in Table II. Despite the slightly elevated compartment pressure on the right side, it was felt that the right leg did not meet indications for emergency surgery. BLE radiographs and venous Doppler ultrasound tests were unremarkable. The initial creatine phosphokinase level was elevated to 23,601 IU/L (normal: 40 to 180 IU/L); all other laboratory values were unremarkable.
Following the first procedure (Table II), the patient was given 0.9% normal saline solution at 200 mL/hour to help prevent acute renal failure secondary to rhabdomyolysis9,10. There were no acute events on the first postoperative day, and the creatine phosphokinase level decreased to 12,218 IU/L. Examination of the operative leg showed diminished sensation in the first dorsal web space, intact plantar and dorsiflexion of the ankle, and no firing of the extensor hallucis longus. It was thought that the deep peroneal nerve likely sustained an injury as a result of high compartment pressures in the anterior compartment. On postoperative day three, the patient complained of increasing pressure in the lateral aspect of the right leg. Examination revealed diminished sensation in the first dorsal web space and decreased ankle eversion strength to 3/5, both of which were new findings. The decision was made to perform immediate anterior and lateral compartment fasciotomies of the right leg in addition to the planned reassessment and debridement of the left leg (Figs. 2-A through 2-D). Following the procedure, the patient was continued on intravenous fluids, and the examination remained stable. Two days later, he underwent closure of the left leg wound as well as washout with debridement of the right leg. The final procedure was performed two days later, and the patient was discharged the next day. Details about all of the procedures are provided in Table II.
At approximately ten months postoperatively, the patient was examined by a blinded independent physical therapist who was unaware of the surgical history. The patient demonstrated a normal gait and was able to perform bilateral heel raises and limited single leg hops. He also demonstrated active foot-eversion strength bilaterally (right greater than left) and intact (slightly diminished) sensation bilaterally, as noted in detail in Table III. The treating surgeon then confirmed these physical examination findings.
Compartment syndrome of the lower leg most often affects the anterior compartment, either alone or concomitantly with other compartments. The lateral compartment is rarely affected in isolation5,11,12. Acute compartment syndrome usually occurs immediately following a single traumatic event. Because such traumatic events generally affect only a single limb, an acute bilateral presentation is extremely uncommon. Additionally, while chronic exertional compartment syndrome, which is often exercise-induced, can present bilaterally, the symptoms almost always self-resolve after cessation of activity.
In our patient, the clinical suspicion for acute compartment syndrome was initially low, given the extremely atypical history and presentation. The lack of even minor trauma was not consistent with an acute compartment syndrome. Furthermore, the lack of prior similar symptoms with exercise was not consistent with chronic exertional compartment syndrome. This led to a misdiagnosis on the patient’s first ED visit since he had likely been developing compartment syndrome at that time. On repeat examination, however, the diagnosis became clear.
The progressive development of clinically noteworthy compartment syndrome of the right leg was most unusual. Although both legs were affected on initial presentation, the left was clearly worse; although the right side was concerning, it was elected to monitor this side with serial examinations. The indications for emergency fasciotomy included the clinical presentation and a documented compartment pressure within 30 mm Hg of the diastolic blood pressure (delta P)4,13-15. It is possible that the edema within the right lateral compartment increased during three days of aggressive fluid resuscitation, leading to an increase in pressure beyond the compartment’s intrinsic threshold for reabsorption into the circulation. The swelling was likely exacerbated when the peroneal muscles started to necrose, which may explain the increasing pain in the right leg on postoperative day three. This theory is consistent with the general etiology of all compartment syndromes, in which an increase in a compartment’s volume results in a pressure increase above the closing pressure of the capillaries, which in turn can lead to ischemia. For example, following leg trauma, compartment pressure will likely increase because of hematoma formation, soft-tissue swelling, and increased capillary permeability. Excess fluid then accumulates within a compartment, leading to tissue ischemia, which further damages the capillaries and leads to additional edema7,16,17.
In 2002, Ebenezer and Dust reported on three cases of unilateral isolated lateral compartment syndrome that were missed because of unusual presentations18. In each case, the patient was diagnosed with acute compartment syndrome only after multiple visits to the ED, following progression of symptoms two to three days after initial presentation. A subsequent emergency fasciotomy was performed in each case.
In the report perhaps most similar to our patient, Goldfarb and Kaeding described a case of acute BLE exertional lateral compartment syndrome in a twenty-one-year-old male athlete5. Unlike our patient, this man had actually been previously diagnosed one year prior with BLE chronic exertional compartment syndrome. The symptoms typically had started thirty minutes after beginning exercise and self-resolved twenty minutes after resting. The patient presented nine hours after collegiate football practice with unrelenting pain and numbness of the BLEs, and bilateral lateral compartment syndrome was evident. After emergency anterolateral fasciotomies, during which the lateral musculature was found to be initially dusky and subsequently healthy, the patient eventually returned to full athletic activity.
A review of the English-language literature reveals multiple case reports describing acute isolated lateral compartment syndrome5,8,18-30, almost exclusively secondary to acute trauma. There are also reports of bilateral lateral compartment syndrome5,6,8,31 and acute-on-chronic lateral compartment syndrome5,32. However, to the best of our knowledge, there are currently no reports describing a patient with acute bilateral exertional lateral leg compartment syndrome, especially with delayed presentation. As described, the etiology of isolated lateral compartment syndrome is almost always traumatic, often caused by ankle inversion injuries22,24,26,29,30. While not involving direct trauma to the leg, these types of injuries frequently occur as a result of a twisting mechanism that results in substantial swelling that raises intracompartmental pressures33,34. Acute anterolateral compartment syndromes following minimal trauma and/or exertion have also been reported6,29,34-36.
Despite the irreversible damage that occurred in our patient, he feels no functional compromise. He remains active, has a normal gait, and although he lost the peroneal longus and brevis muscles bilaterally, he is able to evert the ankles. It is not known how he is able to achieve this active motion. This report represents an unusual case of lateral compartment syndrome with an acute, progressive, nontraumatic, exertional presentation. Clinicians should be aware of atypical presentations of a potentially devastating clinical scenario. This case also demonstrates the possibility of a good functional recovery in a patient with subclinical compartment syndrome despite lower-extremity muscle loss.