A Vascular Cause of Painful Lumbar Transverse Syndrome

Abstract

Background:

Despite typical signs of Leriche syndrome, this patient was misdiagnosed with disk prolapse. Vascular insufficiency, such as aortic occlusion, may underlie a motor-sensory deficit in the lower extremities.

Method:

Case report.

Findings:

A 67-year-old woman was admitted for cramping pain in the lumbar spine, hips, and legs; diffuse weakness and sensory disturbances of both legs for weeks; and a cold feeling and livid colorization of the legs. Her complaints were attributed to a disk prolapse L5/S1, for which she underwent immediate laminectomy. Postoperatively, symptoms and signs were aggravated, abdominal pain developed, and C-reactive protein and creatinine were elevated. On the fourth postoperative day, femoral pulses were no longer palpable, anuria developed, and computed tomographry angiography showed occlusion of the abdominal aorta distal to the superior mesenteric artery and absent kidney perfusion. The patient died one day later. Postmortem review showed a visible intra-aortic mass on the previous lumbar magnetic resonance image.

Conclusions:

The differential diagnosis of painful lumbar transverse syndrome should include vascular disease. Misinterpretation of a Leriche syndrome as a disk prolapse has not been reported.

INTRODUCTION

The original 1940 description of Leriche syndrome meant only the complete obliteration of the subrenal aortic bifurcation (1). Since then, the term has been commonly used to describe a variety of clinical manifestations attributable to the obstruction of the infrarenal aorta (2). The typical clinical manifestations of Leriche syndrome include claudication of the hip, buttocks, and thighs and diminished femoral pulses (3). Color Doppler ultrasonography reveals absence of flow in both common iliac arteries. The obliteration is confirmed by computed tomography angiography with contrast material by means of a multislice computed tomographic image (3). When the obliteration has been longstanding, superficial collateral circles and revascularization of the common femoral arteries may occur. Depending on the luminal diameters of the popliteal arteries, subtypes IA, IB, IC, IIA-1, IIA-2, and IIB are differentiated (4). The treatment of choice is aortofemoral reconstruction by means of a bifurcated aortofemoral prosthesis (Y-Roux prosthesis) (5). In single cases, thoracoepigastric anastomosis (6), endovascular treatment (7), or stenting of the renal arteries (8) have been performed.

This patient presented with atypical painful lumbar transverse syndrome that was attributed to vertebral disk prolapse. Although Leriche syndrome may be occasionally confused with other diagnoses, such as primary intraluminal malignoma of the aorta (9–11), misdiagnosis of Leriche syndrome as a disk prolapse has not been reported.

CASE REPORT

A 67-year-old white woman who tested negative for human immunodeficiency virus infection was admitted for pain in the lumbar spine, hips, and legs and diffuse weakness and sensory disturbances of both legs. She had a past history of nicotine abuse (20 cigarettes/d), diabetes mellitus, arterial hypertension, chronic obstructive pulmonary disease, right heart strain, polyglobulia (elevated erythrocyte count), proximal peripheral artery disease, and cholecystolithiasis. Six weeks earlier, she had been evaluated for claudication, and peripheral artery disease was diagnosed. Ten days later, she was admitted for leg edema and exertional dyspnea and orthopnea.

A chest radiograph showed pulmonary congestion; echocardiography revealed right heart strain and moderate tricuspid insufficiency. C-reactive protein was 3.0 mg/dL; polyglobulia was noted, but renal function was normal. After administration of furosemide and discontinuation of diclofenac, the patient lost 10 kg and her leg edema resolved. Despite this improvement, she complained of diffuse abdominal pain. Because of persistent polyglobulia, a single phlebotomy was carried out. On hospital day 10, a sudden increase in renal function parameters and C-reactive protein (without fever) was noted. Because ultrasonography of the abdomen showed only cholecystolithiasis, her complaints were attributed to cholecystitis; amoxicillin and clavulanic acid were administered, and her condition improved. At her request, she was discharged on day 16 with instructions to continue furosemide (40 mg/d), spironolactone (50 mg/d), and 7 more days of amoxicillin and clavulanic acid.

Nineteen days later, she was readmitted for cramping pain in the lumbar spine, hips, and legs; diffuse weakness and sensory disturbances of both legs; and a cold feeling and livid colorization of the legs. Because abdominal computed tomography without contrast medium was uninformative, the surgeon recommended magnetic resonance angiography and referral to a neurologist. Despite this recommendation, magnetic resonance angiography was not carried out. Neurologic examination revealed weakness of both lower legs with right-sided predominance and reduced patellar tendon reflex on the right side, and absent Achilles tendon reflexes bilaterally. There was bilateral hypesthesia of both lower legs for pin and prick sensation. Polyneuropathy, vertebral stenosis, or vascular myelopathy was suspected.

Magnetic resonance imaging of the lumbar spine showed disk prolapse at L5/S1 with bilateral lateral and cranial extension (Figure 1). Contrast medium was not given, because polyglobulia was regarded as a contraindication (12). Neurosurgeons and the treating physicians concluded that the prolapse was the cause of symptoms and signs. Laminectomy and diskectomy at L5/S1 were performed the same day. Postoperatively, however, symptoms and signs were aggravated and abdominal pain developed. Three days after surgery, marked elevations of C-reactive protein, 37.7 mg/dL (n ≤ 0.6 mg/dL), creatinine, 3.2 mg/dL (n ≤ 1.1 mg/dL), and creatine kinase, 3,952 U/L (n ≤ 144 U/L) were observed. On the fourth postoperative day, creatinine increased to 3.9 mg/dL and creatine kinase to 4,050 U/L. Pulses in the inguinal region were no longer palpable. Anuria developed, and computed tomography angiography showed occlusion of the abdominal aorta distal to the superior mesenteric artery and absent kidney perfusion (Figure 2). Shortly after angiography, she experienced an epileptic seizure. The vascular surgeons decided against further surgery; the patient died 1 day later. Retrospectively, an intra-aortic mass was visible on the previous lumbar magnetic resonance image (Figure 1).

Figure 1.

Sagittal T2-weighted magnetic resonance image of the lumbar spine shows a disk prolapse L5/S1 (asterisk) and an extensive hypodensity within the abdominal aorta (arrow) interpreted as a clot.

Figure 2.

Computed tomography angiography image showing occlusion of the abdominal aorta distal to the superior mesenteric artery (arrow) and absent kidney perfusion (Panel B). At the level of the truncus coeliacus, the aorta was perfused (Panel A).

DISCUSSION

This presented case of Leriche syndrome is interesting for several reasons. Initially, the patient was admitted for peripheral artery disease, but during the first hospitalization, treatment concentrated on heart failure. No risk stratification for peripheral artery disease was performed, and proximal extension of vascular pathology was not considered. Also, the abdominal pain was attributed to cholecystitis without addressing the differential diagnoses, despite the presence of lumbar pain and pain in the hips and the legs, that is, the typical clinical features of Leriche syndrome. Although she underwent phlebotomy during the initial hospitalization for polyglobulia, no other preventive measures for arterial thrombosis were instituted, despite other risk factors, including smoking, peripheral artery disease, diabetes mellitus, and arterial hypertension.

This patient was misdiagnosed for several reasons. First, differential diagnoses for lumbar pain, including Leriche syndrome, were not considered. Second, workup for arterial occlusive disease was not done, despite the history of peripheral artery disease and the recommendations of the neurosurgeons. Although surgeons suspected a vascular cause at the second admission and recommended magnetic resonance angiography, it was not carried out for unknown reasons. Vascular myelopathy was considered, but vascular disease of the aorta was overlooked. Third, symptoms and signs were not typical for disk prolapse (normal Lasègue sign, abdominal pain, hip pain, and leg pain together; diffuse pain instead of radicular pain; diffuse weakness instead of monoradicular weakness). Fourth, there was no urinary dysfunction, although prolapse that causes paraparesis should also compromise urination.

Surgery should not have been attempted with this atypical presentation unless all differential diagnoses for this presentation had been considered. Contrast medium should not have been withheld for the abdominal computed tomograph; mild polyglobulia is not a contraindication. That peripheral pulses were actually palpable on the day of the second admission is puzzling, given the lumbar magnetic resonance image with the presumed clot in the aorta the same day. Either the presence of the pulses was erroneously recorded, or, if they were present, there was an incomplete obstruction of the aorta at that time. Renal failure was inadequately addressed. Whether there was a causal relationship between C-reactive protein and renal failure remains speculative.

Aortic thrombosis should be considered in patients who present with pain in the lumbar spine, hips, and lower limbs. This pain may be due to a vascular problem beyond the lumbar plexus. Whenever there is lumbar or leg pain, the femoral and more peripheral pulses should be tested. Questionable pulses should be assessed by Doppler investigations.

CONCLUSIONS

This case shows that occlusion of the aorta presenting as acute back and leg pain and paraparesis may be incorrectly attributed to lumbar disk prolapse and that lumbar magnetic resonance imaging may reveal more than one relevant pathology. Leriche syndrome should be considered as a differential diagnosis of painful, lumbar transverse syndrome.