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J Thorac Cardiovasc Surg 2003;125:409-410
© 2003 The American Association for Thoracic Surgery
Brief Communications |
From the Department of Thoracic and Cardiovascular Surgery, Sendai City Medical Center,a and the Department of Cardiovascular Surgery, Graduate School of Medicine, Tohoku University,b Sendai, Japan.
Received for publication April 25, 2002. Accepted for publication June 26, 2002. Address for reprints: Naotaka Motoyoshi, MD, Department of Thoracic and Cardiovascular Surgery, Sendai City Medical Center, 22-1, Tsurugaya 5-choume, Miyagino-ward, Sendai-City 983-0824 Japan (E-mail: naotaka{at}openhp.or.jp).
Aortic intramural hematoma (IMH) has previously been reported both at necropsy and in vivo and was recently described as a distinct entity. IMH appears to be characterized primarily by aortic wall hematoma without demonstrable intimal flap and occurs as a result of spontaneous rupture of the vasa vasorum
1 or intimal fracture of an atherosclerotic plaque, which allows blood propagation into the aortic media. 2
On the basis of its pathologic feature, mesenteric ischemia by means of branch occlusion is supposed to be complicated with IMH. However, few incidents of perfusion disorder after IMH have been reported, whereas 2.5% to 12.0% of aortic dissections are complicated. 3 We report a rare case of IMH resulting in ischemia of the spinal cord and the right iliopsoas muscle, representing atypical paraplegia.
Clinical summary
At 5 PM on February 1, 2002, a 46-year-old patient had sudden onset of chest pain. On admission, he was hypertensive (182/80 mm Hg), and extremity pulses were palpated normally. Electrocardiography revealed a normal sinus rhythm and no ST-T-segment change. Computed tomography (CT) revealed IMH from the ascending to infrarenal abdominal aorta. Echocardiography showed no aortic regurgitation or pericardial effusion. Type A IMH with a 40-mm maximum diameter was diagnosed, and therefore antihypertensive therapy was preferred.
Weakness of the right thigh and calf was noted at 7 PM. After rapid, severe progress involving the left leg, the symptom was stabilized (modified Tarlov score of 0, Table 1). Sensory loss was observed on the right lower thigh. There was no other neurologic disorder. Cerebrospinal fluid (CSF) drainage at the lumber subarachnoid space was initiated at 10 PM. Primary CSF pressure was greater than 20 cm H2O. Rapid regression of the pressure to 10 cm H2O was marked after drainage. Two hours after drainage, the weakness was resolved, and within the subsequent 6 hours, the neurologic status had improved to 3/5 on the left and 2/5 on the other. However, localized motor dysfunction on the right thigh remained even after drainage. An increase of serum creatine kinase (>20,000 IU) and myoglobinuria was detected. CT showed a lower-density area in the right iliopsoas muscle (Figure 1). After confirmation of ischemic rhabdomyolysis, mild hydration and diuretics were needed for renal protection. The enzyme level normalized within 35 days.
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Discussion
Ischemic spinal cord injury after IMH has been reported previously. 4,5 In our 104 cases of IMH, including types A and B, this was the first patient with spinal cord and muscle ischemia accompanied by IMH. It has become common that an urgent operation should be required for patients with type A IMH because dissection or rupture tends to develop in these patients. 6 Others reported the maximum acute aortic diameter as either a risk factor for survival or a predictor for aortic enlargement in patients with IMH. 7 In the present case the maximum diameter of disease was less than 45 mm. No aortic regurgitation or pericardial effusion was detected. Therefore medical therapy was preferred. 8 Finally, spontaneous regression of ascending aortic IMH was shown at 28 days after admission.
CSF drainage and its protective effect against ischemic spinal cord dysfunction after thoracoabdominal aortic surgery have been reported. 9 Reduction of symptomatic paraparesis with IMH was achieved by means of CSF drainage in sporadic case reports. 5 Moreover, we have performed 42 cases of CSF drainage as an adjunct with thoracoabdominal aortic replacement, and therefore we had no reason to hesitate with this drainage.
As in other reports, CSF pressure was primarily high in this patient. Also, pressure relief could regress symptomatic paraplegia immediately. The pressure might require ambient duration to be stabilized after ischemic insult without the drainage. Safi and colleagues 9 proposed that a 3-day course of the drainage was effective; however, the duration is not based on other reports. In our case, even at 5 days after drainage, the limb function became worse immediately after the pressure increase caused by unexpected kinking. The 3-day course is not absolutely practical. Prolongation of the course might be considered by patient. At 6 days, after limb function became stabilized on the on-off drainage test, the tube was removed.
Right hip joint flexion remained disordered, even after CSF drainage, in this patient. A high serum creatine kinase level and a low density within the right iliopsoas muscle on CT suggested ischemic rhabdomyolysis. This is the first reported case in which aortic IMH was able to occlude lumbar arteries branching out from the iliopsoas muscle. Care should be taken to determine partial involvement of atypical paraparesis in this type of case.
References
This article has been cited by other articles:
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  Y. Moizumi, T. Komatsu, N. Motoyoshi, and K. Tabayashi Clinical features and long-term outcome of type A and type B intramural hematoma of the aorta J. Thorac. Cardiovasc. Surg., February 1, 2004; 127(2): 421 - 427. [Abstract] [Full Text] [PDF]
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