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LETTER TO EDITOR
Year : 2010  |  Volume : 58  |  Issue : 4  |  Page : 675-676

Hematomyelia due to anterior spinal artery aneurysm in a patient with coarctation of aorta


Department of Neuroradiology, AIIMS, New Delhi, India

Date of Acceptance31-Jan-2010
Date of Web Publication24-Aug-2010

Correspondence Address:
Sandeep Sharma
Department of Neuroradiology, AIIMS, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.68697

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How to cite this article:
Sharma S, Kumar S. Hematomyelia due to anterior spinal artery aneurysm in a patient with coarctation of aorta. Neurol India 2010;58:675-6

How to cite this URL:
Sharma S, Kumar S. Hematomyelia due to anterior spinal artery aneurysm in a patient with coarctation of aorta. Neurol India [serial online] 2010 [cited 2019 Aug 23];58:675-6. Available from: http://www.neurologyindia.com/text.asp?2010/58/4/675/68697


Sir,

Coarctation of aorta causes multiple enlarged collaterals which shunt blood from the supra to infra coarctation segment of aorta and anterior spinal artery (ASA) is one such route. Such collaterals can result in compressive myelopathy. However, hematomyelia secondary to aneurysmal dilatation of the ASA is distinctly rare.

A 36-year-old male presented to the department of neuroradiology with sudden onset weakness of all four limbs associated with loss of sensation of below the neck and urinary and fecal incontinence of two months duration. On examination he had 0/5 motor power in the lower limbs and 3/5 power in the upper limbs and sensory loss below C6 level Deep tendon reflexes were brisk and plantars were bilaterally up going. Magnetic resonance images (MRI) [Figure 1]a to d showed hematomyelia [Figure 1]a to d extending into the subarachnoid space (not seen in the image). Digital subtraction angiography (DSA) [Figure 2] a to c showed coarctation of aorta and multiple collaterals bypassing the narrowing to supply the descending aorta from branches of both subclavian arteries. There was a large radiculomedullary artery from the right thyrocervical trunk supplying the anterior spinal artery with an aneurysm of ASA at C6 level [Figure 2]b and c. Selective injection of this artery showed contrast flowing retrogradely through a lower dorsal segmental artery into the abdominal aorta [Figure 2]b and c. Embolization was attempted but failed due to technical reasons. Aneurysm was exposed surgically and clipped. Patient made a gradual recovery and his lower limb power improved to 4/5 while his upper limb power and bowel bladder control returned to near normal at four months of follow-up.
Figure 1 :(a) T1spin echo, (b) T2 fast spin echo, (c) T2 STIR and (d) T2*GRE sagittal images of cervical cord, show an aneurysm anterior to the cord at C6 level along with linear anterior cord hematomyelia extending from C2 to C6 which is best evident in the T2* weighted images

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Figure 2 :(a) Arch aortogram showing coarctation of aorta. (b) Angiogram of the branches of the right subclavian artery showing enlarged anterior spinal artery and the aneurysm. (c) Late arterial phase study after injection into the branches of subclavian artery showing reformation of aorta distal to coarctation through the intra and extra spinal collaterals

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The incidence of coarctation of aorta is 0.3 to 0.4/1000 live births. The clinical presenting features depend on the age and severity of narrowing. Most of the patients survive to adulthood and the diagnosis may be incidental. [1] About 25% of patients with untreated aortic coarctation, however, die of cardiac failure. The neurological manifestation include both ischemic and hemorrhagic strokes. Coarctation of aorta is often associated with intracranial aneurysms. However, aneurysms of spinal artery are very rare and occur as a result of chronic hemodynamic stress of shunting of blood flow into the descending aorta. Such hemodynamic forces in the spinal artery generally result in engorgement of the vessels in the tight bony canal and consequent cord compression. [2] The first angiographic demonstration of enlarged ASA in patients with coarctation of aorta was by Doppman and colleagues. [3] These authors reviewed the aortographic features in 40 patients with coarctaion of aorta and found an enlarged trortuous ASA at the cervico-thoracic level in seven patients. Only four case of spinal artery aneurysms in association with coarctation have been reported. [2]

Hematomyelia is more commonly due to trauma. Non-traumatic causes include vascular anomalies, tumors, bleeding disorders, drug abuse, syphilis, hemorrhagic myelitis, and aortic aneurysms. [4] This case illustrates that hematomyelia can be the initial presenting feature of patients with coarctation of aorta and is due to hemodynamic flow-related spinal artery aneurysm. Aortogram and extensive spinal angiogram is advisable to investigate the cause of hematomyelia in patients with coartication of aorta.

 
  References Top

1.Mohamed AH. Coarctation of the Aorta: a comprehensive review. J Arab Neonatal Forum 2006;3:5-13.  Back to cited text no. 1      
2.Jiarakongmun P, Chewitt P, Pongpech S. Ruptured anterior spinal artery aneurysm associated with coarctation of aorta. Intervent Neuroradiol 2002;8:285-92.  Back to cited text no. 2      
3.Doppman JL, Di Chiro G, Glancy DL. Collateral circulation through dilated spinal cord arteries in aortic coarctation and extra spinal arteriovenous shunts. An arteriographic study. Clin Radiol 1969;20:192-7.  Back to cited text no. 3  [PUBMED]    
4.Leep Hunderfund AN, Wijdicks EF. Intramedullary Spinal Cord Hemorrhage (Hematomyelia). Rev Neurol Dis 2009;6:E54-61.  Back to cited text no. 4  [PUBMED]    


    Figures

  [Figure 1], [Figure 2]

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