| Article Access Statistics | | | Viewed | 6289 | | | Printed | 195 | | | Emailed | 0 | | | PDF Downloaded | 80 | | | Comments | [Add] | | | Cited by others | 9 | | |
|
Click on image for details.
|
|
| 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
Sandeep Sharma, Subhash Kumar
Department of Neuroradiology, AIIMS, New Delhi, India
| Date of Acceptance | 31-Jan-2010 |
| Date of Web Publication | 24-Aug-2010 |
Correspondence Address:
Sandeep Sharma
Department of Neuroradiology, AIIMS, New Delhi
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0028-3886.68697
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 |
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
Click here to view |
 | 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
Click here to view |
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 | |  |
| 1. | Mohamed AH. Coarctation of the Aorta: a comprehensive review. J Arab Neonatal Forum 2006;3:5-13.  |
| 2. | Jiarakongmun P, Chewitt P, Pongpech S. Ruptured anterior spinal artery aneurysm associated with coarctation of aorta. Intervent Neuroradiol 2002;8:285-92. |
| 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. [PUBMED] |
| 4. | Leep Hunderfund AN, Wijdicks EF. Intramedullary Spinal Cord Hemorrhage (Hematomyelia). Rev Neurol Dis 2009;6:E54-61. [PUBMED] |
[Figure 1], [Figure 2]
| This article has been cited by | | 1 |
Ruptured isolated spinal artery aneurysms: Case series of five patients and a review of the literature on management strategies |
|
| Laura Stone McGuire, Angelica Fuentes, Fady T Charbel, Ali Alaraj, Sepideh Amin-Hanjani | | Interventional Neuroradiology. 2023; : 1591019922 | | [Pubmed] | [DOI] | | | 2 |
Clip Ligation and Disconnection of a Ruptured Ventral Subaxial Cervical Isolated Spinal Aneurysm Using Tailored Access Osteotomies: An Operative Technique |
|
| Tyler Scullen, Christina Ng, Mansour Mathkour, R. Shane Tubbs, Cuong Bui, James Kalyvas | | Operative Neurosurgery. 2022; Publish Ah | | [Pubmed] | [DOI] | | | 3 |
Ruptured Isolated Aneurysm of a Coronal Branch of the Anterior Spinal Artery: Case Report and Discussion of Treatment Strategies |
|
| Alessandra Turrini, Marcello D'Andrea, Vincenzo Antonelli, Luigino Tosatto | | World Neurosurgery. 2021; 145: 119 | | [Pubmed] | [DOI] | | | 4 |
Isolated aneurysms of the spinal circulation: a systematic review of the literature |
|
| Safwan Alomari, Risheng Xu, Judy Huang, Rafael Tamargo, Ali Bydon | | Neurosurgical Review. 2021; | | [Pubmed] | [DOI] | | | 5 |
Endovascular treatment for ruptured cervical anterior spinal artery aneurysm caused by occlusive disease of bilateral vertebral arteries: A case report and literature review |
|
| Shogo Shima, Shinsuke Sato, Tatsuki Motizuki, Yasunari Niimi | | Clinical Neurology and Neurosurgery. 2021; 208: 106862 | | [Pubmed] | [DOI] | | | 6 |
Anterior spinal artery aneurysm in aortic stenosis of different etiology: Report of three cases |
|
| Vivek Singh,Suprava Naik,Sanjeev K Bhoi,RV Phadke | | The Neuroradiology Journal. 2017; 30(2): 180 | | [Pubmed] | [DOI] | | | 7 |
Spinal aneurysms: clinicoradiological features and management paradigms |
|
| Venkatesh S. Madhugiri,Sudheer Ambekar,V. R. Roopesh Kumar,Gopalakrishnan M. Sasidharan,Anil Nanda | | Journal of Neurosurgery: Spine. 2013; 19(1): 34 | | [Pubmed] | [DOI] | | | 8 |
A Ruptured Aneurysm in the Branch of the Anterior Spinal Artery |
|
| Tae Ki Yang | | Journal of Cerebrovascular and Endovascular Neurosurgery. 2013; 15(1): 26 | | [Pubmed] | [DOI] | | | 9 |
Ruptured Spinal Artery Aneurysm Associated with Coarctation of the Aorta: Case Report and Literature Review |
|
| Salah G. Aoun,Maryam Soltanolkotabi,Sameer A. Ansari,Franklin A. Marden,H. Hunt Batjer,Bernard R. Bendok | | World Neurosurgery. 2012; | | [Pubmed] | [DOI] | |
|
|
|
|
|
|
|
|
