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LETTER TO EDITOR
Year : 2014  |  Volume : 62  |  Issue : 3  |  Page : 318-320

Hematomyelia due to ruptured intramedullary aneurysm associated with arteriovenous malformation masquerading as demyelination


Department of Neuroaurgery, Jawaharlal Institute of Postgraduate Medical Education and Research, Pondicherry, India

Date of Submission23-Jul-2014
Date of Decision01-Jun-2014
Date of Acceptance01-Jun-2014
Date of Web Publication18-Jul-2014

Correspondence Address:
V R Roopesh Kumar
Department of Neuroaurgery, Jawaharlal Institute of Postgraduate Medical Education and Research, Pondicherry
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.137003

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How to cite this article:
Roopesh Kumar V R, Madhugiri VS, Ramesh A S, Prabhu A S. Hematomyelia due to ruptured intramedullary aneurysm associated with arteriovenous malformation masquerading as demyelination. Neurol India 2014;62:318-20

How to cite this URL:
Roopesh Kumar V R, Madhugiri VS, Ramesh A S, Prabhu A S. Hematomyelia due to ruptured intramedullary aneurysm associated with arteriovenous malformation masquerading as demyelination. Neurol India [serial online] 2014 [cited 2019 Dec 10];62:318-20. Available from: http://www.neurologyindia.com/text.asp?2014/62/3/318/137003


Sir,

Spinal aneurysms (SA) are very rare lesions. [1],[2] They can be isolated (Type 1) or associated with arteriovenous malformations (AVM) (Type 2). [3],[4],[5] We report a spinal aneurysm that bled, causing hematomyelia. This was mistaken for a demyelinating lesion and treated as such.

An 18-year-old fsmale presented with sudden onset of weakness and parasthesiae of all 4 limbs. She also complained of non-radiating neck pain. On evaluation (at another hospital), she was found to have flaccid paralysis of all 4 limbs with complete loss of sensation below C4. She had shallow respiration and was intubated and electively ventilated. Magnetic resonance imaging (MRI) cervical spine T 2 W images revealed diffuse hyperintensity of the cord extending from the pontomedullary junction till the lower dorsal spine [Figure 1]a. T 1 W images revealed an ill-defined focal hyperintensity at C4 with cord expansion [Figure 1]b. A diagnosis of acute demyelination was made and she was administered corticosteroids and subsequently IV immunoglobulins. She recovered gradually and power improved to 3/5 in the upper limbs and 2/5 in the lower limbs. Repeat MRI one month later revealed partial resolution of the T 2 signal changes and multiple flow voids at C4-5 [Figure 2]. She was then referred to our institute. A spinal angiogram with cannulation of the left thyrocervical trunk revealed aperimedullary AVM at C4-5 level with a large intranidal aneurysm [Figure 3]a and b. MRI confirmed the position of aneurysm to be intramedullary with surrounding hemosiderin deposition suggestive of old rupture [Figure 4]a and b.

Since this was a Type 1A in SA, she underwent surgery. Via C4-6 laminectomy and medial 1/3 C4-5 left facetectomy, the lesion was accessed. A leash of abnormal vessels was found on the pial surface [Figure 5]a. The arterial feeder was identified in the C4-5 foramen, coagulated and divided [Figure 5]b . Subsequently, the nidus on the perimedullary region was circumferentially dissected and excised. The aneurysm was buried within the cord substance and the radiculo-pial arterial feeder [Figure 5]c was coagulated following which the aneurysm become lax. Fine needle aspiration of fundus was done and aneurysm sac was emptied [Figure 5]d. Further dissection of aneurysm was not attempted fearing damage to normal surrounding cord substance.
Figure 1: (a) MRI T2W image showing holocord edema extending till pontomedullary junction (b) MRI T1W image showing

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Figure 2: MRI T2W image after 6 weeks showing flow voids at C4-5 level

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Figure 3: (a) Spinal DSA revealing intranidal aneurysm with Arteriovenous malformation (b) Spinal DSA showing venous drainage of the AVM

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Figure 4: (a) MRI T2W axial image showing the intramedullaryy location of the aneurysm (b) MRI T2W axial image showing multiple flow voids in the left foraminal region

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Figure 5: (a) Intraop picture showing the nidus (b) Main arterial feeder from the radicular artery (c) Showing the lateral surface of the aneurysm wall (main fundus buried within the cord substance) (d) Aneurysm fundus being punctured to empty the sac

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Postoperative MRI spine revealed no residual nidus [Figure 6]a and b and spinal angiography confirmed total excision of the AVM and complete obliteration of the aneurysm [Figure 7]a and b. At 6 weeks follow-up, she was ambulant with support and her sphincter functions were normal.
Figure 6: (a) - A Postoperative T2W Sagittal MRI showing complete excision of nidus (b) Postoperative T2W Axial

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In an extensive review of literature by Madhugir et al., most SA occurred in the cervical spine and were intraduralextra-medullaryin location. [5] Most of these patients presented with spinal (and/or cranial) subarachnoid hemorrhage. [2],[3],[4] Only 12 cases of intramedullary aneurysms are reported till date. [5] Hematomyelia due to SA is rare (14.7% of SA) and is associated with poor outcome. [5] In the present patient, hematomyelia in the initial MRI scan was masked by the extensive cord edema and the focal hyperintense signal at C4 level in T1W image (suggestive of bleed) was overlooked resulting in the erroneous diagnosis of demyelination. The diagnostic error could also be due to the extreme rarity of this lesion.
Figure 7: Postoperative Spinal DSA showing complete obliteartion of AVM & aneurysm and (a) right side and (b) left side injection

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Both microsurgical and endovascular obliteration are described to manage SA. However, microsurgical excision is the preferred option, if the SA is associated with an AVM (Type 1). [3],[5] Microsurgically, aneurysms can either be excised or parent vessel can be clipped. Endovascular coiling would add to increased mass within cord substance resulting itself in compressive myelopathy. In the present case, the aneurysm could be successfully obliterated by coagulating the radiculo-pial feeder vessels, thereby reliving the mass effect. It is probably not necessary to excise the aneurysm, if it is completely buried within the cord to prevent further cord damage during circumferential dissection of the aneurysm.

 
  References Top

1.Biondi A, Merland JJ, Hodes JE, Pruvo JP, Reizine D. Aneurysms of spinal arteries associated with intramedullary arteriovenous malformations. I. Angiographic and clinical aspects. AJNR Am J Neuroradiol 1992;13:913-22.  Back to cited text no. 1
    
2.Cavuþoðlu H, Ozdilmaç A, Sahin Y, Aydin Y. Isolated posterior spinal artery aneurysm causing intracranial acute subarachnoidal hemorrhage. Acta Neurochir (Wien) 2010;152:721-4.  Back to cited text no. 2
    
3.Gonzalez LF, Zabramski JM, Tabrizi P, Wallace RC, Massand MG, Spetzler RF. Spontaneous spinal subarachnoid hemorrhage secondary to spinal aneurysms: Diagnosis and treatment paradigm. Neurosurg 2005;57:1127-31.  Back to cited text no. 3
    
4.Kurita M, Endo M, Kitahara T, Fujii K. Subarachnoid haemorrhage due to a lateral spinal artery aneurysm misdiagnosed as a posterior inferior cerebellar artery aneurysm: Case report and literature review. Acta Neurochir (Wien) 2009;151:165-9.  Back to cited text no. 4
    
5.Madhugiri VS, Ambekar S, Roopesh Kumar VR, Sasidharan GM, Nanda A. Spinal aneurysms: Clinicoradiological features and management paradigms. J Neurosurg Spine 2013;19:34-48.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]

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[Pubmed] | [DOI]



 

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