Case Report Symptomatic Spinal Migration of Subarachnoid Hemorrhage due to Ruptured Intradural Vertebral Artery Aneurysm ¨ ¨ un ¨ Yılmaz Ovalı, MD, Gurhan ¨ Gulg Adam, MD, Celal C ¸ ınar, MD, Halil Bozkaya, MD, Cem C ¸ allı, MD, Omer Kitis¸, MD, ˙Ismail Oran, MD From the Department of Radiology, Celal Bayar University, Manisa, Turkey (GYO); Department of Radiology, Canakkale Onsekiz Mart University, Canakkale, Turkey (GA); and ¨ IO). Department of Radiology, Ege University, Izmir, Turkey (CC ¸ , HB, CC ¸ , OK,

ABSTRACT A 55-year-old patient was admitted to the hospital with severe acute back pain. Thoracolumbar magnetic resonance (MR) imaging showed hemorrhage in subarachnoidalsubdural space. On cranial MR imaging and MR angiography, an aneurysm was suspected in the V4 segment of the right vertebral artery. Angiography showed a fusiform dissecting aneurysm in the V4 segment of right vertebral artery. The final diagnosis was ruptured V4 segment aneurysm with subsequent symptomatic migration of hemorrhage into the spinal subarachnoidal-subdural space. The patient was treated endovascularly by coil occlusion of both the aneurysm and vertebral artery. This rare cause and possible mechanisms for spinal migration of intracranial hemorrhage after aneurysmal rupture is discussed.

Keywords: Aneurysm, subarachnoid hemorrhage, vertebral artery. Acceptance: Received April 24, 2014, and in revised form July 18, 2014. Accepted for publication September 13, 2014. Correspondence: Address correspondence to Gurhan Adam, MD, ¨ Assistant Professor, Department of Radiology, Canakkale Onsekiz Mart University, Canakkale, Turkey. E-mail: [email protected] J Neuroimaging 2015;25:668-670. DOI: 10.1111/jon.12189

Introduction Spinal subarachnoid hemorrhage (SAH) and subdural hematoma are most frequently caused by iatrogenic or traumatic mechanisms or by defective coagulation.1 Rarely, isolated anterior spinal artery aneurysm may rupture into the spinal subarachnoid space.2 Spinal migrating hemorrhage has been reported infrequently after intracranial operations.3 Intracranial intradural aneurysm rupture associated with spinal migration of hemorrhage is another very rare cause leading to the presence of hemorrhage in the spinal subarachnoid-subdural space; there have been only a few case reports.4 We describe clinical presentation, magnetic resonance (MR) imaging, and angiography findings of a patient with migrating spinal hemorrhage from a ruptured intracranial aneurysm.

Case Report A 55-year-old female patient was admitted to the hospital with severe acute back pain radiating to both (but predominantly to left) buttocks and numbness in both legs that began 2 days ago. On further conversation, she additionally mentioned that she had severe headache that resolved spontaneously within a week 3 weeks ago. She had paresthesia on both lower extremities; her neurologic examination was otherwise normal. Thoracolumbar MR imaging showed high signal intensities on T1-weighted and low signal intensities on T2-weighted images consistent with hemorrhage in subarachnoidal space beginning

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at the level of T10 to the end of dural sac. Axial images revealed patchy involvement of subarachnoid space around the spinal cord especially in the left side (Figs 1A and 1B). On cranial MR imaging and angiography, an aneurysm was suspected in the V4 segment of right vertebral artery. On catheter angiography, a fusiform-shaped aneurysm accompanied by internal dissection flap was discovered in the V4 segment of right vertebral artery in vicinity of the right PICA origin (Fig 2). The final diagnosis was made as ruptured V4 segment aneurysm with subsequent symptomatic migration of hemorrhage into the spinal subarachnoidal space. The decision was to embolize the aneurysm with parent vessel occlusion just before the PICA origin. Under general anesthesia, the aneurysm together with parent vessel was occluded with detachable coils. The left vertebral angiogram confirmed the patency of vertebrobasilary system and retrograde filling of the right PICA as well as small remnant of aneurysm at superior aspect (Fig 3A). The patient was discharged 3 days later. Her pain gradually subsided within 2 weeks. One month later, thoracolumbar MRI showed disappearance of all hemorrhagic signal intensities. A 6-month control angiography demonstrated persistent occlusion of the aneurysm and disappearance of small aneurysm remnant (Fig 3B).

Discussion Spinal hematomas are rare entities that require early diagnosis to prevent spinal cord compression syndrome. Most cases of

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Fig 1. (A) Sagittal and (B) axial T1-weighted image of the thoracolumbar spine showing high signal intensity in the subarachnoid space due to hemorrhage.

Fig 2. Right vertebral artery angiography. Fusiform dissecting aneurysm was seen in the V4 segment of right vertebral artery close to the right PICA origin.

spinal hematoma have a multifactorial cause, and in up to onethird of patients, no factor can be identified as the cause of the bleeding.1 On MR imaging, the differential diagnosis of spinal hemorrhage includes lymphoproliferative disorders or neoplasms. Isointensity or hypointensity on T1-weighted images in acute phase of hematomas is nonspecific and can be seen in association with other conditions, including lymphoma and metastasis. Contrast enhancement of these two conditions is not always sufficient for differential diagnosis because it can also be seen in spinal hematomas due to hyperemia of dura mater resulting in thickening of adjacent meninges. It is easier to diagnose hematoma on subacute stage having high T1 signal as we observed in our case.

Intracranial vertebral artery dissection has 3 clinical presentations: ischemia, hemorrhage, and mass effect. Imaging findings of intracranial vertebral artery dissections vary according to clinical presentation. Dissections located on the intradural V4 segment are uncommon, and presentation with SAH is rare. In a study by Arnold et al5 comprising 169 patients with 195 vertebral dissections, only 22 dissections (11%) were located in the intradural V4 segment and 6 (4%) occurred with SAH. SAH related to dissecting V4 segment aneurysm is generally reported to be very rare comprising well below 1% of all ruptured intradural aneurysms.6,7 Cranial MR imaging revealed no positive findings consistent with SAH in our patient in contrast to widespread hemorrhage in the thoracolumbar subarachnoid space. This may be due to the 3-week interval between headache and MR examination. Interestingly, the patient’s first clinical symptom that led her to the hospital was the radiating back pain accompanying with numbness in both legs. These spinal symptoms were likely to be related to the direct effect of hemorrhage on the lower spinal radicular nerves. The mechanism of spinal SAH after rupture of intracranial subarachnoid aneurysm is simple; migration or extension of blood from the intracranial to spinal level. It occurs probably more likely in patients with significant amount of subarachnoid blood or after rapid ambulation of the patient. Blood migration in reversed direction, from spinal subarachnoid space to intracranial subarachnoid space, is also possible after rupture of spinal vascular lesions.8 The migration of blood to spinal subdural space after rupture of the intracranial intradural aneurysm has also been reported in the literature, however, the pathologic mechanism remains unclarified. One explanation is that it may be the result of hemorrhage under high pressure, leading to pia-arachnoid rupture and extravasation of blood into the subdural space.4 Kobayashi et al claimed that a clot large enough could rupture through the arachnoid into the subdural space.9 Bernser et al defined the combination of subarachnoid and subdural spinal hemorrhage in the same patient.10 They mentioned that the rare combination of subarachnoid and subdural

Ovalı et al: Ruptured Intradural Vertebral Artery Aneurysm

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Fig 3. (A) Left vertebral angiogram showed the patency of vertebrobasilary system, retrograde filling of the right PICA, and small remnant of aneurysm at superior aspect. (B) Control angiography demonstrated persistent occlusion of the aneurysm and disappearance of aneurysm remnant. Note the remodeling (narrowing) of the patent distal right vertebral artery due to diminished (and retrograde) flow.

spinal hematomas may have been caused either by rupture of a small vessel in the arachnoid membrane or by rupture of the arachnoid membrane itself, secondary to a massive hemorrhage in the subarachnoid space. Yamaguchi et al defined a mechanism in their patient that spinal subdural hematoma migrated from intracranial subdural space due to low CSF pressure because of continuous drainage and intrathecal thrombolytic therapy.11 When spinal hemorrhage is present, the progressive neurological deficit requires urgent surgery. However, in some cases whose neurological findings are not severe and/or progressive, like our case, conservative management is an option. In such cases, to control the spontaneous resolution, repeat MR examination should be performed. In conclusion, symptomatic spinal SAH may be the first complaint of patient with intracranial intradural ruptured aneurysm associated with migrating hemorrhage. The spinal migration of hemorrhage from intracranial subarachnoid space should be kept in mind in differential diagnosis of spinal intradural hemorrhage.

References 1. Kreppel D, Antoniadis G, Seeling W. Spinal hematoma: a literature survey with meta-analysis of 613 patients. Neurosurg Rev 2003;26:149. 2. Berlis A, Scheufler K, Schmahl C, et al. Solitary spinal artery aneurysms as a rare source of spinal subarachnoid hemorrhage:

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potential etiology and treatment strategy. AJNR Am J Neuroradiol 2005;26:405-410. Kim MS, Lee CH, Lee SJ, et al. Spinal subdural hematoma following intracranial aneurysm surgery: four case reports. Neurol Med Chir (Tokyo) 2007;47:22-25. Gilad R, Fatterpekar GM, Johnson DM, et al. Migrating subdural hematoma without subarachnoid hemorrhage in the case of a patient with a ruptured aneurysm in the intrasellar anterior communicating artery. AJNR Am J Neuroradiol 2007;28:20142016. Arnold M, Bousser MG, Fahrni G, et al. Vertebral artery dissection: presenting findings and predictors of outcome. Stroke 2006;37:2499-2503. Peluso JPP, Van Rooij WJ, Sluzewski M, et al. Endovascular treatment of symptomatic intradural vertebral dissecting aneurysms. AJNR Am J Neuroradiol 2008;29:102-106. Yoon W, Seo JJ, Kim TS, et al. Dissection of the V4 segment of the vertebral artery: clinicoradiologic manifestations and endovascular treatment. Eur Radiol 2007;17:983-993. van Beijnum J, Straver DCG, Rinkel GJE, et al. Spinal arteriovenous shunts presenting as intracranial subarachnoid hemorrhage. J Neurol 2007;254:1044-1051. Kobayashi N, Abe T, Imaizumi Y. Lumbosacral subdural hematoma following a ruptured aneurysmal subarachnoid hemorrhage. Neurol India 2007;55:431. Bernser RA, Hoogenraad TV. A spinal hematoma occurring in the subarachnoid as well as in the subdural space in a patient treated with anticoagulants. Clin Neurol Neurosurg 1992;94:35-37. Yamaguchi S, Hida K, Akino M, et al. Spinal subdural hematoma: a sequela of a ruptured intracranial aneurysm? Surg Neurol 2003;59:408-412.

Symptomatic Spinal Migration of Subarachnoid Hemorrhage due to Ruptured Intradural Vertebral Artery Aneurysm.

A 55-year-old patient was admitted to the hospital with severe acute back pain. Thoracolumbar magnetic resonance (MR) imaging showed hemorrhage in sub...
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