Acta Neurol Belg DOI 10.1007/s13760-014-0352-5
LETTER TO THE EDITOR
Vertebral artery dissection as cause of sulcal artery syndrome Nadia Younan • Thierry P. Duprez Christian J. M. Sindic
•
Received: 17 June 2014 / Accepted: 4 August 2014 Ó Belgian Neurological Society 2014
Keywords Vertebral artery dissection Spinal cord infarction Sulcal artery syndrome Vertebral artery dissection (VAD) is a rare cause of ischaemic injury to the cervical spinal cord. We report the case of a young man presenting an extended right VAD leading to a stroke within both the posterior inferior cerebellar artery (PICA) area and within that of a right sulcal branch of the anterior spinal artery responsible for a Brown-Sequard-like syndrome, but with sparing of the lateral part of the medulla oblongata.
Case report In September 2013, a 33-year-old man presented with sudden onset of vertigo and dizziness increased by postural changes and accompanied by vomiting. He had no tinnitus, diplopia, hearing or visual complaints. Medical history consisted of psoriasis and smoking. He practiced regularly kick-boxing. He was initially admitted to the ENT Department for suspicion of vestibular neuronitis and was treated with tapering doses of methylprednisolone for 6 days without improvement. One week later, he complained with right nuchal pain, slight right ptosis, numbness in the left hemibody under a Th3 level, and paraesthesia in the right hand and forearm. N. Younan C. J. M. Sindic (&) Service de Neurologie, Cliniques Universitaires Saint-Luc, Avenue Hippocrate, 10, 1200 Brussels, Belgium e-mail: [email protected] T. P. Duprez Service d’Imagerie Me´dicale, Cliniques Universitaires Saint-Luc, Avenue Hippocrate, 10, 1200 Brussels, Belgium
The neurological examination at that time revealed a horizontal and rotatory nystagmus in the right gaze and a right Claude-Bernard–Horner syndrome. Perception of pain and temperature was disturbed only under the left Th3 dermatoma. Right-sided deep tendon reflexes were increased. No other neurological abnormalities were detectable. Biological investigations including CSF analysis were normal, except for dyslipidemia. Brain MRI (Fig. 1) demonstrated a subacute ischaemic stroke of the right cerebellum within the PICA territory, and a tiny right vertebral artery. There was no lesion in the lateral part of the medulla oblongata. MR angiography showed a dissection of the right vertebral artery with a mural thrombus in its V2 segment, and a complete arterial occlusion from its ostium. Spinal cord MRI revealed a right lesion of 3 mm in diameter at the level of C3, matching perfectly with the neurological examination. The patient was treated with atorvastatin and acetylsalicylic acid. In July 2014, he had recovered from balance disturbances, but not from left thermoanalgesia. A control MR angiography showed no recanalization of the right vertebral artery.
Discussion Spinal cord infarction (SCI) is rarely the consequence of a VAD. In a series of 111 patients with such a dissection, only two presented with spinal manifestations [1]. More recently, Hsu et al. [2] made a systematic review of the literature and found 17 cases (nine men and eight women) with VAD-associated SCI. The mean age was 40 years. The main symptoms were headache or neck pain (88 %), presence of a sensory level (76 %), and a Brown-Sequard
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Acta Neurol Belg Fig. 1 a Axial-transverse FLAIR view showing an acute stroke in the right PICA territory (arrows). Note the sparing of the lateral part of the medulla oblongata. b Unenhanced fat-saturated T1weighted axial-transverse view showing a highly hyperintense mural thrombus rich in methemoglobin within the right vertebral artery at the level of C4 (arrow). c Right parasagittal STIR image showing a small hyperintense lesion at the level of C3 consistent with an ischaemic injury (arrow). d Axial-transverse T2-weighted image at C3 level confirming the presence of a small rightsided ischaemic focus within cord (arrow)
syndrome (53 %). The most common regions of dissections were at the V1 or proximal V2 segments, and the infarcted area was mainly located at C2–C5 levels. Posterior spinal artery infarction was noted in 29 % of patients, spinal sulcal infarction in 42 %, and anterior spinal artery infarction in 29 %. Indeed, one vertebral artery frequently supplies the dominant feeder to the anterior spinal artery. Therefore, compromise of one vertebral artery often causes bilateral ischaemic injury to the cord [3]. In turn, unilateral cervical cord involvement following ipsilateral VAD is uncommon, and results from infarction within the territory of the sulcal artery, which is the penetrating branch of the anterior spinal artery. The clinical features of the condition are a Brown-Sequard-like syndrome associated as in our case to a Claude-Bernard–Horner syndrome [4, 5]. In the review of Hsu et al. [2], smoking, age above 50 years, and anterior spinal artery infarction were
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associated with poor outcome, while sulcal artery infarctions had a better prognosis. Only seven cases of sulcal artery syndromes have been reported with similar characteristics as in our patient [6– 12] and prognosis was indeed good in most of them, although a delayed painful spinothalamic syndrome remains possible. In our patient, the regular practice of kick-boxing may have favoured repetitive microtraumatisms of the vertebral arteries walls progressively leading to VAD. Unexpectedly lateral neck pain appeared only after a 2 weeks’ delay after balance disorder. No other potential cause of VAD was present. It should be noted that spontaneous dissections affect more often women than men (77 %) and are bilateral in 60 % of cases [13]. In conclusion, VAD-associated SCI should be considered in a patient presenting neck pain or headache combined with a Brown-Sequard syndrome and/or a sensory level.
Acta Neurol Belg Conflict of interest
None.
References 1. Crum B, Mokri B, Filgham J (2000) Spinal manifestations of vertebral artery dissection. Neurology 55:304–306 2. Hsu CY, Cheng CY, Lee JD et al (2013) Clinical features and outcomes of spinal cord infarction following vertebral artery dissection: a systematic review of the literature. Neurol Res 35:676–683 3. Machnowska M, Moien-Afshari F, Voll C, Wiebe S (2008) Partial anterior cervical cord infarction following vertebral artery dissection. Can J Neurol Sci 35:674–677 4. Gutowski NJ, Murphy RP, Beale DJ (1992) Unilateral upper cervical posterior spinal artery syndrome following sneezing. J Neurol Neurosurg Psychiatry 55:841–843 5. Goldsmith P, Rowe D, Jager R, Kapoor R (1998) Focal vertebral artery dissection causing Brown-Se´quard’s syndrome. J Neurol Neurosurg Psychiatry 64:415–416 6. Cheshire WP, Santos CC, Massey EW et al (1996) Spinal cord infarction: etiology and outcome. Neurology 47:321–330
7. Weidauer S, Claus D, Gartenschla¨ger M (1999) Spinal sulcal artery syndrome due to spontaneous bilateral vertebral artery dissection. J Neurol Neurosurg Psychiatry 67:550–551 8. Laufs H, Weidauer S, Heller C et al (2004) Hemi-spinal cord infarction due to vertebral artery dissection in congenital afibrinogenemia. Neurology 63:1522–1523 9. Li Y, Jenny D, Bemporad JA, Liew CJ, Castaldo J (2010) Sulcal artery syndrome after vertebral artery dissection. J Stroke Cerebrovasc Dis 19:333–335 10. Takahashi PG, Cury RG, Lopes CG, Simabukuro MM, Marchiori PE (2012) Unilateral non traumatic vertebral artery dissection with cervical spinal cord infarction. Arq Neuropsiquiatr 70:162 11. Muscat P, Lidov M, Nahar T, Tuhrim S, Weinberger J (2001) Vertebral artery dissection in Turner’s syndrome: diagnosis by magnetic resonance imaging. J Neuroimaging 11:50–53 12. Bas DF, Oguz KK, Yavuz K, Topcuoglu MA (2009) Spinal cord infarction in congenital afibrinogenemia: a case report and review of the literature. J Stroke Cerebrovasc Dis 18:298–303 13. Labouret P, Tranchant C, Jesel M et al (1993) Mye´lopathie cervicale symptomatique d’une dissection de l’arte`re verte´brale extracraˆnienne. Rev Neurol 149:559–561
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LETTER TO THE EDITOR
Vertebral artery dissection as cause of sulcal artery syndrome Nadia Younan • Thierry P. Duprez Christian J. M. Sindic
•
Received: 17 June 2014 / Accepted: 4 August 2014 Ó Belgian Neurological Society 2014
Keywords Vertebral artery dissection Spinal cord infarction Sulcal artery syndrome Vertebral artery dissection (VAD) is a rare cause of ischaemic injury to the cervical spinal cord. We report the case of a young man presenting an extended right VAD leading to a stroke within both the posterior inferior cerebellar artery (PICA) area and within that of a right sulcal branch of the anterior spinal artery responsible for a Brown-Sequard-like syndrome, but with sparing of the lateral part of the medulla oblongata.
Case report In September 2013, a 33-year-old man presented with sudden onset of vertigo and dizziness increased by postural changes and accompanied by vomiting. He had no tinnitus, diplopia, hearing or visual complaints. Medical history consisted of psoriasis and smoking. He practiced regularly kick-boxing. He was initially admitted to the ENT Department for suspicion of vestibular neuronitis and was treated with tapering doses of methylprednisolone for 6 days without improvement. One week later, he complained with right nuchal pain, slight right ptosis, numbness in the left hemibody under a Th3 level, and paraesthesia in the right hand and forearm. N. Younan C. J. M. Sindic (&) Service de Neurologie, Cliniques Universitaires Saint-Luc, Avenue Hippocrate, 10, 1200 Brussels, Belgium e-mail: [email protected] T. P. Duprez Service d’Imagerie Me´dicale, Cliniques Universitaires Saint-Luc, Avenue Hippocrate, 10, 1200 Brussels, Belgium
The neurological examination at that time revealed a horizontal and rotatory nystagmus in the right gaze and a right Claude-Bernard–Horner syndrome. Perception of pain and temperature was disturbed only under the left Th3 dermatoma. Right-sided deep tendon reflexes were increased. No other neurological abnormalities were detectable. Biological investigations including CSF analysis were normal, except for dyslipidemia. Brain MRI (Fig. 1) demonstrated a subacute ischaemic stroke of the right cerebellum within the PICA territory, and a tiny right vertebral artery. There was no lesion in the lateral part of the medulla oblongata. MR angiography showed a dissection of the right vertebral artery with a mural thrombus in its V2 segment, and a complete arterial occlusion from its ostium. Spinal cord MRI revealed a right lesion of 3 mm in diameter at the level of C3, matching perfectly with the neurological examination. The patient was treated with atorvastatin and acetylsalicylic acid. In July 2014, he had recovered from balance disturbances, but not from left thermoanalgesia. A control MR angiography showed no recanalization of the right vertebral artery.
Discussion Spinal cord infarction (SCI) is rarely the consequence of a VAD. In a series of 111 patients with such a dissection, only two presented with spinal manifestations [1]. More recently, Hsu et al. [2] made a systematic review of the literature and found 17 cases (nine men and eight women) with VAD-associated SCI. The mean age was 40 years. The main symptoms were headache or neck pain (88 %), presence of a sensory level (76 %), and a Brown-Sequard
123
Acta Neurol Belg Fig. 1 a Axial-transverse FLAIR view showing an acute stroke in the right PICA territory (arrows). Note the sparing of the lateral part of the medulla oblongata. b Unenhanced fat-saturated T1weighted axial-transverse view showing a highly hyperintense mural thrombus rich in methemoglobin within the right vertebral artery at the level of C4 (arrow). c Right parasagittal STIR image showing a small hyperintense lesion at the level of C3 consistent with an ischaemic injury (arrow). d Axial-transverse T2-weighted image at C3 level confirming the presence of a small rightsided ischaemic focus within cord (arrow)
syndrome (53 %). The most common regions of dissections were at the V1 or proximal V2 segments, and the infarcted area was mainly located at C2–C5 levels. Posterior spinal artery infarction was noted in 29 % of patients, spinal sulcal infarction in 42 %, and anterior spinal artery infarction in 29 %. Indeed, one vertebral artery frequently supplies the dominant feeder to the anterior spinal artery. Therefore, compromise of one vertebral artery often causes bilateral ischaemic injury to the cord [3]. In turn, unilateral cervical cord involvement following ipsilateral VAD is uncommon, and results from infarction within the territory of the sulcal artery, which is the penetrating branch of the anterior spinal artery. The clinical features of the condition are a Brown-Sequard-like syndrome associated as in our case to a Claude-Bernard–Horner syndrome [4, 5]. In the review of Hsu et al. [2], smoking, age above 50 years, and anterior spinal artery infarction were
123
associated with poor outcome, while sulcal artery infarctions had a better prognosis. Only seven cases of sulcal artery syndromes have been reported with similar characteristics as in our patient [6– 12] and prognosis was indeed good in most of them, although a delayed painful spinothalamic syndrome remains possible. In our patient, the regular practice of kick-boxing may have favoured repetitive microtraumatisms of the vertebral arteries walls progressively leading to VAD. Unexpectedly lateral neck pain appeared only after a 2 weeks’ delay after balance disorder. No other potential cause of VAD was present. It should be noted that spontaneous dissections affect more often women than men (77 %) and are bilateral in 60 % of cases [13]. In conclusion, VAD-associated SCI should be considered in a patient presenting neck pain or headache combined with a Brown-Sequard syndrome and/or a sensory level.
Acta Neurol Belg Conflict of interest
None.
References 1. Crum B, Mokri B, Filgham J (2000) Spinal manifestations of vertebral artery dissection. Neurology 55:304–306 2. Hsu CY, Cheng CY, Lee JD et al (2013) Clinical features and outcomes of spinal cord infarction following vertebral artery dissection: a systematic review of the literature. Neurol Res 35:676–683 3. Machnowska M, Moien-Afshari F, Voll C, Wiebe S (2008) Partial anterior cervical cord infarction following vertebral artery dissection. Can J Neurol Sci 35:674–677 4. Gutowski NJ, Murphy RP, Beale DJ (1992) Unilateral upper cervical posterior spinal artery syndrome following sneezing. J Neurol Neurosurg Psychiatry 55:841–843 5. Goldsmith P, Rowe D, Jager R, Kapoor R (1998) Focal vertebral artery dissection causing Brown-Se´quard’s syndrome. J Neurol Neurosurg Psychiatry 64:415–416 6. Cheshire WP, Santos CC, Massey EW et al (1996) Spinal cord infarction: etiology and outcome. Neurology 47:321–330
7. Weidauer S, Claus D, Gartenschla¨ger M (1999) Spinal sulcal artery syndrome due to spontaneous bilateral vertebral artery dissection. J Neurol Neurosurg Psychiatry 67:550–551 8. Laufs H, Weidauer S, Heller C et al (2004) Hemi-spinal cord infarction due to vertebral artery dissection in congenital afibrinogenemia. Neurology 63:1522–1523 9. Li Y, Jenny D, Bemporad JA, Liew CJ, Castaldo J (2010) Sulcal artery syndrome after vertebral artery dissection. J Stroke Cerebrovasc Dis 19:333–335 10. Takahashi PG, Cury RG, Lopes CG, Simabukuro MM, Marchiori PE (2012) Unilateral non traumatic vertebral artery dissection with cervical spinal cord infarction. Arq Neuropsiquiatr 70:162 11. Muscat P, Lidov M, Nahar T, Tuhrim S, Weinberger J (2001) Vertebral artery dissection in Turner’s syndrome: diagnosis by magnetic resonance imaging. J Neuroimaging 11:50–53 12. Bas DF, Oguz KK, Yavuz K, Topcuoglu MA (2009) Spinal cord infarction in congenital afibrinogenemia: a case report and review of the literature. J Stroke Cerebrovasc Dis 18:298–303 13. Labouret P, Tranchant C, Jesel M et al (1993) Mye´lopathie cervicale symptomatique d’une dissection de l’arte`re verte´brale extracraˆnienne. Rev Neurol 149:559–561
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