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Brainstem infarction in a patient with internal carotid dissection and persistent trigeminal artery: a case report
© Iancu et al; licensee BioMed Central Ltd. 2010
Received: 22 March 2010
Accepted: 2 July 2010
Published: 2 July 2010
The primitive trigeminal artery (PTA) is the most commonly described fetal anastomosis between the carotid and vertebrobasilar circulations.
We report a 42-year-old patient presenting with internal carotid dissection, and imaging features of brainstem infarction.
Based on the imaging studies we presume occlusive carotid dissection with extensive thrombosis within a persistent trigeminal artery as the cause of this brainstem ischemia.
Several fetal anastomoses have been described between the carotid and vertebrobasilar circulations. These anastomoses regress while the P1 segments develop, but they can occasionally persist in adult age . The primitive trigeminal artery (PTA) is the most common of them representing about 85% of cases with prevalence between 0.1% and 0.76% .
We report a patient with brainstem infarction caused by a persistent PTA thrombosis secondary to occlusive dissection of the homolateral internal carotid artery (ICA).
A 42-year-old woman presented with right-side motor deficit and dysarthria. She experienced diffuse headaches, regressive episodes of ill-defined visual disturbance and right-side numbness the previous day. She reported osteopathic cervical manipulations in the previous week.
Neurologic examination revealed right-sided hemiparesis, hypoesthesia, central facial palsy and dysarthria. No Horner's sign, cranial nerve palsy, abnormal cardiac or carotid bruit were found.
A digital subtraction angiography (DSA) showed irregular localized filling defect within the distal hypoplastic BA (Figure 2B) and a flame-shaped occlusion of the left ICA which is characteristic of occlusive dissection (Figure 2C).
Further investigation did not reveal concomitant cardiac or coagulation disorders. Intravenous Heparin was initiated and the patient was discharged 3 weeks later with residual motor deficit. MRA follow-up showed persistent ICA and PTA occlusion.
Persistent PTA is often associated with intracranial aneurysms, arteriovenous malformations, carotid cavernous fistulas, Moyamoya and cerebellar hemangioblastoma [4–7]. Its clinical significance is usually uncertain but presentation may include cranial nerve dysfunction or subarachnoid haemorrhage [4–7].
Very few cases of brainstem or occipital infarction due to embolism from the ICA stenosis via persistent PTA have been reported [8, 9]. To our knowledge, this is the only case report documenting a persistent PTA thrombosis responsible for a brainstem infarction. The diagnosis was difficult since flow was completely absent in the PTA even on DSA. One clue was the diminutive aspect of both VA and proximal BA, usually found in persistent PTA. Moreover, concomitant dissection of the ICA and BA would have been unlikely.
The PTA courses from the ICA cavernous segment to the BA between the origins of the anterior inferior cerebellar arteries (AICA) and the SCA. It usually follows the trigeminal nerve in the cisternal part and Meckel's cave with some anatomic variants . The localization of the thrombosis on MRI appears consistent with the known anatomy. Embryologically, the PTA is a metameric artery arising from the first aortic arch, supplying central and peripheral nervous structures. It follows the trigeminal nerve, ending in a plexus at the trigeminal ganglion. Later on, PTA is the most important supply of the posterior structures but ultimately it regresses usually at the carotid edge . The pontine artery could give an accessory branch to the trigeminal ganglion but its main territory is the protuberance . In this case the anterolateral pontine infarction corresponds to this territory.
Stroke secondary to cervical ICA dissection generally involves embolic mechanisms instead of hypoperfusion. Since dissection rarely extends beyond the petrous segment of the ICA , we propose that extensive thrombosis, due to the dissection, is the mechanism that occluded the intracavernous carotid segment and the PTA. The DSA aspect showing small VA and proximal BA with localized endovascular filling defect is suggestive of pre-existent PTA pattern with a distal extension of the thrombosis into the PTA and BA.
This is a very rare case of MRI documented persistent PTA thrombosis responsible for brainstem infarction. In patients presenting with brainstem ischemia associated with occlusion or stenosis of the homolateral ICA, persistent PTA should be considered.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent was provided to the editorial office of this journal.
We acknowledge Dr. Marshall Wilkinson, Dr. Thomas Mammen and Dr. Andreea Nistor from the University of Manitoba, for reviewing and helping with editing this manuscript.
- Lasjaunias P, Berenstein A: Surgical neuroangiography: functional anatomy of craniofacial arteries. 1987, Berlin, Springer-Verlag, 1:View ArticleGoogle Scholar
- Piotin M, Miralbes S, Cattin F, Marchal H, Amor-Sahli M, Moulin T, Bonneville JF: MRI and MR angiography of persistent trigeminal artery. Neuroradiology. 1996, 38: 730-733. 10.1007/s002340050337.View ArticlePubMedGoogle Scholar
- Leclerc X, Lucas C, Godefroy O, Nicol L, Moretti A, Leys D, Pruvo JP: Preliminary experience using contrast-enhanced MR angiography to assess vertebral artery structure for the follow-up of suspected dissection. Am J Neuroradiol. 1999, 20: 1482-1490.PubMedGoogle Scholar
- Redekop GJ: Extracranial carotid and vertebral artery dissection: a review. Can J Neurol Sci. 2008, 35: 146-152.View ArticlePubMedGoogle Scholar
- Hurst RW, Howard RS, Zager E: Carotid cavernous fistula associated with persistent trigeminal artery: endovascular treatment using coil embolization. Skull Base Surg. 1998, 8: 225-228. 10.1055/s-2008-1058188.View ArticlePubMedPubMed CentralGoogle Scholar
- Komiyama M, Nakajima H, Nishikawa M, Yasui T, Kitano S, Sakamoto H, Fu Y: High incidence of persistent primitive arteries in moyamoya and quasi-moyamoya diseases. Neurol Med Chir. 1999, 39: 416-420. 10.2176/nmc.39.416.View ArticleGoogle Scholar
- Murai Y, Kobayashi S, Tateyama K, Teramoto A: Persistent primitive trigeminal artery aneurysm associated with cerebellar hemangioblastoma. Case report. Neurol Med Chir. 2006, 46: 143-146. 10.2176/nmc.46.143.View ArticleGoogle Scholar
- Gasecki AP, Fox AJ, Lebrun LH, Daneault N: Bilateral occipital infarctions associated with carotid stenosis in a patient with persistent trigeminal artery. The Collaborators of the North American Carotid Endarterectomy Trial (NASCET). Stroke. 1994, 25: 1520-1523.View ArticlePubMedGoogle Scholar
- Foerch C, Berkefeld J, Halbsguth A, Ziemann U, Neumann-Haefelin T: Brain stem infarction caused by proximal internal carotid artery stenosis in a patient with a persisting primitive trigeminal artery. Cerebrovasc Dis. 2006, 22: 200-202. 10.1159/000093808.View ArticlePubMedGoogle Scholar
- Abe T, Matsumoto K, Aruga T: Primitive trigeminal artery variant associated with intracranial ruptured aneurysm and cerebral arteriovenous malformation - case report. Neurol Med Chir. 1994, 34: 104-107. 10.2176/nmc.34.104.View ArticleGoogle Scholar
- The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2342/10/14/prepub
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