Persistent Trigeminal Artery and Dural Arteriovenous Fistula: A Case Report

Abstract

Introduction

The persistent trigeminal artery is a carotid-vertebrobasilar anastomosis, mostly unilateral. Embryonic arteries typically disappear during prenatal development, but in rare instances, they may persist. The prevalence of this anastomosis in adulthood is 0.4% in individuals without vascular abnormalities.

Case Report

A 41-year-old female patient sought emergency care with complaints of neck pain associated with a headache, starting one month ago, and worsening associated with paresthesia in the left upper limb. The magnetic resonance angiography of the skull revealed a left sigmoid sinus dural arteriovenous fistula (dAVF), irrigated by the left middle meningeal and ipsilateral occipital arteries. The same examination revealed the presence of a persistent trigeminal artery originating from the cavernous segment of the right internal carotid artery.

Discussion

The persistent trigeminal artery is associated with other arteriovenous abnormalities. The most common symptom is trigeminal neuralgia. Complications may result from aneurysm ruptures, and the treatment of the condition still depends on the associated clinical presentations.

Conclusion

The persistent trigeminal artery is a rare incidental finding in medical practice, as are studies related to the topic. Recognizing this abnormality in affected patients is essential, as there have been reports of complications related to the finding, especially before surgical procedures. Therefore, there is a need to expand the literature data on the persistent trigeminal artery and its appropriate management.

 

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Resumo

Introdução

A artéria trigeminal persistente é uma anastomose carótida-vertebrobasilar, em sua maioria unilateral. As artérias embrionárias costumam desaparecer ao longo da evolução pré-natal, mas em ocasiões raras tendem a persistir. A prevalência na vida adulta dessa anastomose é de 0,4% em pessoas que não apresentam anormalidades vasculares.

Relato de Caso

Paciente, sexo feminino, 41 anos. Buscou atendimento em pronto atendimento com queixas de cervicalgia associada à cefaleia, iniciada há um mês e piora associada à parestesia em membro superior esquerdo. Em angioressonância magnética, o crânio apresentou fístula arteriovenosa dural (dAVF) no seio sigmoideo esquerdo, com irrigação pela artéria meningeia média esquerda e occipital ipsilateral. Neste mesmo exame, foi evidenciada a presença de artéria trigeminal persistente de origem em segmento cavernoso da artéria carótida interna direita.

Discussão

A artéria trigeminal persistente é associada a outras anormalidades arteriovenosas. O sintoma mais comum é a neuralgia trigeminal. As complicações podem decorrer de rupturas de aneurismas e o tratamento da condição ainda depende do quadro associado e suas apresentações clínicas.

Conclusão

A artéria trigeminal persistente é um achado incidental raro na prática médica, assim como os estudos relacionados ao tema. O reconhecimento da anormalidade, em pacientes portadores, se torna essencial, pois já ocorreram relatos de complicações relacionadas ao achado, principalmente antes de procedimentos cirúrgicos. Sendo assim, é necessário ampliar em literatura dados sobre a artéria trigeminal remanescente e seu manejo adequado.

 

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Keywords

persistent trigeminal artery - remnant trigeminal artery - persistent embryonic arteries - dural arteriovenous fistula - neurovascular malformation - cerebral angiography

Palavras-chave

artéria trigeminal persistente - artéria trigeminal remanescente - artérias embrionárias persistentes - fístula arteriovenosa dural - malformação neurovascular - angiografia cerebral

Introduction

Persistent trigeminal artery (PTA) is a carotid-vertebrobasilar anastomosis, predominantly unilateral. During embryonic development, there are four connections between the carotid and basilar arteries: the trigeminal, optic, hypoglossal, and proatlantal.[1] Embryonic arteries typically disappear during prenatal evolution, but in rare instances, they persist. The persistence of the trigeminal artery was first described in 1844 by Richard Quinn. The prevalence of this anastomosis in adulthood is 0.4% in individuals without vascular abnormalities, making it the most common remnant connection, accounting for 80% of the cases.[2] [3]

There is an anatomical classification for this anastomosis, known as the Saltzman classification, comprising subtypes I, II, and III. Type I involves the connection of the internal carotid with the basilar artery. Type II refers to trigeminal-cerebellar arteries that can be further divided based on their irrigation into superior, anterior, and posterior, and type III relates to the union with other primitive arteries, forming a complex network.[2] [4] In addition to this classification, there is the Salas classification, which divides it into lateral and medial, with the former being petrosal and the latter being sphenoidal.[5]

This study reports the case of a patient undergoing investigation for headaches who presented with a persistent trigeminal artery on imaging. Due to the rarity of this occurrence, the study aims to analyze and discuss information about the case, addressing relevant data found in the literature for the diagnosis and definition of management strategies.

 

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Case Report

A female patient, 41 years old, presented to the emergency department with complaints of neck pain associated with a headache that started a month ago and worsened with paresthesia in the left upper limb. She denied any comorbidities, medication use, smoking, or relevant medical history. Upon physical examination at admission, she did not exhibit cognitive deficits or difficulties in performing daily work activities. Upper cortical functions were preserved, pupils were isocoric and reactive to light, and there were no focal neurological deficits or abnormalities in cranial nerve examinations. Admission laboratory tests revealed anemia (Hb 11), electrolyte disturbance (hyponatremia and hypocalcemia), and lymphocytosis.

Upon performing an angiotomography, what appeared to be four aneurysms were identified in the region of the right internal carotid artery, including the cavernous and ophthalmic segments. ([Fig. 1])

Fig. 1 Angiotomography showing apparent saccular aneurysms in the region of the right internal carotid artery, one posteriorly oriented measuring 0.7 × 0.5 cm, two in the cavernous segment, the former anterior measuring 0.5 × 0.3 cm, and the latter posterior measuring 0.4 × 0.2 cm. The last one is in the ophthalmic segment, posteriorly oriented, measuring 0.5 × 0.4 cm. Source: The authors.

On magnetic resonance angiography of the skull and cervical spine, a dural arteriovenous fistula (dAVF) was identified in the left sigmoid sinus, irrigated by the middle meningeal and ipsilateral occipital arteries. The saccular aneurysms observed before the angiographic study were false positives. The image showed the presence of a persistent trigeminal artery originating in the cavernous segment of the right internal carotid artery, with a parasselar course converging into the basilar artery in its upper third (Saltzman I). Hypoplasia downstream was observed, with no sacculation of arterial origin. The same examination revealed hypoplasia of both posterior communicating arteries. ([Figs. 2] and [3])

Fig. 2 Magnetic resonance angiography showing a dural arteriovenous fistula in the left sigmoid sinus. Source: The authors.

Fig. 3 3D reconstruction on magnetic resonance angiography displaying a persistent trigeminal artery originating from the cavernous segment of the right internal carotid artery. Source: The authors.

An angiography was performed, revealing thrombosis in the left superior sagittal and transverse sinuses, along with a dural fistula in the left sigmoid sinus. According to the Cognard classification of dural arteriovenous fistulas, it was a benign type I fistula, equivalent to type I in the Borden classification. The recommended course of action was urgent embolization of the dural fistula due to the thrombosis in the sagittal and left transverse sinuses, which could lead to the progression of the fistula. The patient was symptomatic, with a correlation between the clinical presentation and radiologic findings, including the risk of bleeding and the necessity for full anticoagulation due to thrombosis. The embolization procedure was successful, and follow-up angiography was scheduled for three months later. ([Figs. 4] [5] [6])

Fig. 4 Persistent trigeminal artery in digital angiography. Source: The authors.

Fig. 5 Dural arteriovenous fistula before embolization. Source: The authors.

Fig. 6 Post-embolization image of the fistula. Source: The authors.

 

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Discussion

The persistent trigeminal artery (PTA) is a carotid-vertebrobasilar anastomosis that is predominantly unilateral.[1] Its existence may be related to other abnormalities such as aneurysms, arteriovenous malformations, fistulas, and Moyamoya disease. Reported complications include ischemic stroke, aneurysm ruptures, emboli, and the fistulas themselves.[5] [6]

In a study involving 33 patients, the most common location of appearance was the cavernous part of the internal carotid artery. Five patients presented with trigeminal neuralgia symptoms, and six cases were associated with intracranial aneurysms.[7] In another study, trigeminal neuralgia occurred in 2.2% of patients with PTA, and the relationship may be associated with neural compression.[8] A study noted a 29% association between PTA and aneurysms. Additionally, there is a possibility of PTA being responsible for forming fistulas, such as cavernous-carotid fistulas.[9]

Diagnosis can be made through magnetic resonance angiography, computed tomography angiography, or catheter angiography.[4] Treatment is directed toward the patient's symptoms or complications. For trigeminal neuralgia, microvascular decompression is a treatment option, while for fistulas, endovascular embolization is a choice.[8]

Persistent trigeminal artery lacks a well-defined etiology, mostly presenting unilaterally in the cavernous segment of the internal carotid artery. The most common associated symptom is trigeminal neuralgia. There is a relationship with other arteriovenous abnormalities, as seen in the reported patient's case. Complications may arise from aneurysm ruptures, and the treatment of the condition depends on the associated clinical presentations.

 

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Conclusion

Persistent trigeminal artery is a rare incidental finding in medical practice, much like studies related to the subject. Recognizing this anomaly in affected patients becomes essential, as there are reports of complications associated with the finding, particularly before surgical procedures. Therefore, there is a need to expand the literature data on persistent trigeminal arteries and their proper management.

 

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Conflict of Interest

None.

• References

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• 9 Shah KA, Katz JM. Ruptured Persistent Trigeminal Artery-Basilar Artery Junction Aneurysm: Case Report and Review of Literature. World Neurosurg 2020; 133: 159-162

Publication History

Received: 30 January 2024

Accepted: 20 March 2025

Article published online:
16 July 2025