Persistent trigeminal artery (PTA) is an uncommon embryological carotid-basilar vascular anastomosis, persisting into adulthood in approximately 0.1–1.0% of individuals. PTA is clinically significant due to its impact on cerebral hemodynamics and association with various cerebrovascular pathologies, including ischemic stroke, aneurysms, and arterial dissection. Early recognition is important to guide diagnostic and therapeutic strategies.

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A case of 45-year-old female with no prior comorbidities who presented with two episodes of generalized tonic-clonic seizures followed by severe headache, bilateral sensory symptoms, and progressive neurological decline including aphasia and hemiplegia.

On arrival MRI demonstrated restricted diffusion in the left fronto-parieto-temporo-occipital regions and bilateral thalami with hemorrhagic transformation, consistent with acute ischemic infarcts. CT Angiography revealed the presence of a persistent trigeminal artery.

The PTA influenced the ischemic pattern and collateral circulation, complicating patient management. Literature review confirmed PTA as a rare but clinically relevant variant, associated with complex vascular presentations and increased stroke risk depending on its anatomic variant and associated vascular abnormalities.

The patient was treated with standard medical therapy, modified Rankin scale at discharge was zero.

PTA in adults, while rare, constitutes an important consideration in stroke etiology and management. Advanced neuroimaging plays a pivotal role in the detection and characterization of PTA. Awareness of this vascular anomaly is crucial to optimize diagnostic accuracy, tailor treatment approaches, and improve clinical outcomes in neurovascular patients.