To describe a rare clinical presentation of Artery of Percheron infarction, emphasizing its unique symptomatology that differs from the classical triad of altered mental status, vertical gaze palsy, and memory impairment. By detailing this unusual manifestation, the report aims to expand the current understanding of AOP stroke phenotypes, aid in earlier recognition, and underscore the diagnostic challenges given the subtle or atypical presentation.

Bilateral thalamic infarction is a rare stroke syndrome that can occur due to occlusion of the artery of Percheron (AOP), an uncommon anatomical variation supplying both paramedian thalami and the rostral midbrain. The clinical presentation is often atypical and may resemble neuromuscular or other disorders making diagnosis challenging.

An initial concern for myasthenia gravis was ruled out after imaging revealed bilateral thalamic infarcts. Workup for embolic sources, including transthoracic echocardiogram and hypercoagulability testing, was unremarkable. He was treated with dual antiplatelet therapy and secondary prevention measures.

This case highlights the diagnostic complexity of AOP infarction, particularly when it mimics neuromuscular conditions, and underscores the importance of considering stroke in the differential diagnosis of bilateral ptosis and ocular movement abnormalities.