Background <p>Acute transient myopia with shallowing of the anterior chamber is a rare idiosyncratic response to many systemic and topical medications, including topiramate. Several such cases have been reported in the past as a chronic complication, but are less frequently reported as a bilateral acute myopia. We report a case of acute transient myopia due to Topiramate – a drug used for epilepsy and migraine prophylaxis.</p> Case presentation <p>A 22 years old male recently diagnosed with epilepsy started on topiramate for 8 days after which he developed sudden drop in vision bilaterally. He was diagnosed with acute transient bilateral myopia based clinical findings and exclusion. The medication was stopped immediately and the patient showed significant improvement after the 2nd day from the discontinuation.</p> Literature review <p>A review of 9 studies, which included 16 cases for topiramate induced acute myopia encompassing 18 patients revealed a predominance of females (12). Symptoms included sudden myopia, anterior chamber shallowing, ciliochoroidal effusion, and elevated intraocular pressure. Notably, discontinuation of topiramate resulted in symptom reversal. These findings underscore the need for awareness of acute bilateral myopia secondary to topiramate as a rare adverse effect across diverse patient demographics and dosages.</p> Conclusion <p>This case and the existing literature emphasize the significance of educating patients and the necessity for immediate discontinuation of the drug when ocular symptoms happen. This case also outlines the importance of interdisciplinary collaboration in addressing the adverse effects linked to neurological medications such as topiramate. We present an additional case that illustrates the ophthalmic complications associated with topiramate.</p>