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ObjectiveThe aim of the study was to describe the electroclinical features of visual sensitivity in patients with familial cortical myoclonic tremor and epilepsy.MethodsWe searched the EEG database using the terms “familial cortical myoclonic tremor and epilepsy” and “visual sensitivity” over a seven-year period from March 2013 to April 2020 in the Xijing hospital, Xi’an, China. The inclusion criteria were demonstrable electroclinical visual sensitivity in the form of eye-opening sensitivity, eye-closure sensitivity, eyes-closed sensitivity and photosensitivity. Clinical, EEG and imaging records of patients were screened, and subsequently, detailed analysis of their data was undertaken.ResultsWe enrolled six patients with visual sensitivity, five of whom suffered with rare generalized tonic-clonic seizures. Neuroimaging was negative in all cases. All patients demonstrated photosensitivity; eye-opening sensitivity associated with cortical myoclonic in one patient, eyes-closed sensitivity associated with cortical myoclonic tremor status in three patients, and eye-closure sensitivity in two patients. At the last follow-up visit, cortical myoclonic tremor and epilepsy in all patients was well controlled with first-line treatment.SignificanceVisual sensitivity is therefore likely to be an important reflex trait in some patients with familial cortical myoclonic tremor and epilepsy, and should be routinely evaluated in order to better define the electroclinical features in FCMTE syndrome. [Published with video sequences].