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<p><strong>Objective</strong> To discuss the major indicing factors, clinical features, therapy and prognosis of toothbrushing-induced epilepsy.  <strong>Methods </strong> The clinical manifestations, auxiliary examinations, treatment and prognosis were studied in 2 patients of toothbrushing-induced epilepsy, and related literatures were reviewed.  <strong>Results </strong> Two sisters with toothbrushing-induced epilepsy were recruited in our study. Seizures began with facial numbness and convulsive tic, then involving right arm tic, and followed by loss of consciousness. One case was associated with tongue bite. They recovered after a few minutes without administration of any antiepileptic drugs (AEDs). Physical and neurological examination and brain MRI scan were normal. Prolonged video EEG (VEEG) recordings during wakefulness and sleep showed normal in the older sister, and spike-slow waves in the younger sister. After brushing their teeth less vigorously in shorter time, no seizure occurred. The patients had no further seizures during follow-up.  <strong>Conclusions</strong> Toothbrushing-induced epilepsy is a rare kind of reflex epilepsy, and has tightly time-locked relationship with toothbrushing. Mild cases can be released by avoiding prolonged and vigorous brushing.</p><p> </p><p>doi: 10.3969/j.issn.1672-6731.2014.12.012</p>