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Purpose: To test nicotine patch treatment for a patient with a defined mutation for autosomal dominant nocturnal frontal lobe epilepsy (ADNFLE) whose seizures were refractory to standard antiepileptic therapy., Methods: Open and double-blind trials of nicotine patches in an "n-of-one" study. The double-blind trial comprised periods during which either placebo or nicotine patches were each used for three periods of 2 weeks, randomized in a double-blind manner., Results: In an open study, nicotine patches reduced seizures from 1.65 +/- 2.36 to 0.01 +/- 0.0 seizures per day (p < 0.0001). In a double-blinded placebo-controlled phase, the average frequency of seizures on nicotine versus placebo was 0 +/- 0 versus 0.56 +/- 1.14 seizures per day (p < 0.0001)., Conclusions: Nicotine patches may be of benefit to some individuals with ADNFLE.