Paroxysmal kinesigenic dyskinesia (PKD) is an uncommon neurological disorder, consisting of brief attacks of involuntary movements triggered by sudden action. Patients with PKD generally respond positively to antiepileptic drugs. We compared the efficacy and tolerability of oxcarbazepine (OXC) and carbamazepine (CBZ) in the treatment of PKD, in order to find the optimal prescription. This retrospective study reviewed monotherapy use of CBZ or OXC in 28 patients with PKD during 2005-2011, dividing into two groups. The frequency and severity of attacks and adverse events were recorded. Ten patients in the OXC group and 12 in the CBZ group continued the therapy for more than 12 months. The 12-month retention rate was 76.92% and 80.00%. Both groups showed a marked reduction in attack frequency and the degree of reduction did not differ significantly between the groups. Side effects in patients with OXC included headache, diplopia, and elevated hepatic enzymes, while diplopia, nausea, and leukopenia were recorded in CBZ group. Another three cases were found with better tolerance when converted to OXC from CBZ for rash, drowsiness, diplopia, and nervousness. In conclusion, OXC and CBZ are similarly effective and tolerated in the treatment of PKD, however, more evidence from larger and blind prospective trials are needed.