We present 2 patients with bipolar disorder and persistent sexual arousal syndrome (PSAS). Because they were refractory to several trials of antidepressants and mood stabilizers, electroconvulsive therapy (ECT) was prescribed. They underwent a course of ECT with remission of both their depressive and their PSAS symptoms. They have remained on maintenance ECT with good results. Although the etiology of PSAS is unknown and its relationship to bipolar disorder is unclear, the successful resolution of PSAS with ECT is of clinical and theoretical interest.