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Burning mouth syndrome (BMS) is an orofacial pain disorder characterized by a chronic, idiopathic burning sensation of the oral mucosa that mostly affects middle-aged women. Although both psychological and neuropathological factors have been postulated to underlie BMS, the pathogenic mechanism of the condition remains controversial, as do the treatment strategies.A single case was reported.Ms A, a 66-year-old woman with BMS type 1, which is characterized by daily burning pain associated with circadian variation, underwent electroconvulsive therapy (ECT). After the completion of 12 ECTs, the pain markedly diminished and the pronounced ECT effect persisted over the subsequent 24-week period of observation.To our knowledge, this is the first clinical report on the efficacy of ECT for treating pain associated with BMS. ECT can be considered to be an option for treating individuals with enduring and intractable intraoral burning pain.