Background: Patients with gamma-hydroxybutyric acid withdrawal symptoms are at high risk of developing organic delirium, which can be fatal. The recommended first-line treatment is benzodiazepines, but treatment-resistant cases are frequent. Here we describe a case of successful bilateral electroconvulsive therapy in a patient with severe and highly agitated acute organic delirium induced by gamma-hydroxybutyric acid withdrawal and complicated by polydrug use resistant to first-line treatment. To our knowledge, this is the first report on the effect of electroconvulsive therapy on treatment-resistant delirium caused by gamma-hydroxybutyric acid withdrawal. Case presentation: A 21-year-old Danish man diagnosed with untreated attention deficit hyperactivity disorder developed severely agitated acute organic delirium caused by gamma-hydroxybutyric acid withdrawal in a Danish psychiatric ward. The patient was subjected to physical restraints and transferred to the intensive care unit for treatment. During the next 10 days, the patient showed no clinical improvement despite first-line, high-dose benzodiazepines along with intense supportive treatment with propofol, phenobarbital, and antipsychotics. On day 11, bilateral frontotemporal electroconvulsive therapy treatment was initiated and full clinical recovery was obtained after four sessions. Discussion: The full clinical remission after four electroconvulsive therapy sessions, strongly supports that electroconvulsive therapy may be an effective treatment when severe delirium induced by gamma-hydroxybutyric acid withdrawal is resistant to conventional first-line treatment with benzodiazepines. Moreover, this case illustrates that clinically effective seizures were achieved despite intensive concurrent exposure to anticonvulsive drugs. Therefore, this case report encourages consideration of electroconvulsive therapy in patients with gamma-hydroxybutyric acid delirium who are resistant to psychopharmacological treatment.