The prevalence of gamma-butyrolactone/gamma-hydroxybutyric acid (GBL/GHB) use is increasing. The gravity and number of incidents with this drug are relatively high. A feared complication is addiction and its withdrawal syndrome, which can be life-threatening and is difficult to treat. We present the case of a 31-year-old man, admitted to the ICU because of accidental GBL withdrawal. The patient was tachycardic, sweaty, extremely agitated, and showed signs of psychosis. High doses of benzodiazepines, propofol, sufentanil, and quetiapine could not sedate the patient sufficiently. Dosing with pharmaceutical GHB was challenging due to severe gastric retention. As the patient developed hyperthermia and rhabdomyolysis, signs of a neuroleptic malignant syndrome (NMS), he was treated with dantrolene. After 14 days, the patient was discharged to a psychiatric clinic for further treatment. GHB affects multiple neurotransmitters and chronic use causes the up- or down-regulation of several receptors. During GHB withdrawal, the patient developed a hyperexcitable state, in which there was insufficient gamma-aminobutyric acid (GABA) (the most important inhibiting neurotransmitter) and an abundance of glutamate (the most important excitatory neurotransmitter). High-dose benzodiazepines are often advocated as the first-line treatment, but benzodiazepine resistance has frequently been reported. Therefore, treatment with pharmaceutical GHB is advised. Patients with GHB-withdrawal who have clinical signs of NMS can be treated with dantrolene because it regulates the distorted calcium secretion and affects the serotonin and cholinergic system.