We describe a case of severe neuroexcitatory reaction with hyperthermia after administration of propofol in a 43-year-old patient suspected of a relapse of sarcoidosis who underwent bronchoscopy. This resulted in a lengthy stay in intensive care and long-term neuropsychological impairment. A review of the literature shows that severe neuroexcitatory symptoms (seizure-like phenomena, abnormal hypertonic, and/or jerky movements) occur rarely after propofol administration and may be life-threatening. Due to the paucity of data, the treatment is mostly empirical. The diagnosis can also be delayed owing to underrecognition. We conclude that health practitioners who frequently use propofol should be aware of this specific manifestation of drug toxicity, which albeit rare can be devastating for the patient.