A 32-year-old man with a history of heroin dependence was discovered unconscious in his home after heroin injection; brought to the hospital, he regained consciousness, but he developed a confusional state, which turned to stupor, and weakness of the upper and lower limbs on the right side. The neurological examination revealed an altered conscious level with difficult awakening. The patient maintained a decorticate posture at rest. Cranial nerves were intact. There was a right hemiparesis. Plantar responses were extensor on the right side and downgoing on the left.Brain MRI (Fig. 1a, b, c) revealed diffuse, symmetric areas of hyperintensity in the periventricular white matter and in the semiovale centers; the DWI sequence showed a restricted diffusion compatible with cytotoxic edema and axonal damage. These findings were suggestive of a toxic spongiform leukoencephalopathy [1].The patient had no exposure to other white matter toxins. Serology was negative for HBV, HCV, and HIV. CSF was unremarkable (glucose, proteins, cell counts) and negative for oligoclonal bands and JC virus.Stupor resolved in few weeks and motor signs improved, so the patient was discharged and he started a neurological and psychiatric rehabilitation program.