Organophosphorus (OP) pesticide poisoning which is common in rural India, usually presents with acute cholinergic crisis, which is due to increased acetylcholine because of the inhibition of the enzyme acetyl-cholinesterase. Central Nervous System (CNS) manifestations are rare, and even rarer is the presentation with extrapyramidal symptoms. The authors report a case of organophosphorus poisoning presenting with torticollis 4 days after organophosphorus exposure. The patient had inhalational exposure to organophosphorus compound while spraying in the elds and developed intermediate syndrome 4 days later. But rather than having respiratory depression, the patient developed torticollis. The patient was successfully managed with Inj Pralidoxime and supportive care. We wish to bring awareness in the medical profession and the general public regarding this rare but life threatening presentation of organophosphorus poisoning.