PIP: Chloroquine is considered essentially nontoxic when used for the chemosuppression of malaria, but gastrointestinal upsets, headache, blurring of vision, pruritus, and uritcaria may occur during chloroquine therapy. Recently, Bhargava et al. and Eronini and Eronini have reported the extrapyramidal syndrome (EPS) following chloroquine therapy in adults. The clinical manifestations included upward rolling of the eyeballs, retraction of neck and back, trismus with marked difficulty in speech, and coarse tremors. Observations of 4 instances of EPS in children following chloroquine therapy for malaria are reported. A 2-1/2 year old girl was admitted to the All India Institute of Medical Sciences Hospital with a 4 day history of intermittent high grade pyrexia with chills and rigors. Following treatment with oral chloroquine in the recommended therapeutic dosage, the fever responded, but the child became drowsy and developed paroxysms of involuntary movements of the tongue, torticollis, torsion dystonia of the limbs, and parosysms of tonic muscular spasms. She completely recovered spontaneously within 48 hours. The 2nd case was that of a 12-year old female brought to the hospital with a 15-day history of intermittent high grade fever with chills and rigors. The patient was started on chloroquine sulfate in the recommended therapeutic dose. After an interval of 4 days she developed coarse tremors of the hands, upward rolling of the eyeballs, episodic deviation of the angle of the mouth towards the left, and trismus. These symptoms disappeared spontaneously within 8 hours. A 6-year old girl, the 3rd case, developed episodes of opisthotonous, upward rolling of the eyeballs, protrusions of the tongue, intermittent writhing movements of the upper limbs, and drowsiness following the ingestion of 6 tablets of chloroquine sulfate for suspected diagnosis of malaria. She spontaneously recovered from EPS over a period of about 48 hours. The 4th case, a 7-year old boy, gave a history of high grade fever with chills and rigors of 1 day's duration. He developed drowsiness, tonic spasms of the neck, upward rolling of the eyeballs, and writhing contortions of all limbs about 2 hours following intravenous administration of 100 mg of chloroquine. 8 hours later an additional 100 mg chloroquine was given intravenously for the mistaken diagnosis of cerebral malaria. On examination the child was drowsy, had generalized stiffness, torticollis, and trismus. He recovered gradually over a 48-hour period without any specific therapy. The exact mechanism of production of EPS remains uncertain.