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Water intoxication is a potentially fatal hypo-osmolar syndrome with brain function impairment. Isolated symptomatic excessive ingestion of free water is very rare in childhood. We report a case of acute hyponatremia due to water intoxication without Antidiuretic Hormone (ADH) excess in a child with sickle cell disease. The boy was admitted to our Emergency Department because of new-onset prolonged generalized seizures. Blood test showed hyponatremia, and elevated creatine kinase value; neuroimaging was negative. His recent medical history revealed that on the day before he had drunk about 4 liters of water in 2 hours to prevent sickling, because of back pain. He was treated with mild i.v. hydration with normal saline solution and showed progressive clinical improvement and normalization of laboratory test. Rhabdomyolysis is a rare complication of hyponatremia whose underlying mechanism is still unclear.