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Sildenafil is a phosphodiesterase 5 inhibitor that is widely used for the treatment of pulmonary hypertension in children. It is generally well tolerated and its safety data is largely derived from small uncontrolled trials, published reviews and case reports.

We report a case of twenty months old boy with Trisomy 21, who was admitted to our paediatric intensive care unit following a ‘stiffening episode’ at home followed by sudden onset of breathing difficulty. He had had been taking sildenafil for pulmonary hypertemsion secondary to brochiolitis obliterans. During admission he required artificial mechanical ventilation for poor respiratory effort. CT scan of the brain showed extensive bilateral infarction in the middle cerebral artery territory with cerebral oedema and subuncal herniation. He was confirmed brainstem dead after 24 hours of admission. In view of the poor clinical condition and severe brain injury, intensive care was withdrawn after consulting family.

On literature search we found only one case report of a 52-year-old man who developed bilateral middle cerebral artery infarction soon after taking sildenafil and this entity has not been described in a child before to our knowledge.

Conclusion: Cerebral ischaemic event should be considered in a child on sildenafil who presents with altered neurological signs or symptoms. To our knowledge there is no case report of bilateral middle cerebral artery stroke associated with sildenafil in the paediatric population.