To report a paediatric case of retinopathy-positive cerebral malaria, emphasizing the clinical significance of long-term neurological and ophthalmological follow-up (5,5 years). After a recent journey in Ghana, a 17-month-old African female child was admitted at the Paediatric Emergency Room with fever and vomiting. Blood smear confirmed a Plasmodium Falciparum parasitaemia. Iv quinine was promptly administered, but after a few hours, the child developed generalized seizures, requiring benzodiazepine therapy and assisted ventilation for severe desaturation. Brain imaging (CT and MRI), lumbar puncture and several electroencephalograms showed data compatible with cerebral involvement of malaria. Schepens ophthalmoscopy and Ret-Cam pictures acquisition revealed macular haemorrhages in the left eye with central whitening and bilateral capillary abnormalities, typical signs of malarial retinopathy. Antimalarial therapy and iv Levetiracetam allowed neurological improvement. Eleven days after the admission, the child was discharged, showing no neurological symptoms and with an improved EEG signal, a normalized fundus oculi and brain imaging. Neurological and ophthalmological long-term follow-up were conducted: EEG controls didn't reveal abnormalities and the complete ophthalmological assessment showed a regular visual acuity and fundus oculi, as well as a normal SD-OCT and electrophysiologic testing. Cerebral malaria is a severe complication, characterized by a high fatality rate and challenging diagnosis. A helpful instrument for diagnostic and prognostic evaluation is the ophthalmological detection of malarial retinopathy and its monitoring over time. In our patient the long term visual follow-up didn't reveal any adverse outcome.