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Background In children, migraine with or without aura is a common entity, however variants like recurrent painful optic neuropathy (RPON) is rarely encountered. Case result A 9 year old boy presented with headache for 1 week and restricted movements and drooping in both eyes for last 3 days. On examination he had bilateral ophthalmoplegia and ptosis. History of migrainous headache was present in the patient as well as his mother. His MRI brain with venogram, serum autoimmune markers, serum and urine toxicology screen and repetitive nerve stimulation test were normal. He received intravenous pulse followed by oral steroids for 6 weeks and was started on antimigraine prophylaxis. Eighteen months since the attack, he has improved completely with mild asymmetric mydriasis persisting. Discussion and conclusion This may represent first attack of RPON in a child with migraine. Rarely this may herald the onset of migraine as well, index of suspicion should be high as it is a diagnosis of exclusion and a treatable entity. [ABSTRACT FROM AUTHOR]