SummaryThe authors report a case of pituitary apoplexy whose sole clinical manifestation was painful ophtalmoplegia. No other neurological, endocrine or visual symptoms were present. The classic investigations (radiological findings as computed tomography, endocrine and visual investigations, biology) were normal. The reasons of this are the small size of the pituitary adenoma, the absence of upward enlargment or secretion. This case illustrates the carefulness necessary before concluding to the Tolosa-Hunt syndrome and the ophthalmoplegia migraine, which are diagnoses of exclusion of painful ophthalmoplegia. Many other lesions can simulate their clinical manifestations.