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International Journal of Case Reports and Images (IJCRI) is an international, peer reviewed, monthly, open access, online journal, publishing high-quality, articles in all areas of basic medical sciences and clinical specialties.Aim of IJCRI is to encourage the publication of new information by providing a platform for reporting of unique, unusual and rare cases which enhance understanding of disease process, its diagnosis, management and clinico-pathologic correlations. We present the clinical case of a 72-year-old Caucasian male with ophthalmological history of right eye amblyopia caused by an untreated esotropia, who went to the emergency room presenting holocranial headache, ptosis and temporary diplopia. The pupils were symmetric with a present but slowed down right direct reflex, and a doubtful Marcus Gunn pupil. The patient was orthotropic. Ocular motility examination showed a limitation in adduction, elevation and depression of the right eye. The patient had a computed tomography (CT) of the brain and orbits, a analytical study, a CT angiography, a brain magnetic resonance, a lumbar puncture, a CT scan of the neck, chest, abdomen and pelvis, a flexible cystoscopy, and a transurethral resection. Once made, the histology of the biopsies of TUR revealed fragments of a infiltrative urothelial carcinoma of high grade. IJCRI publishes Conclusion:The oculomotor nerve palsy may be the first manifestation of a serious systemic disease and appear in an atypical form in a patient with a previous esotropia. Careful assessment and investigation should be taken.

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