Brown syndrome is a rare form of strabismus that is characterised by restriction of elevation of the eye in adduction. This is a case report of an eleven year old Nigerian girl who presented with a history of squint and poor vision. She had visual acuities of 6/24 and counting fingers in her right and left eyes respectively. There was a left hypotropia in the primary position of gaze with associated marked restriction of elevation in adduction and a positive forced duction test. Refraction revealed a refractive error of +4.50 diopters in each eye. The right visual acuity improved significantly to 6/9 with the refractive correction while the left visual acuity improved marginally to 6/60. This report demonstrates the occurrence of Brown syndrome with associated severe amblyopia in Africa. Health care providers are encouraged to promptly refer all patients, especially children, who have ocular motility disorders for early specialist intervention.