Hamartomas are benign neoplastic malformations characterized by disorganized growths of tissue in their native location. They may grow in numerous sites throughout the body. We report the case of a 3-year-old boy with unilateral compressive optic neuropathy caused by a brain hamartoma that mimicked strabismic amblyopia. To our knowledge, this is the first case report in the literature of prechiasmal optic nerve compression from a brain hamartoma without ipsilateral bony defect or encephalocele.
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