Absent and anomalous superior oblique and superior rectus muscles

Anomalous or absent superior oblique or superior rectus muscles have usually been reported in the context of craniofacial disorders. We report a case of absent superior oblique and superior rectus muscles in the right eye and anomalous superior oblique and superior rectus muscles in the left eye in an otherwise healthy 4-year-old boy. The possibility of anomalous or absent extraocular muscles should be considered in unusual and complex presentations of strabismus, and alternative surgical strategies should be considered preoperatively. The clinical triad of levator palpebrae superioris, superior rectus and superior oblique dysfunction should suggest embryologic abnormalities of these muscles.