Purpose: To evaluate the characteristics and outcomes of strabismus in patients with hydrocephalus. Methods: A retrospective chart review of patients with strabismus and hydrocephalus is performed in the Department of Pediatric Ophthalmology between 2012 and 2018. Results: Seventeen patients between the ages of 6 months and 13 years met the criteria of strabismus and hydrocephalus. Although all had developmental delay, five patients out of 17 were premature (lower than 36 weeks of gestation). All patients had ventriculoperitoneal shunt placement for congenital hydrocephalus. Three patients had exotropia, whereas 14 had esotropia. Glasses were prescribed to 13 patients: hyperopic correction in 12 and myopic correction in one patient. Surgical correction with bimedial recession was performed in five patients. Four of them achieved successful ocular alignment. Conclusions: Children with hydrocephalus most likely have esotropia. Although good ocular alignment is achieved with surgical correction in some patients, some patients may benefit from glasses.