A 2-year-old boy with Alagille syndrome who had esotropia underwent bilateral medial rectus recession. One month postoperatively, esotropia reoccurred and was aggravated thereafter. Right lateral rectus resection was unsuccessful because the insertion site could not be identified. Careful preoperative imaging may be necessary in patients with Alagille syndrome to detect any abnormal extraocular muscle insertion. [J Pediatr Ophthalmol. 2018;55:e36-e38.].
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