To report a case of congenital eyelid retraction with eyelid asymmetry.

12-year-old male patient visited our clinic with eyelid asymmetry, which had found 6 months after birth. On examination, his marginal reflex distance (MRD1) was 4.0 mm for right eye, and 7.5 mm for left eye. There was no significant notice from orbital computed tomography or laboratory findings and other factors that might have caused eyelid retraction were not found. As treatment, Botulinum toxin type A was injected under the upper tarsal conjunctiva. The upper eyelid retraction was relieved to MRD1 = 4.0 mm after 1 week and MRD1 = 5.5 mm after 1 month. This case was diagnosed as congenital eyelid retraction of upper eyelid.

Diagnosis of congenital eyelid retraction should be considered to patients with eyelid asymmetry, and the symptom can be relieved with Botulinum toxin type A injection.