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ary 28, 2022 (retrospectively registered). UMIN000046769 (UMIN-CTR); first registered on January 28, 2022 (retrospectively registered). PITX3 has been reported to be associated with congenital cataracts, anterior segment mesenchymal dysgenesis, Peters' anomaly, and microphthalmia. In this case, an infant with unilateral buphthalmos, corneal staphyloma and corneal fistula carrying a variant in PITX3 was reported. We describe a 4-month-old female infant who was referred to our Eye Clinic because of gradual enlargement of the eyeball in the