To assess the presence of rare GPATCH3 variants in congenital glaucoma patients, we screened this gene by Sanger sequencing in a cohort of 130 unrelated congenital glaucoma cases with no known gene mutations and in a second cohort composed of 40 secondary congenital glaucoma cases associated with ocular malformations such as microphthalmia or anterior segment dysgenesis. This evidence concerns the gene GPATCH3 and anterior segment dysgenesis.