To report a case of congenital lacrimal outflow dysgenesis with supernumerary lacrimal puncta.

A 45-years-old woman presented with chronic bilateral epiphora with no specific medical history. Slit-lamp examination revealed bilateral upper and lower double puncta. The accessory puncta were situated along the lid margin, medial to the normal one and had a typical slit configuration. Additionally, there was a lower canalicular system block that appeared unresponsive to simple probing. Bilateral endoscopic conjunctivodacryocystorhinostomy with Jones tube was performed. Chronic bilateral epiphora was relieved after bilateral endoscopic conjunctivodacryocystorhinostomy with Jones tube.

A case of congenital lacrimal outflow dysgenesis with supernumerary lacrimal puncta was observed, which has not been previously reported in Korea. Surgical repair of the congenital lacrimal outflow dysgenesis with supernumerary lacrimal puncta should be considered to achieve a functional recovery of the lacrimal drainage system.