A case with acquired lacrimal fistula due to Sjögren's syndrome

Purpose: To report a case with lacrimal fistula presumably associated with Sjögren's syndrome. Observations: A patient with Sjögren's syndrome was referred to our hospital for fistula from the inferior lacrimal canaliculus to the palpebral conjunctiva of her left lower eyelid. She also revealed severe dry eye in both eyes. She had no history of trauma or congenital lacrimal fistula. She was administering 0.3% purified sodium hyaluronate and 3% diquafosol sodium solution 6 times a day for dry eye. The Schirmer-1 test indicated that tear secretion was 0 mm for 5 min for both eyes. She was diagnosed with Sjögren's syndrome based on increased levels of blood Sjögren's-syndrome-related antigen A and Sjögren's-syndrome-related antigen B antibodies, decreased saliva volume, and lip biopsy. We performed silicone tube intubation and patched the fistula with conjunctiva. We observed the lacrimal sac and nasolacrimal duct under lacrimal micro-endoscopy; there was no bacterial concretion, obstruction, and inflammation of lacrimal mucosa. After the operation, her symptoms improved and lacrimal perforation healed after the removal of the silicone tube. Conclusions and importance: Sjögren's syndrome can cause not only corneal perforation but also mucosal perforation, which may lead to a lacrimal fistula. Sjögren's syndrome patients with severe dry eye should be managed carefully. Keywords: Lacrimal fistula, Sjögren's syndrome, dry eye