Purpose: To report a case of conjunctivitis due to Mycobacterium abscessus in the setting of keratoconjunctivitis sicca due to Sjögren's syndrome in the absence of other known risk factors such as surgery, trauma or immunosuppressive therapy.

Observations: A 61-year-old woman with a history of keratoconjunctivitis sicca secondary to Sjögren's syndrome presented with dryness, irritation, redness, and discharge in her left eye for 2 months. She was diagnosed with chronic conjunctivitis and began a regimen of moxifloxacin and an ocular ointment of dexamethasone, neomycin, and polymyxin with no improvement of symptoms. Concurrent cultures grew Mycobacterium abscessus and the patient began treatment with amikacin drops, oral clarithromycin and intravenous imipenem, followed by amikacin drops, oral clarithromycin, and oral clofazimine, but her course was complicated by a perforated corneal ulcer that required a corneal patch graft. The patient eventually recovered despite persistent colonization.

Conclusions/importance: We present a case of Mycobacterium abscessus conjunctivitis in a patient with keratoconjunctivitis sicca secondary to Sjögren's syndrome without previous history of surgery, trauma, or other known risk factors. Because of low suspicion and clinician awareness, ocular nontuberculous mycobacteria (NTM) infection may have a delayed diagnosis and treatment. Clinicians should consider NTM in the differential diagnosis in patients with autoimmune disease such as Sjögren's syndrome. Treatment may be lengthy, requiring topical and systemic antibiotics and is often complicated due to resistance.