Bilateral marginal keratitis associated with engraftment syndrome after hematopoietic stem cell transplantation

PURPOSE: To report the first case of bilateral marginal keratitis in the setting of engraftment syndrome in a patient who had undergone hematopoietic stem cell transplantation. METHODS: A 63-year-old man with a history of myelodysplastic syndrome presented with a 5-day history of red eyes. Two weeks before presentation, the patient had received a matched unrelated donor peripheral blood stem cell transplant and subsequently developed engraftment syndrome with a rapid white blood cell count recovery, noninfectious fever, skin rash, and shortness of breath. Ocular symptoms coincided with the timing of the white blood cell recovery. On examination, vision was 20/20 OU with +1 conjunctival injection with bilateral corneal subepithelial infiltrates in the periphery, particularly in areas of corneal pannus from previously existing contact lens-related neovascularization. There was no evidence of blepharitis, meibomian gland dysfunction, or uveitis. Ocular bacterial and viral cultures were negative, and a conjunctival biopsy was negative for viral inclusions or ocular graft-versus-host disease. RESULTS: The patient was initially treated with topical antibiotics for 2 days without any improvement and treated with topical corticosteroids 4 times daily. Examination 7 days after starting topical corticosteroids showed complete resolution of the marginal keratitis. CONCLUSIONS: Engraftment syndrome is notable for a rapid recovery of the white blood cell count after hematopoietic stem cell transplantation. Patients who present with presumed conjunctivitis in the setting of autologous and allogeneic stem cell transplantation should be evaluated for engraftment syndrome-related marginal keratitis.