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To report a rare case of filamentary keratitis associated with stiff person syndrome.Case report.A 26-year-old woman with several autoimmune conditions was referred because of filamentary keratitis. Conservative management using lubrication, mucolytics, and bandage contact lenses failed to adequately relieve symptoms. Despite the addition of oral prednisone, cyclosporine, and azathioprine, the patient's condition persisted with numerous filaments and severe dry eye. One month later, the patient had an episode of sudden muscle cramps in her back, for which she was hospitalized. Investigations revealed a diagnosis of stiff person syndrome, positive for glutamic acid decarboxylase autoantibodies. Plasmapheresis and high-dose intravenous steroids were provided as initial therapy. On follow-up, her filamentary keratitis resolved and at the last visit, her ocular symptoms had completely resolved.Filamentary keratitis is a chronic, recurrent condition that can be challenging to treat. Effective management requires careful consideration of possible predisposing causes, especially in refractory cases.