The purpose of this report is to present a case of multiple peripheral kerati-tis associated with chronic dacryocystitis and to discuss its management. A 58-year-old woman with redness, pain, and decrease of vision in her right eye that remained refractory to medical therapy with antibiotics and cycloplegics was referred to our clinic. The patient had history of chronic dacryocystitis for 2 years. Ocular examination revealed conjunctival injection, diffuse corneal ede-ma and multiple peripheral corneal infiltrates two of which were associated with corneal thinning. Streptococcus pyogenes was isolated from the corneal and conjunctival cultures. Since there was no any clinical improvement and corneal thinning continued despite medical treatment, external dacryocystorhinostomy (DCR) and amniotic membrane transplantation (AMT) were performed at the same session. After surgery, the patient responded well to medical treatment, symptoms relieved and corneal findings improved. Streptococcus pyogenes can cause multiple peripheral keratitis with corneal thinning in the patient with chronic dacryocystitis. The surgical treatment for dacryocystitis may be neces-sary for management of the keratitis.