| Summary: | Cowpox is a rare zoonosis transmitted to humans mainly from cats. The disease usually causes skin lesions; however, the ocular form may lead to other serious complications.We describe a case of cowpox in a rare location of the upper eyelid of an immunocompetent male, which lead to necrosis of the upper eyelid, keratitis and leucomatous opacity, and the neovascularization of the cornea. The patient underwent several surgeries, including reconstruction surgery of the eyelids, correction of the medial canthus, and corneal neurotization with supraorbicular nerve transplantation.Suspicion of cowpox should be made in patients where there are poorly healing skin lesions accompanied by a painful black eschar with erythema and local lymphadenopathy. Ocular cowpox may lead to serious complications and possibly mimic anthrax. Diagnosis of cowpox can be confirmed by detection of cowpox virus DNA by polymerase chain reaction. Patients should be advised to protect themselves while handling sick animals.
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