Leishmaniasis is a group of diseases with varied clinical manifestations. Ocular involvement is an unusual presentation of leishmaniasis, and the eyelid is not a common site of cutaneous lesions, likely due to the mobility of the lids. Some case reports of conjunctival involvement are either a contiguous dissemination from lid margin or in the setting of disseminated leishmaniasis in an immunocompromised host. To our knowledge, isolated involvement of the bulbar conjunctiva has not been reported. We present the first case in the literature of a patient with an erythematous fibrovascular lesion in the interpalpebral zone that was clinically diagnosed as pterygium, but recurred at the site of surgical excision. After histopathologic diagnosis, the lesion was treated with intralesional injection of amphotericin B and improved completely within a few weeks. An accurate diagnosis of leishmaniasis in the eye may be challenging in many clinical settings. To our knowledge, an isolated pterygium.like lesion has not been reported in literature. In addition, intralesional injection of amphotericin B is a novel treatment method in this setting.