Odontogenic keratocysts are odontogenic cysts which have locally infiltrative behavior. They mostly occur in second and fourth decades but can occur at any age throughout life. In majority of cases, they are located in the posterior region of mandible, but can also be found in the maxilla especially in the canine region. In this article, we have discussed the diagnosis and management of a large odontogenic keratocyst cyst involving the maxillary sinus. A 22 years old male patient presented with pain and swelling on right side of the face since last one year. There was history of pus discharge and gradual increase in size of the swelling since last 3-4 months. Patient took antibiotics but there was no improvement. Odontogenic keratocyst is a developmental pathology which arises from additional remnants of dental lamina of oral epithelium. They present with swelling, pain and pus formation/discharge as signs of infection. Often, when the lesion is large there can be paresthesia of the lower lip. These lesions can also be clinically asymptomatic. Mostly they appear as well-demarcated unilocular radiolucency having a thin, sclerotic well-defined margin. There are many treatment options like enucleation, marsupialization, decompression, curettage, use of chemical agents like Carnoy's solution, cryotherapy with liquid nitrogen or peripheral osteotomy and surgical resection of lesion.