The odontogenic keratocyst is an enigmatic developmental cyst that deserves special attention. It has characteristic histopathological and clinical features; but, what makes this cyst special is its aggressive behavior and high recurrence rate. Odontogenic keratocyst is a benign odontogenic cyst, commonly affecting the mandible. These lesions have posed a great difficulty for the surgeons and pathologists. Few cases affecting the maxilla are also reported. Literature search suggests that odontogenic keratocysts may affect maxillary posterior and canine region. The present case reports a lesion of odontogenic keratocyst occuring in the maxillary incisor region, crossing midline which was initially misdiagnosed as infected residual cyst. After careful evaluation, it was reported as an odontogenic keratocyst. Key Words: Odontogenic keratocyst, Maxilla, Incisor teeth.