Squamous cell carcinomas arising from keratocystic odontogentic tumors are a rare phenomenon in head and neck cancer, accounting for only 1.45% of oral squamous cell carcinomas, and are classified as primary intraosseous carninomas. This cancer is locally aggressive, has a high potential for recurrence, and requires radical resection and subsequent rehabilitation. This case report describes a patient diagnosed with this uncommon tumor who was treated by dental specialists at Mayo Clinic. During convalescence, orthodontic changes to the maxillary dental archform were observed secondary to alterations in the soft tissue equilibrium following mandibulectomy and reconstruction with a microvascular fibula free flap. This highlights the need for treatment plan adaptability, the role of orthodontists in maintenance or treatment of pre-resection archforms, and the interdisciplinary nature necessary in managing the complex oncologic, functional, and esthetic needs in patients undergoing treatment for head and neck cancer. Schlagwörter: head and neck cancer, interdisciplinary oral rehabilitation, orthodontic archform management