Giant cell arteritis is a systemic disease with a broad range of clinical signs and symptoms. Although the most frequently involved vessel is the superficial temporal artery, other arteries can be affected. Vasculitic changes in the facial artery usually present as jaw claudication. A report of a case of giant cell arteritis is presented in which facial artery involvement first manifested itself clinically as a submandibular mass. This is a very rare and atypical form of clinical presentation.