Wilson's Disease (WD) is a rare autosomal recessive hereditary disease with copper accumulation in the body, particularly the liver, brain and cornea. WD is widely treated with chelation agents who enable the copper excretion. Since high concentrations of copper are toxic, WD is associated with hepatocellular carcinoma and cholangiocarcinoma, with low incidence of other types of cancer. We present a case of a 33 year old man who was treated in the Oral and Maxillofacial Surgery Department of the Aristotle University of Thessaloniki with multiple dysplasia lesions and an in situ carcinoma of the tongue, which is to our knowledge the first case report of oral lesions to a patient with WD. Literature is reviewed on copper levels on patients with head and neck cancer, and on chelation agents and their effect on cancer cells.