Celiac sprue, also called nontropical sprue, is a malabsorption syndrome with symptoms that are triggered mainly by gluten ingestion. Non-Hodgkin's lymphoma of the gastrointestinal tract has been reported in patients with a long history of celiac sprue. This case report describes the occurrence of primary oral lymphoma in such a patient. This case is unusual because lymphomas associated with celiac sprue usually develop in the small intestine or in the stomach, and primary lymphoma in the mouth is itself uncommon.