Isolated extramedullary relapse of acute lymphoblastic leukemia in a submandibular salivary gland

Salivary glands are uncommonly involved in leukemia. Moreover, isolated leukemic relapse in salivary glands is an extremely rare event. We report a young adult male with acute lymphoblastic leukemia who presented with an isolated extramedullary relapse of his leukemia in a left sided submandibular salivary gland just before his planned allogeneic bone marrow transplant. The patient underwent a wide surgical excision of the involved salivary gland then he received a conditioning protocol composed of total body irradiation and cyclophosphamide followed by a successful allograft.