Midface bones are an unusual site for primary presentation and relapse in acute lymphoblastic leukemia. Herein, we describe a case of acute pre B cell lymphoblastic leukemia with leukemic infiltration of maxilla and bone marrow involvement. At the time of relapse, the patient presented again with maxilla involvement and the phenotype changed to biphenotypic lymphoblastic leukemia. Our case suggests that leukemic involvement of the maxilla should be considered in differential diagnosis of patients presenting with acute painful midface swelling. These patients should be evaluated using imaging modalities, blood parameters and histopathology. Keywords: Face, bone, leukemia, relapse, acute lymphoblastic leukemia.

Keywords: Face, bone, leukemia, relapse, acute lymphoblastic leukemia.

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