Secondary osteosarcoma arising after treatment for childhood hematologic malignancies

Abstract

Secondary osteosarcoma arising after the treatment of hematologic malignancies other than Hodgkin's lymphoma is rare. We report two cases of secondary osteosarcoma arising after treatment for childhood hematologic malignancies (non-Hodgkin's lymphoma and lymphoblastic leukemia). A 10-year-old boy, at the age of 3, was diagnosed with non-Hodgkin's lymphoma. He received chemotherapy, radiation, and bone-marrow transplantation and then was in complete remission. At 6 years, he complained of increasing pain of the right thigh and was diagnosed with osteoblastic osteosarcoma. A 26-year-old man, at the age of 6, was diagnosed as having acute lymphoblastic leukemia (ALL). He received chemotherapy, radiation, and peripheral blood stem cell transplantation (PBSCT). At 11 years after PBSCT, he visited with the complaint of left lumbar swelling. He was diagnosed with chondroblastic osteosarcoma. In both cases alkaline phosphatase (ALP) had already increased prior to the onset of the symptom. We should rule out secondary osteosarcoma at the abnormal elevation of ALP during clinical follow-up of patients after treatment of childhood hematologic malignancies.

Figure 1.

Case1. Plain radiograms of the proximal femur revealed a mixed sclerotic and lytic intramedullary lesion with the periosteal perpendicular spiculation with extensive periosteal reaction and “Codman's triangle” reaction.

Figure 2.

Case1. (A) MRI revealed an isointense lesion within the quadriceps femoris muscle on T1-weighed images. (B) T2-weighed images showed a heterogenous hyperintense lesion. (C) The lesion was enhanced heterogeneously on fat-suppressed T1-weighed images after gadolinium injection.

Figure 3.

Case1. Histological appearances of the biopsied specimen demonstrated spindle or polygonal cells with atypical nuclei, eosinophilic amorphous osteoid and matured bone tissue.

Figure 4.

Case 2. Plain CT showed a lytic lesion in the posterior superior iliac spine (arrow).

Figure 5.

Case 2. (A) MRI showed a low-intense lesion around the left sacroiliac joint on T1-weighted images. (B) T2-weighed images confirmed a heterogenous hyperintense lesion. (C) The lesion was enhanced on fat-suppressed T1-weighed images.

Figure 6.

Case 2. Needle biopsy of the left iliac spine showed polygonal cells with hyperchromatic nuclei. The stroma consisted of chondroid and osteoid tissues.