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Journal of Clinical Oncology, Vol 22, No 11 (June 1), 2004: pp. 2242-2243 © 2004 American Society of Clinical Oncology. DOI: 10.1200/JCO.2004.08.019
Paraneoplastic Syndromes in CancerCASE 2. Leucocytosis Associated With Liposarcoma Recurrence: Original Presentation of Liposarcoma RecurrenceHôpital Avicenne, Bobigny; and Institut Curie, Paris, France A 50-year-old woman was admitted to our hospital because of acute abdominal pain and marked leucocytosis. A physical examination revealed no spasm or organomegaly. She had pain in the left side. Past medical history included treatment of a retroperitoneum liposarcoma by chemotherapy (cyclophosphamide, vincristine, doxorubicin, and dacarbazine) in 1997 and resection of residual disease. In September 2001, local disease relapse occurred. Treatment was administered successfully with chemotherapy (mesna, adriamycin, ifosphamide, and dacarbazine). The patient was evaluated on computed tomography (CT) scan after four cycles and a partial response was noted (Fig 1). The patient then had resection of disease. Pathology showed a high-grade dedifferentiated sarcoma (pleomorphic sarcoma) without lipoblasts but compatible with relapse of the known liposarcoma (Fig 2).
Initially and during the first relapse, no major changes in WBC were noted. One month later, however, she had abdominal pain that became intense within a few days. Laboratory findings on admission showed 100,000 leukocytes. Eighteen days earlier, a remarkable increase in leukocytes had been observed without fever, eventually ruling out infection. Abdominal CT scan showed bulky tumor masses that measured more than 5 or 10 cm each. CT scan showed metastatic disease and a pleural effusion (Fig 3). Bone marrow aspiration showed no excessive blasts or metastatic infiltration. A cytologic study of bone marrow aspirate and chromosomal analysis showed myeloid hyperplasia consisting of granulocytes in various stages of maturation.
Liposarcoma often develops in the retroperitoneum. Dedifferentiated liposarcomas are frequently localized in the abdominal cavity with a peak of frequency around 60 years. Retroperitoneal soft tissue sarcomas are rare tumors estimated to account for 15% of all patients with soft tissue sarcomas seen in referral populations.1 The dedifferentiated character is frequently observed during a relapse.2,3 As our patient showed a remarkable degree of leukocytosis without any obvious focus of infection, we suspected a paraneoplastic syndrome with granulocyte colony-stimulating factor or granulocyte macrophage colony-stimulating factor production. These cytokines enhance the production of granulocytes and their release from bone marrow into the peripheral blood.4 A previous case of liposarcoma and another sarcoma were published with documented leukemoid reactions.5,6 In connective tissue tumors, this syndrome appears particularly rare. Increased production of granulocyte macrophage colony-stimulating factor has also been documented in two patients with lung cancer and leukocytosis and eosinophilia.7 Initially, our patient's tumor didn't produce a leukemoid reaction, but 1 month after surgery, leucocytosis rapidly increased. Even though the lung CT scan and x-rays were normal and the tumor masses had been removed, 1 month later increased tumor growth was associated with cytokine production. It is well known that mild leukemoid reactions can be associated with surgery, but such a high increase in leukocytes is rare. The treatment options for primary retroperitoneal sarcomas include chemotherapy, radiation therapy, surgery, or a combination of these modalities.2,8 The surgical principles important for local tumor control are complete en bloc excision of the tumor, and pathology confirmation of tumor-free margins. However, in this particular case, with relapse, prognosis seemed poor with no possibility of additional surgery and radiotherapy.3, 8,9 Chemotherapy was therefore started. A decrease in both the tumor and WBC occurred with palliation of symptoms. Authors' Disclosures of Potential Conflicts of Interest The authors indicated no potential conflicts of interest.
REFERENCES 1. Pisters PW, O'Sullivan B: Retroperitoneal sarcomas: Combined modality treatment approaches. Curr Opin Oncol14:400–405, 2002[CrossRef][Medline] 2. Eilber FC, Eilber KS, Eilber FR: Retroperitoneal sarcomas. Curr Treat Options Oncol1:274–278, 2000[Medline] 3. Lewis JJ, Leung D, Woodruff JM, et al: Retroperitoneal soft-tissue sarcoma: Analysis of 500 patients treated and followed at a single institution. Ann Surg228:355–365, 1998[CrossRef][Medline]
4. Melhem MF, Meisler AI, Saito R, et al: Cytokines in inflammatory malignant fibrous histiocytoma presenting with leukemoid reaction. Blood82:2038–2044, 1993 5. Nasser SM, Choudry UH, Nielsen GP, et al: A leukemoid reaction in a patient with a dedifferentiated liposarcoma. Surgery129:765–767, 2001[CrossRef][Medline] 6. Hisaoka M, Tsuji S, Hashimoto H, et al: Dedifferentiated liposarcoma with an inflammatory malignant fibrous histiocytoma-like component presenting a leukemoid reaction. Pathol Int47:642–646, 1997[Medline] 7. Sawyers CL, Golde DW, Quan S, et al: Production of granulocyte-macrophage colony-stimulating factor in two patients with lung cancer, leukocytosis, and eosinophilia. Cancer69:1342, 1992[CrossRef][Medline]
8. Linehan DC, Lewis JJ, Leung D, et al: Influence of biologic factors and anatomic site in completely resected liposarcoma. J Clin Oncol18:1637–1643, 2000 9. Kinsella TJ, Sindelar WF, Lack E, et al: Preliminary results of a randomized study of adjuvant radiation therapy in resectable adult retroperitoneal soft tissue sarcomas. J Clin Oncol6:18–25, 1988[Abstract]
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Copyright © 2004 by the American Society of Clinical Oncology, Online ISSN: 1527-7755. Print ISSN: 0732-183X
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