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Journal of Clinical Oncology, Vol 23, No 3 (January 20), 2005: pp. 641-643
© 2005 American Society of Clinical Oncology.
DOI: 10.1200/JCO.2005.03.090

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DIAGNOSIS IN ONCOLOGY

Benign Lesions in Cancer Patients

CASE 2. Unusual Association of Osteosarcoma and Sarcoidosis

Kyoji Okada, Masahiro Sasaki, Hiroshi Nanjo, Manabu Ito, Shu Takahashi, Eiji Itoi

Akita University School of Medicine, Akita, Japan

A 29-year-old Japanese woman first noticed pain around her left knee in May 1997. The pain gradually increased, and the patient visited our outpatient clinic in June 1998. Radiographs showed an osteolytic lesion in the distal femur with irregularly distributed mineralization (Fig 1A). In the lateral view, periosteal reaction of the posterior part of the distal femur and a partially mineralized soft tissue mass were observed (Fig 1B). A biopsy specimen contained proliferations of cartilaginous tissue and spindle cells with marked nuclear atypia. Scattered osteoid formation was also observed. A diagnosis of osteosarcoma of the left distal femur was made. The patient began a 12-week course of preoperative chemotherapy including high-dose methotrexate (10 g/m2) with leucovorin rescue, doxorubicin hydrochloride (30 mg/m2 x 2 days), and cisplatinum (100 mg/m2). She underwent a wide excision with replacement by a metal prosthesis in December 1998. The histologic evaluation of chemotherapy effect was poor. She received postoperative chemotherapy including high-dose methotrexate with leucovorin rescue, doxorubicin hydrochloride, cisplatinum, and ifosfamide (2 g/m2 x 4 days) for 6 months. In June 2003, chest radiographs and computed tomography scans (Fig 2) showed enlarged bilateral hilar and paratracheal lymph nodes. Her serum angiotensin-converting enzyme was within the normal range (11.5 U/L; normal range, 8.3 to 21.4 U/L). Gallium scintigram showed an increased uptake in bilateral hilar regions. Clinical and radiological patterns suggested the possibility of metastatic osteosarcoma, though most metastases from osteosarcoma are hematogenous. A biopsy specimen taken by thoracoscopy showed diffuse noncaseating granulomas (Figs 3 and 4). Sarcoidosis associated with osteosarcoma was diagnosed, but no other lesion was detected in a further systemic investigation. The chest computed tomography scan has remained unchanged. The patient is healthy 5 years 3 months after surgery, without any evidence of local recurrence or distant metastasis.



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Fig 4.
 
It is well known that sarcoidosis or sarcoid reaction often occurs in association with several kinds of epithelial malignancies.1-3 However, this condition associated with sarcoma is quite rare.1,4-6 To our knowledge to date, only one case of osteosarcoma associated with sarcoidosis has been reported in the literature.7 In both this previously reported case and the current case, the patients developed sarcoidosis after the completion of systemic chemotherapy for osteosarcoma.7 The relationship between chemotherapy and sarcoidosis should be further investigated.

Authors’ Disclosures of Potential Conflicts of Interest

The authors indicated no potential conflicts of interest.

REFERENCES

1. Brinker H: Sarcoid reactions in malignant tumours. Cancer Treat Rev 13:147-156, 1986[CrossRef][Medline]

2. Reich JM, Mullooly JP, Johnson RE. Linkage analysis of malignancy-associated sarcoidosis. Chest 107:605-613, 1995[Abstract/Free Full Text]

3. Haluska P, Luetmer PH, Inwards CY, et al: Diagnostic dilemma: Sarcoidosis simulating metastatic malignancy. J Clin Oncol 21:4653-4654, 2003[Free Full Text]

4. Sumiyoshi A, Sannoe Y, Tanaka K: Rhabdomyosarcoma of the esophagus: A case report with sarcoid-like lesions in its draining lymphnodes and the spleen. Acta Pathol Jpn 22:581-589, 1972[Medline]

5. Nakamura M, Iemura A, Kojiro M, et al: Leiomyosarcoma of the rectum with sarcoid reaction: A case report. Kurume Med J 37:171-175, 1990[Medline]

6. Matsuhashi N, Nakajima A, Fukushima Y, et al: Sarcoid reactions in association with a leiomyosarcoma of the stomach. Am J Gastroenterol 91:2623-2624, 1996[Medline]

7. Sybert A, Butler TP: Sarcoidosis following adjuvant high-dose methotrexate therapy for osteosarcoma. Arch Intern Med 138:488-489, 1978[Abstract/Free Full Text]


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Copyright © 2005 by the American Society of Clinical Oncology, Online ISSN: 1527-7755. Print ISSN: 0732-183X
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