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| Year : 2003 | Volume
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| Issue : 1 | Page : 75-77 |
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| Metachronous multifocal osteosarcoma |
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JP Singh, R Shrimali, L Garg, V Setia
Mohan Das Oswal Cancer Hospital, G.T. Road, Sherpur, By Pass, Ludhiana, India
Click here for correspondence address and email
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Keywords: Osteosarcoma, Metastasis
How to cite this article:
Singh J P, Shrimali R, Garg L, Setia V. Metachronous multifocal osteosarcoma. Indian J Radiol Imaging 2003;13:75-7 |
 Introduction | |  |
Metachronous multifocal osteosarcoma (MFOS) is a rare form of osteosarcoma manifested by one or more new tumors developing after the initial treatment of primary osteosarcoma. The pathogenesis of metachronous MFOS is still obscure whether it represents multiple true primaries or metastatic disease. However there is a clinical significance that metachronous MFOS is a potentially curable disease [1]. Metachronous MFOS mainly occurs in adolescents or young adults and present as asymmetric lesions in the metaphyses of long tubular bones. We present here a case of metachronous MFOS because of its unusual presentation and with review a literature.
| Case history | | |
A 40-year-old male patient presented to the orthopaedics dept. with swelling over the right knee for the last 2 months and pain in the back for the last 1 month. Plain radiograph of the right knee [Figure - 1] showed an osseous lesion with minimal periosteal reaction near the lower end of the right femur. CT cuts [Figure - 2] through the lesion showed obliteration of the medullary cavity by a dense osseous lesion with spiculations into the surrounding soft tissue. Chest radiograph [Figure - 3] showed multiple rounded shadows in both lung fields with a dense opacity in the left lower zone s/o an ossified soft tissue opacity. Sagittal MR image [Figure - 4] showed a hypointense lesion in D10 vertebrae on both T1 & T2 W sequences. Axial T2W MR image [Figure - 5] through D9 vertebra showed the spinal cord being pushed to the left by a hypointense lesion (which was a calcified soft tissue lesion on CT scan, image not shown) causing widening of the canal on the right side. The bone scan was done; it showed an abnormal increased uptake in D9, 10,11& L5 vertebrae and a large hot spot measuring approximately 1.0 X 0.8 cm occupying the left hemithoracic region s/o metastasis. A diagnosis of multicenteric osteosarcoma was made that was confirmed on biopsy of the knee lesion, as a 'highly malignant osteosarcomatous tumor'. Since all the lesions were of the same size and occurred within a period less than 5 months a diagnosis of metachronous MFOS was made
| Discussion | | |
Osteosarcoma is a highly malignant bone tumor, and it mainly involves a single location in the metaphysis of long bones [2]. MFOS is a rare condition and the incidence has been reported as 1-10% of all cases of osteosarcoma [3]. A slight male predominance exists among younger patients (<18 years old), and a much greater male predominance is present, approximately 3:1, among older patients (>18 years old). The most frequently encountered site of the primary lesion (in the decreasing order of frequency) are as follows: the distal end of femur, the proximal end of tibia, the proximal end of femur together with the pelvis and proximal end of humerus [4].
Two major theories exist in the literature regarding the multifocality of osteogenic sarcoma. These include: (a) multisite lesions arising simultaneously, presumably all representing multiple, synchronous, primary lesions; (b) single-site origin, with one dominant site and then early and rapidly progressive metastatic disease. The most commonly quoted classification of multifocal osteogenic sarcoma is that of Amstutz [4].
Type I- Patients < 18 years of age with multiple, synchronously occurring bone lesions with rapid progression within 5 months of diagnosis.
Type II- Patients > 18 years of age with multiple, synchronously occurring bone lesions arising within 5 months of presentation with a dominant mass.
Type III a- Patients of any age with metachronous metastatic bone lesions appearing 5-24 months after diagnosis.
Type III b- Patients of any age with metachronous metastatic bone lesions occurring more than 24 months after diagnosis.
There are still ongoing debates whether MFOS represent multiple true primaries or metastatic disease. Many authors have proposed that MFOS is caused by multiple primary tumors because osteosarcoma manifests as a single primary bone lesion that most frequently metastasizes to the lung and less frequently has bony metastasis [1]. Parham et al. [5] reviewed the clinical and pathologic features of nine pediatric patients with MFOS and found no significant difference between patients presenting with or without pulmonary metastases. They concluded that the absence of pulmonary metastasis is not sufficient to prove the hypothesis of multiple primary origins. Mohoney et al. [6] agreed that metachronous MFOS probably represent metastatic osteosarcoma to bone because the pulmonary metastasis is frequently combined. However, they thought that some cases of metachronous MFOS might represent new primary lesions occurring in damaged or dystrophic mesenchymal tissue which have a propensity to undergo sarcomatous degeneration at a later stage.
Jaffree et al. [7] studied the metastatic patterns of osteosarcoma and concluded that the median time for lung metastasis (LM) was 5-6 months after starting treatment, for extra pulmonary metastasis (EPM) 9-10 months. First metastases after 24 months were infrequent, especially in children. With delay in the appearance of metastases, whether LM or EPM, post-metastatic survival lengthened. Local recurrence usually appeared within 6-8 months and was shown to lead to increased frequency of osseous metastases. It is suggested that terminal dissemination may often be tertiary but not always from a pulmonary secondary.
We conclude that metachronous MFOS is an extremely rare disease that seems to be osteosarcoma with metastases and we believe that our patient fits into type II metachronous MFOS as per Amstutz classification. If we make use of multiple modalities like Bone scan, CT and MRI for patient evaluation we can improve the sensitivity of detection and extent of progression of the disease.
| References | | |
| 1. | Lee HJ, Kim IO, Kim WS, Cheon JE, Kim KW, Yeon KM. Metachronous multifocal osteosarcoma: A case report and literature review. Journal of Clinical Imaging 2000 Jan- Feb; 26 (1): 63-68.  |
| 2. | Resnick D, Kyriacos M, Greenway GD. Tumors and tumor like lesions of bone: imaging and pathology of specific lesions. In: Resnick D, editor. Diagnosis of bone and joint disorders. Philadelphia: Saunders, 1995: 3697-9. |
| 3. | Unni KK. Dahlin's bone tumors: general aspect and data on11087 cases. 5th ed. Philadelphia: Lippincott-Raven, 1996:143-83. |
| 4. | Olson PN, Prewitt L, Griffiths HJ, Cherkna B. Case report 703. Skeletal Radiology 1991; 20 (8): 624-7. |
| 5. | Parham DM, Pratt CB, Parvey LS, Webber BL, Champion J. Childhood multifocal osteosarcoma. Clinicopathologic and radiologic correlates. Cancer 1985; 55: 2653-8. |
| 6. | Mohoney JP, Spanier SS, Morris JL. Multifocal osteosarcoma: a case report and review of literature. Cancer 1979; 44:1897-907. |
| 7. | Jeffree GM, Price CHG, Sissons HA. The metastatic patterns of osteosarcoma. British Journal of Cancer 1975; 32:87-107. |
Correspondence Address:
J P Singh
Dept of Radiodiagnosis, Mohan Das Oswal Cancer Hospital, G.T. Road, Sherpur, By Pass, Ludhiana
India
 Source of Support: None, Conflict of Interest: None  | Check |
Figures
[Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4], [Figure - 5] |
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