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Journal of Clinical Oncology, Vol 23, No 18 (June 20), 2005: pp. 4226-4229 © 2005 American Society of Clinical Oncology. DOI: 10.1200/JCO.2005.05.055
CNS Manifestations of MalignanciesCASE 2. Skull and Brain Metastasis From Tibial OsteosarcomaSurgical Neurology Branch, National Institutes of Neurological Disorders and Stroke, Bethesda, MD
Laboratory of Pathology, National Cancer Institute, National Institutes of Health, Bethesda, MD A 26-year-old man presented with a history of right-leg pain, first with exercise, then, over the course of several weeks, at rest. Physical examination revealed a 3 x 2 cm firm mass along the tibia of the right leg and plain x-rays suggested an osteolytic lesion. Simultaneously, chest x-ray revealed multiple small nodules in both lungs suggestive of metastatic cancer. Needle biopsy of the tibial lesion demonstrated an osteosarcoma (OS). The patient was treated with two cycles of methotrexate and cisplatin, followed by resection of the right tibial lesion and cadaveric bone graft, then video-assisted thoracoscopic wedge resection of one remaining left pulmonary tumor. He was then treated with cisplatin, methotrexate, etoposide, and ifosfamide for 36 weeks. The patient had an asymptomatic recurrence in the left femur 18 months after initial presentation treated with resection of the femoral neck and left hip arthrodesis, from which he recovered well. Methotrexate and cisplatin were resumed for 12 months, with no evidence of recurrent disease. The patient did well until 3 years after initial presentation, at which point he bumped his head firmly against a car door. Later that day he noticed a 3 x 3 cm swelling under his scalp at that location. It was soft, minimally tender, and moved with the scalp. Two weeks later, the scalp mass had not resolved and he sought medical attention. On examination, it measured 4 x 4 cm and was elevated 2 cm above the scalp contour. The patient's neurological examination was normal. Needle biopsy of the lesion was inconclusive. Neuroimaging imaging (Fig 1) demonstrated a 6 x 6 x 3 cm extra-axial mass with extension through the inner and outer tables of the skull, with compression of the underlying brain. A lateral skull x-ray shows thinning of calvarium (Fig 1A, arrow) and a soft tissue mass above. On T1-weighted, gadolinium-enhanced magnetic resonance (MR) images (Fig 1B, axial; Fig 1C, coronal view) one sees the full extent of the mass, which goes beneath the scalp, penetrates the dura, and compresses the brain; the small bars at the bottom of the frames of Figure 1B and C are 1-cm markers. Chest x-ray demonstrated new bilateral pulmonary nodules that were felt to be surgically accessible. The patient underwent craniotomy and gross total resection of the tumor, which arose from the calvarium and elevated but did not invade the scalp. A 1 to 2 cm rim of normal bone was removed circumferentially around the bone tumor. The tumor invaded through the dura and compressed the brain just anterior to the cortical motor strip. All tumor was removed except for a small 2 x 2 mm cuff of tumor that invaded the lateral wall of the superior sagittal sinus; the dura overlying the tumor was removed, taking with it a 1-cm border of normal-appearing dura. Pathology was consistent with OS. Figure 2 shows low- (A, 100x), medium- (B, 200x), and high-power (C, 400x) views of a typical osteoblastic OS, composed of osteoid-producing, spindle-shaped malignant tumor cells. The patient refused additional treatment with chemotherapy and/or focused radiotherapy. Within 2 months, the intracranial disease progressed rapidly, with a large recurrence locally, as well as spread within the dura to the left orbit, causing proptosis and blindness. The patient died within 4 months of the initial presentation of his scalp mass due to cranial and pulmonary disease.
OS, the most common primary malignant bone tumor, is composed of pleomorphic, spindle-shaped cells that produce osteoid.1-4 The most common clinical presentation is pain in the involved bone, with or without a tissue mass.1-4 OS can occur in any bone, but generally, in children and young adults, in the metaphysis of a long bone; the most common sites are, in order, the distal femur, proximal tibia, and proximal humerus.1-4 The axial skeletal is involved in 10% of the time and rarely is the skull the site of tumor origin.5-7 While clinically evident metastases are found in 20% of patients, nearly all patients have subclinical, micrometastatic disease at presentation. Metastatic deposits are most common in the lungs, CNS and gastrointestinal tract.1,5-7 Survival of patients with metastatic disease reaches 40% at 5 years with aggressive surgical resection of the primary and intensive chemotherapy.5-7 Patients who relapse after intensive induction and adjuvant chemotherapy and surgery tend to fare poorly.4-7 Death as a result of OS is almost always secondary to progressive pulmonary metastatic disease.1,4-7 However, in situations such as this, failure within the CNS may lead to death due to the difficulty of gaining adequate local or regional control. Authors' Disclosures of Potential Conflicts of Interest The authors indicated no potential conflicts of interest.
REFERENCES 1. Meyers PA, Gorlick R: Osteosarcoma. Pediatr Clin North Am 44:973-989, 1997[CrossRef][Medline] 2. Gurney JG, Severson RK, Davis S, et al: Incidence of cancer in children in the United States: Sex-, race-, and 1-year age-specific rates by histologic type. Cancer 75:2186-2195, 1995[CrossRef][Medline] 3. Harris MB, Gieser P, Goorin AM, et al: Treatment of metastatic osteosarcoma at diagnosis: A Pediatric Oncology Group study. J Clin Oncol 16:3641-3648, 1998[Abstract]
4. Saeter G: ESMO Minimum Clinical Recommendations for diagnosis, treatment and follow-up of osteosarcoma. Ann Oncol 14:1165-1166, 2003 5. Saeter G, Hoie J, Stenwig AE, et al: Systemic relapse of patients with osteogenic sarcoma: Prognostic factors for long term survival. Cancer 75:1084-1093, 1995[CrossRef][Medline]
6. Ferrari S, Briccoli A, Mercuri M, et al: Postrelapse survival in osteosarcoma of the extremities: Prognostic factors for long-term survival. J Clin Oncol 21:710-715, 2003 7. Giuliano AE, Feig S, Eilber FR: Changing metastatic patterns of osteosarcoma. Cancer 54:2160-4, 1984[CrossRef][Medline]
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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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