- Case Report
- Open Access
A case of recurrent giant cell tumor of bone with malignant transformation and benign pulmonary metastases
© Miller et al; licensee BioMed Central Ltd. 2010
- Received: 11 August 2010
- Accepted: 22 September 2010
- Published: 22 September 2010
Giant cell tumor (GCT) of bone is a locally destructive tumor that occurs predominantly in long bones of post-pubertal adolescents and young adults, where it occurs in the epiphysis. The majority are treated by aggressive curettage or resection. Vascular invasion outside the boundary of the tumor can be seen. Metastasis, with identical morphology to the primary tumor, occurs in a few percent of cases, usually to the lung. On occasion GCTs of bone undergo frank malignant transformation to undifferentiated sarcomas. Here we report a case of GCT of bone that at the time of recurrence was found to have undergone malignant transformation. Concurrent metastases were found in the lung, but these were non-transformed GCT.
- Multinucleated Giant Cell
- Giant Cell Tumor
- Giant Cell Tumor
- Undifferentiated Pleomorphic Sarcoma
- Lineage Specific Marker
Giant cell tumor of bone is a locally destructive tumor that occurs predominantly in long bones of post pubertal adolescents and young adults, where it occurs in the epiphysis. The majority are treated by aggressive curettage or resection. Histologically, giant cell tumor of bone classically shows many large multinucleated giant cells with interspersed haphazardly arranged mononuclear cells, and the nuclear features of both elements are described as similar. Some tumors also have areas with a fascicular or storiform pattern devoid of giant cells resembling a benign fibrous histiocytoma. Vascular invasion outside the boundary of the tumor can be seen. The rate of local recurrence varies among centers and is influenced by the completeness of surgical treatment, with high speed burring, adjuvants, and bone cement adding to the effectiveness of curettage treatment. Unresectable tumors such as large sacral masses can be treated with radiation. New therapies targeting the Receptor Activator of NF-κB (RANK) signaling pathway, such as with the anti-RANK ligand antibody denosumomab are in early stages of investigation. Metastasis, with identical morphology to the primary tumor, occurs in a few percent of cases, usually to the lung. These cases are treated with wedge resection with good long term outlook. On occasion giant cell tumors of bone undergo frank malignant transformation to undifferentiated sarcomas. Here we report a case of giant cell tumor of bone that at the time of recurrence was found to have undergone malignant transformation. Concurrent metastases were found in the lung, but these were non-transformed giant cell tumor. Contemporaneous histologically benign pulmonary metastases and locally recurrent giant cell tumor of bone with transformation to sarcoma has not to our knowledge been previously reported.
This study was performed with the approval of the Rush University IRB, ORA#: 09092501-CA01. The study included detailed clinical information, imaging and pathology. Tissue was fixed in 10% buffered formalin at room temperature and dehydrated and paraffin embedded in overnight processing. Immunohistochemical stains were performed as follows: The following antibodies were used on Ventana Benchmark and the manufacturer's solution CC1 for antigen retrieval.: CD4 (Biocare Medical, Concord, CA) 1:10 dilution, CD43 (Cell Marque, Rocklin, CA) prediluted, P63 (Fisher Scientific, Pittsburgh, PA) 1:500. Ki-67 immunostaining was performed on the Dako autostainer PLUS with FLEX Envision chemistry using clone MIB-1 (Dako, Carpentaria, CA), at 1:400 dilution after citrate buffer antigen retrieval under pressure in a microwave oven. Images were captured on an Olympus BX41 microscope with a Spot Insight color camera with Spot Advanced software.
A 29 year-old male was diagnosed with a giant cell tumor of the left proximal tibia at an outside institution in January 2005. He underwent intralesional curettage followed by heat cauterization and methacrylation. Eight months later, in September 2005, the patient had a local recurrence of the tumor which was treated with cement removal and repeat curettage followed by argon beam ablation and repeat cementing.
We report here the case of a giant cell tumor of the proximal tibia of a 29 year old man treated with aggressive curettage that recurred 4 years later with malignant transformation and "benign" metastases to the lung. Recurrence, malignant transformation, and metastasis with "benign" morphology all occur in giant cell tumor of bone. This is the first report of all three occurring in a single patient. The influence of local recurrence on malignant transformation and pulmonary metastases is largely unknown. Malignant transformation has been reported most frequently with radiation even in nonrecurrent tumors. Similarly, benign pulmonary metastases have been reported to occur in recurrent and nonrecurrent tumors. The current report adds little to the discussion of the fate of locally recurrent giant cell tumor of bone. The molecular pathways leading to giant cell tumors of bone are still largely uncharacterized.
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in Chief of this Journal.
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