PMC9691950_01
Lateral plain radiography of the tibia (A) showed a polycystic expansive bone destruction area in the anterior cortex of the upper segment of the right tibia with a sclerotic edge that was irregular and lobulated, and the long axis of the lesion was consistent with the tibia. The sagittal reformation image of CT (B) and the axial image of CT (G) clearly showed that the lesion is an eccentric polycystic expansive transparent area while the bone cortex expands and thins. Cortical interruption could be seen in some layers with a small periosteal reaction in some parts, and cortical hyperplasia, sclerosis, and cortical destruction could be visualized around the lesions. MRI showed abnormal signal lesions of the upper tibia on the right side with low signal intensities on T1-weighted images (T1WI) (C) and high signal intensities on sagittal and axial T2-weighted images (T2WI) (D, H). The lesions were significantly enhanced on postcontrast scans (E). The postoperative plain radiography of the lower limb (I-K) showed postoperative changes of tumor resection of the right tibia, free transplantation of the left fibula, and internal fixation. The gross specimen during the operation (F) showed a sagittal view of the tumor and the tibia, which had a good comparison with the lateral plain radiography and sagittal CT and MRI images (A-E). Tumor pathological HE sections (L, M) showed irregularly woven bone trabeculae surrounded by osteoblasts, with many fibrous matrix and small nests of epithelial cells. Immunohistochemistry (N, O) showed nests of small tumor cells positive for cytokeratin CK19.
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The immunohistochemistry results showed Ki-67 (+1%), SATB2 (+), CK19 (1+), AE1/AE3 (1+), CK18 (-), EMA (-), P63 (1+), S-100 (-), MDM2 (2+), SMA (-), and beta-catenin (2+, cytoplasm) ( Figure 1 ).