Gastrointestinal stromal tumors are a heterogeneous group with a wide spectrum of histologic features. We describe the first case of 61-year-old woman who presented gastrointestinal stromal tumors of the ampulla of Vater with osteoclast-like giant cells surrounding osteoid-like material and aneurismal bone cyst-like areas. The phenotype was supported by light microscopy and corroborated by immunohistochemistry analysis. Because of the presence of osteoid-like and aneurismal bone cyst-like components, it is first necessary to make differential diagnosis with other entities such as metastatic osteosarcoma. Our case shows another form of differentiation that has not previously been reported.
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