Introduction The Gorham-Stout Syndrome (Gorham's massive osteolysis) is a rare condition in which spontaneous, progressive resorption of bone occurs. The etiology is poorly understood. Case presentation We reported here a patient who had begun insidiously and was characterized by extensive osteolysis in the left upper limb, with progressive carpal bone resorption. The characteristic radiological and CT findings were presented and the clinical presentation, diagnosis and surgical treatment were discussed in the case. Conclusion Gorham's disease is rarely involved in the whole upper limb. Histopathological evidences suggest that extensive osteolysis is caused by hemangiomatosis. The characteristic imaging findings may help us to differentiate from malignant neoplasm with progressive bony destruction.
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