Japanese Journal of Oral and Maxillofacial Surgery
Online ISSN : 2186-1579
Print ISSN : 0021-5163
ISSN-L : 0021-5163
Case reports
A case of Gorham‒Stout disease with osteolysis from the mandible to the skull base
Hiroki TANIGUCHIKento NAKAMURAKenji OHARAKazuto OKABEShinichiro KATOYasuyuki SHIBUYA
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2025 Volume 71 Issue 3 Pages 139-146

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Abstract
 Gorham-Stout disease (GSD) is a rare lymphangitic disease that causes continuous and destructive dissolution of bone. It is extremely rare for it to occur in the maxillofacial bone.
 A 21-year-old male patient visited his family dentist due to discomfort on the right side of his mandible. Panoramic radiographs revealed a radiolucency in the right mandible, and the patient was referred to the Department of Oral and Maxillofacial Surgery in our hospital for further examination and treatment. CT images showed bone resorption from the right side of the mandible to the base of the skull, and together with pathological findings, the patient was diagnosed with GSD. After the commencement of treatment with zoledronic acid, denosumab and sirolimus, the lesion decreased in size and the osteolytic lesion improved.
 The findings indicate that antiresorptive agents and mTOR inhibitors may serve to stabilize this disease and facilitate gradual improvement in prognosis.
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© 2025 Japanese Society of Oral and Mxillofacial Surgeons
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