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- Title
Treatment for Gorham-Stout syndrome with a combination of teriparatide and denosumab.
- Authors
Zhang, Liqin; Wang, Weihong; Wen, Liang; Xue, Fenglin; Li, Jingyi; Qian, Yemei
- Abstract
Gorham-Stout syndrome is an aggressive, non-hereditary, and rare disease affecting bone metabolism. Its etiology and pathogenesis remain elusive. The syndrome manifests with diverse clinical symptoms, often leading to frequent misdiagnoses and presenting challenges in treatment. In this study, we report a case of cranial and maxillary osteolysis in a 47-year-old female patient with somatic mutations in the VEGF-A, VEGF-B, and VEGF-C genes and the EPHB4 gene. After treatment with bisphosphonates, this patient still had persistent resorption of the mandible, but switching to a teriparatide and denosumab combination yielded substantial improvement. This study is the first report to show that teriparatide combined with denosumab can be used to treat Gorham-Stout syndrome.
- Subjects
THERAPEUTIC use of monoclonal antibodies; BONE resorption; COMBINATION drug therapy; VASCULAR endothelial growth factors; TERIPARATIDE; DIPHOSPHONATES; MAXILLARY diseases; COMPUTED tomography; TREATMENT effectiveness; GENES; GENETIC mutation; MANDIBLE
- Publication
Osteoporosis International, 2024, Vol 35, Issue 4, p727
- ISSN
0937-941X
- Publication type
Article
- DOI
10.1007/s00198-023-06995-1