ArticleBone2020
Dissecting the mechanisms of bone loss in Gorham-Stout disease.
Article in Bone, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 27 papers.
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Who cites it
27 citing papers in PubMed, 43 citations in OpenAlex.
- Gorham-Stout disease with thoracic involvement: pathogenic mechanisms, respiratory complications, and multimodal therapies.Orphanet journal of rare diseases · 2026Review
- Clavicular Gorham-Stout disease: a rare case report and literature review.Frontiers in endocrinology · 2026Review
- Trp53 loss drives the neoplastic transformation of Pik3caH1047R-induced vascular malformation in a mouse model.PloS one · 2026Article
- Structural Rearrangements and Selection Promote Phenotypic Evolution in Anolis Lizards.Genome biology and evolution · 2025Article
- Phloridzin Mitigates Gorham-Stout Disease by Dual Inhibition of Osteoclastogenesis Related to the CaACS omega · 2025Article
- Clinical and CT imaging features of different types of thoracic complex lymphatic anomaly.Quantitative imaging in medicine and surgery · 2025Article
- Exploring the genetic alterations of Gorham-Stout disease.Frontiers in endocrinology · 2025Review
- Analysis of circulating osteoclast and osteogenic precursors in patients with Gorham-Stout disease.Journal of endocrinological investigation · 2024Article
- Treatment for Gorham-Stout syndrome with a combination of teriparatide and denosumab.Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA · 2024Article
- Editorial: Genetic and molecular determinants in bone health and diseases.Frontiers in endocrinology · 2024Article
- Case Report and Literature Review: Bisphosphonate, Sirolimus, and Atenolol Treatment in a 4-Year-Old Child Diagnosed with Gorham-Stout Disease.Pharmaceuticals (Basel, Switzerland) · 2023Article
- The Lymphatic Endothelial Cell Secretome Inhibits Osteoblast Differentiation and Bone Formation.Cells · 2023Article
- Gorham-Stout disease of spine accompanying chylothorax: a case description.Quantitative imaging in medicine and surgery · 2023Article
- Gorham-Stout syndrome, the challenge in diagnosis and unique in treatment: a case report.Journal of medical case reports · 2023Article
- The molecular mechanism of Gorham syndrome: an update.Frontiers in immunology · 2023Review
- Cystic angiomatosis in children: clinical experience and review of literature.World journal of surgical oncology · 2022Review
- Characterization of Extracellular Vesicles in Osteoporotic Patients Compared to Osteopenic and Healthy Controls.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2022Article
- Difficult Therapeutic Decisions in Gorham-Stout Disease-Case Report and Review of the Literature.International journal of environmental research and public health · 2022Review
- Osteopathy in Complex Lymphatic Anomalies.International journal of molecular sciences · 2022Review
- Refractory serositis in Gorham-Stout syndrome.Orphanet journal of rare diseases · 2022Article
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Authors and funding
15 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Gorham-Stout disease (GSD) is a rare disorder characterized by progressive osteolysis and angiomatous proliferation. Since the mechanisms leading to bone loss in GSD are not completely understood, we performed histological, serum, cellular and molecular analyses of 7 patients. Increased vessels, osteoclast number and osteocyte lacunar area were revealed in patients' bone biopsies. Biochemical analysis of sera showed high levels of ICTP, Sclerostin, VEGF-A and IL-6. In vitro experiments revealed increased osteoclast differentiation and activity, and impaired mineralization ability of osteoblasts. To evaluate the involvement of systemic factors in GSD, control cells were treated with patients' sera and displayed an increase of osteoclastogenesis, bone resorption activity and a reduction of osteoblast function. Interestingly, GSD sera stimulated the vessel formation by endothelial cells EA.hy926. These results suggest that bone cell autonomous alterations with the cooperation of systemic factors are involved in massive bone loss and angiomatous proliferation observed in GSD patients.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.