ArticleActa orthopaedica2020
Gorham-Stout disease: good results of bisphosphonate treatment in 6 of 7 patients.
Article in Acta orthopaedica, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers, 1 of them a synthesis that pooled it.
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Who cites it
18 citing papers in PubMed, 1 synthesis or guideline pooled it, 28 citations in OpenAlex.
- Clinical features and current management experience in Gorham-Stout disease: a systematic review.Orphanet journal of rare diseases · 2025Pooled it
- Gorham-Stout disease of the craniovertebral junction presenting with bow hunter syndrome and cerebellar infarction treated by occipitocervical fusion: illustrative case.Journal of neurosurgery. Case lessons · 2026Article
- Therapeutic Efficacy of Sirolimus in Skeletal Manifestations of Gorham-Stout Disease in Adults: A Systematic Review.Cureus · 2026Review
- Pathological Fractures in Rare Bone Diseases: Clinical Cases.Acta medica Lituanica · 2026Article
- Phloridzin Mitigates Gorham-Stout Disease by Dual Inhibition of Osteoclastogenesis Related to the CaACS omega · 2025Article
- Exploring Gorham-Strout syndrome in a young adult: an illustrative case report and literature review.Oxford medical case reports · 2025Article
- An Atypical Cause of a Child Limp: A Gorham-Stout Disease with a Vanishing Hip.Revista brasileira de ortopedia · 2024Article
- Article
- 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
- 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
- The spectrum of imaging manifestations of Gorham-Stout disease: a novel dynamic contrast-enhanced MR lymphangiography.Orphanet journal of rare diseases · 2023Article
- Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report.Frontiers in surgery · 2023Article
- A new therapeutic approach in Gorham-Stout disease: a case report.Frontiers in surgery · 2023Article
- The molecular mechanism of Gorham syndrome: an update.Frontiers in immunology · 2023Review
- Systematic lymphatic abnormality-related osseous lesions: a study based on CT lymphangiography.Quantitative imaging in medicine and surgery · 2022Article
- Current concepts from diagnosis to management in Gorham-Stout disease: a systematic narrative review of about 350 cases.EFORT open reviews · 2022Review
- A Case of Gorham-Stout Disease Treated with Fistula Closure by Transmeatal Approach.Yonago acta medica · 2021Article
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Authors and funding
9 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Background and purpose - Gorham-Stout disease (GSD) is a rare mono- or polyostotic condition characterized by idiopathic intraosseous proliferation of angiomatous structures resulting in progressive destruction and resorption of bone. Little is known about the course of disease and no previous study has evaluated patients' quality of life (QoL).Patients and methods - This is a retrospective analysis of 7 consecutive patients (5 males) with a median age at diagnosis of 14 years and a median follow-up of 7 years who were diagnosed with GSD in our department between 1995 and 2018. Data regarding clinical, radiographic, and histopathological features, and treatment, as well as sequelae and their subsequent therapy, were obtained. QoL was assessed by Musculoskeletal Tumor Society Score (MSTS), Toronto Extremity Salvage Score (TESS), and Reintegration to Normal Living (RNL) Index.Results - 3 patients had a monoostotic and 4 patients a polyostotic disease. Besides a diagnostic biopsy, 4 of the 7 patients had to undergo 8 surgeries to treat evolving sequelae. Using an off-label therapy with bisphosphonates in 6 patients, a stable disease state was achieved in 5 patients after a median of 20 months. The median MSTS, TESS, and RNL Index at last follow-up was between 87% and 79%.Interpretation - Due to its rare occurrence, diagnosis and treatment of GSD remain challenging. Off-label treatment with bisphosphonates appears to lead to a stable disease state in the majority of patients. QoL varies depending on the individual manifestations but good to excellent results can be achieved even in complex polyostotic cases with a history of possibly life-threatening sequelae.
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