ReviewMedicine2018
Successful treatment of Gorham-Stout syndrome in the spine by vertebroplasty with cement augmentation: A case report and literature review.
Review in Medicine, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers, 4 of them syntheses that pooled it.
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Who cites it
23 citing papers in PubMed, 4 syntheses or guidelines pooled it, 29 citations in OpenAlex.
- Japanese clinical practice guidelines for vascular tumors, vascular malformations, lymphatic malformations, and lymphangiomatosis 2022.Surgery today · 2026Guideline
- Japanese Clinical Practice Guidelines for Vascular Tumors, Vascular Malformations, Lymphatic Malformations, and Lymphangiomatosis 2022.The Journal of dermatology · 2026Guideline
- Japanese clinical practice guidelines for vascular tumors, vascular malformations, lymphatic malformations, and lymphangiomatosis 2022.Japanese journal of radiology · 2026Guideline
- Japanese clinical practice guidelines for vascular tumors, vascular malformations, lymphatic malformations, and Lymphangiomatosis 2022.Pediatrics international : official journal of the Japan Pediatric SocietyGuideline
- Skeletal manifestations in lymphatic diseases: nonenhanced magnetic resonance lymphography.Insights into imaging · 2026Article
- Japanese Clinical Practice Guidelines for Vascular Tumors, Vascular Malformations, Lymphatic Malformations, and Lymphangiomatosis 2022.Journal of plastic and reconstructive surgery · 2026Review
- Gorham-Stout disease with thoracic involvement: pathogenic mechanisms, respiratory complications, and multimodal therapies.Orphanet journal of rare diseases · 2026Review
- Reduced expression of semaphorin 3A in osteoclasts causes lymphatic expansion in a Gorham-Stout disease (GSD) mouse model.Journal of Zhejiang University. Science. B · 2024Article
- The molecular mechanism of Gorham syndrome: an update.Frontiers in immunology · 2023Review
- Gorham-Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report.Endocrinology, diabetes & metabolism case reports · 2022Article
- Gorham-Stout disease: good results of bisphosphonate treatment in 6 of 7 patients.Acta orthopaedica · 2020Article
- Unusual case of chylothorax with unilateral limb swelling.BMJ case reports · 2020Article
- A Large Skull Defect Due to Gorham-Stout Disease: Case Report and Literature Review on Pathogenesis, Diagnosis, and Treatment.Frontiers in endocrinology · 2020Review
- Gorham-Stout disease of the malleolus: a rare case report.BMC musculoskeletal disorders · 2019Article
- Surgical treatment of chondrosarcoma of the sacrum with cement augmentation: A case report.Medicine · 2019Article
- Management of giant benign fibrous histiocytoma in the spinal region with pleural involvement: A case report.Medicine · 2019Article
- Surgical treatment of giant chordoma in the thoracic spine combining thoracoscopic and posterior spinal surgery: A case report.Medicine · 2019Article
- A report of two children with Gorham-Stout disease.BMC pediatrics · 2019Article
- A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report.BMC musculoskeletal disorders · 2019Article
- 99mTc-SC lymphoscintigraphy and SPECT/CT findings in a case report of Gorham-Stout disease presenting with chylothorax and bone pain.Medicine · 2019Article
Corrections and comments
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Authors and funding
6 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
rationaleGorham-Stout syndrome in the spine is extremely rare, and there is no standard curative management thus far. The objective of this article is to report a very rare case of Gorham-Stout syndrome of the lumbar and sacral spine with chylothorax and chyloperitoneum successfully treated by combination of vertebroplasty with cement augmentation and medication treatment. We described the clinical characteristics and postoperative therapy of the patient, and reviewed all of the published cases of Gorham-Stout syndrome of the lumbar and sacral spine. PATIENT CONCERNS: A 31-year-old man presented with increasingly serious abdominal distention and back pain. MRI showed massive bony destruction of the spine and pelvis. CT and ultrasonography demonstrated massive ascites and mild hydrothorax. DIAGNOSES: We believe this is the first report of a case of Gorham-Stout syndrome with both chylothorax and chyloperitoneum.
interventionsChest and abdominal cavity puncture was performed for symptomatic relief and the test results confirmed chylothorax and chyloperitoneum. Tissue biopsy and percutaneous vertebroplasty at L5 were performed and the postoperative pathology together with symptoms and examinations were reported to be consistent with Gorham-Stout syndrome. Subsequently, we administered combination medical treatment consisting of interferon-α-2b, zoledronic acid and calcitriol. OUTCOMES: At the 1-year and 2-year follow-up visit, he had nearly full complete remission and reported palliative back pain. Moreover, the amount of pleural and peritoneal fluid was successfully reduced gradually. LESSONS: Vertebroplasty by cement augmentation may be a treatment option for patients with Gorham-Stout Syndrome in the spine who cannot undergo appropriate surgery or decline open surgery. This represents a safe and minimally invasive approach to sustainably relieve pain and stabilize vertebral bodies with Gorham-Stout syndrome in the spine.
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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.