“…One patient in our case series who just underwent calcium salts and vitamin D supplements achieved partial recovery after a follow-up of 5.5 years. However, Vinée et al (1994) reported that treatment with hormone combined with calcium salts and vitamins showed no efficacy. Interferon may be useful in disorders with vessel proliferation because of its antiangiogenic properties (Takahashi et al, 2005).…”
Section: Discussionmentioning
confidence: 99%
“…Surgical therapy includes resection of the lesion, reconstruction using bone grafts and/or prostheses (Woodward et al, 1981;Vinée et al, 1994;Wang and Lin, 2004;Escande et al, 2008), thoracic duct ligation, pleurodesis, and chest drainage. It has been reported that progression of bone graft has led to failure of reconstruction (Woodward et al, 1981;Kulenkampff et al, 1990;Paley et al, 2005); however, reconstruction using prostheses seems to be more effective.…”
Abstract:Objective: Gorham-Stout syndrome (GSS) is a rare disorder of uncertain etiology and unpredictable prognosis. This study aims to present a comprehensive understanding of this rare entity. Methods: A literature search in PubMed and three Chinese databases was performed to screen histologically proven GSS cases among Chinese residents in the mainland. We analyzed the patients' clinical characteristics, the value of different treatment modalities and their influence on the clinical outcome. Results: Sixty-seven cases were finally enrolled. There were 43 men (64.2%) and 24 women (35.8%). The mean age at diagnosis was 28 years (1.5-71 years). The most common clinical symptoms included pain (n=40, 59.7%), functional impairment (n=13, 19.4%), and swelling (n=12, 17.9%). The radiographic presentation of 37 cases (55.2%) was disappearance of a portion of the bone. The others presented as radiolucent foci in the intramedullary or subcortical regions. A total of 42 cases provided data on therapy, these included surgery (n=27, 40.3%), radiation therapy (n=6, 9.0%), surgery combined with radiation therapy (n=2, 3.0%), and medicine therapy (n=7, 10.4%). For 30 of these 42 cases, follow-up data were available: 21 cases had the disorder locally controlled and 9 had a symptom progression. Fortunately, the disease is not fatal in the majority of cases. Conclusions: GSS has no specific symptoms and it should be taken into consideration when an unclear massive osteolysis occurs. The efficacies of different treatment modalities are still unpredictable and further research is required to assess the values of different treatments.
“…One patient in our case series who just underwent calcium salts and vitamin D supplements achieved partial recovery after a follow-up of 5.5 years. However, Vinée et al (1994) reported that treatment with hormone combined with calcium salts and vitamins showed no efficacy. Interferon may be useful in disorders with vessel proliferation because of its antiangiogenic properties (Takahashi et al, 2005).…”
Section: Discussionmentioning
confidence: 99%
“…Surgical therapy includes resection of the lesion, reconstruction using bone grafts and/or prostheses (Woodward et al, 1981;Vinée et al, 1994;Wang and Lin, 2004;Escande et al, 2008), thoracic duct ligation, pleurodesis, and chest drainage. It has been reported that progression of bone graft has led to failure of reconstruction (Woodward et al, 1981;Kulenkampff et al, 1990;Paley et al, 2005); however, reconstruction using prostheses seems to be more effective.…”
Abstract:Objective: Gorham-Stout syndrome (GSS) is a rare disorder of uncertain etiology and unpredictable prognosis. This study aims to present a comprehensive understanding of this rare entity. Methods: A literature search in PubMed and three Chinese databases was performed to screen histologically proven GSS cases among Chinese residents in the mainland. We analyzed the patients' clinical characteristics, the value of different treatment modalities and their influence on the clinical outcome. Results: Sixty-seven cases were finally enrolled. There were 43 men (64.2%) and 24 women (35.8%). The mean age at diagnosis was 28 years (1.5-71 years). The most common clinical symptoms included pain (n=40, 59.7%), functional impairment (n=13, 19.4%), and swelling (n=12, 17.9%). The radiographic presentation of 37 cases (55.2%) was disappearance of a portion of the bone. The others presented as radiolucent foci in the intramedullary or subcortical regions. A total of 42 cases provided data on therapy, these included surgery (n=27, 40.3%), radiation therapy (n=6, 9.0%), surgery combined with radiation therapy (n=2, 3.0%), and medicine therapy (n=7, 10.4%). For 30 of these 42 cases, follow-up data were available: 21 cases had the disorder locally controlled and 9 had a symptom progression. Fortunately, the disease is not fatal in the majority of cases. Conclusions: GSS has no specific symptoms and it should be taken into consideration when an unclear massive osteolysis occurs. The efficacies of different treatment modalities are still unpredictable and further research is required to assess the values of different treatments.
“…Plain radiographs, 6,28,36,71,77,79,116,136,143 radioisotope bone scans, 50,79,81,111,116,143 computed tomography (CT), 71,76,77,79,87,106,143 and magnetic resonance imaging (MRI) 76,77,87,106,116,138,143 have all been used in such evaluations.…”
“…Gorham disease is an extremely rare and mysterious bone disease of unknown etiology, which is histopathologically characterized by a proliferation of thin-walled vascular channels in bone (Vinée et al 1994). Since first described by Jackson (1838), it was called by various names: vanishing bone disease, acute absorption of bone, phantom bone, primary lymphangioma, progressive osteolysis and disappearing bone (Gorham and Stout 1955).…”
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