Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: a severe case of Gorham-Stout syndrome

Takahashi, Atsushi; Ogawa, Chitose; Kanazawa, Takashi; Watanabe, Hideomi; Suzuki, Makoto; Suzuki, Norio; Tsuchida, Yoshiaki; Morikawa, Akihiro; Kuwano, Hiroyuki

doi:10.1016/j.jpedsurg.2004.11.015

Cited by 72 publications

(76 citation statements)

References 11 publications

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“…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). Interferon α-2b was successfully used to treat one patient at a dosage of (7.5-15) million IU three times a week for five years (Dupond et al, 2010).…”

Section: Discussionmentioning

confidence: 99%

Gorham-Stout syndrome in mainland China: a case series of 67 patients and review of the literature

Yuan

et al. 2013

J. Zhejiang Univ. Sci. B

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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.

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Section: Discussionmentioning

confidence: 99%

Gorham-Stout syndrome in mainland China: a case series of 67 patients and review of the literature

Yuan

et al. 2013

J. Zhejiang Univ. Sci. B

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“…1,9 Although the exact nature of the disease remains unknown, the effects of hyperemia, changes in local pH-stimulating hydrolytic enzyme activity, and increased osteoclast activity have all been proposed as contributing factors. 10 Gorham disease occurs in men or women of any age group but is usually discovered before 40 years of age. It may involve any bone within the axial or appendicular skeleton, though spine and visceral involvement have higher morbidity and mortality.…”

Section: Discussionmentioning

confidence: 99%

“…Medical treatment options include antiosteoclastic agents (bisphosphonates), radiation therapy, and interferon ␣ 2b. 10,11 Although resection has been attempted in localized disease, no single therapy is effective. MR imaging typically shows re-placement of normal bone marrow with high T1 signal intensity from fat.…”

Section: Discussionmentioning

confidence: 99%

Intraosseous CSF Fistula in a Patient with Gorham Disease Resulting in Intracranial Hypotension

Adler

Gupta

Hess

et al. 2011

AJNR Am J Neuroradiol

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“…Zoledronic acid treatment has been effective with no recurrence in young people (15,16). Because of its antiangiogenic properties interferon alfa could be useful in GorhamStout disease with documented vessel proliferation with extended lymph-hemangiomatosis (13,17). Radiotherapy acts accelerating sclerosis of the proliferating blood vessels and prevents re-growth of these vessels which have been found to be extremely radiosensitive.…”

Section: Discussionmentioning

confidence: 99%

Two cases of Gorham-Stout disease with good response to zoledronic acid treatment

Brance¹,

Castiglioni

Cóccaro

et al. 2017

ccmbm

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SummaryGorham-Stout disease -also called vanishing bone syndrome -is a rare bone disease characterized by a progressive intra-osseous proliferation of non-neoplastic vascular tissue resulting in massive osteolysis. Here, we report two clinical cases of Gorham-Stout disease. Case 1: a 56-year-old woman with 20 years of history of pain and swell in elbows, ankles and wrist. Then she was diagnosed as systemic lupus erythematosus (SLE) with glomerulonephritis type III. After other pathologies were ruled out Gorham-Stout disease was diagnosed. Intravenous zoledronic acid (5 mg) was indicated and after third infusion a progressive improvement of pain, mobility and daily activities were observed. Case 2: a 70-years-old man with a history of pain and limited motion in the left shoulder without X-ray abnormality. Six months later pathological fracture in the left humerus occurred and after ruled out other pathologies Gorham-Stout disease was diagnosed. Intravenous zoledronic acid (5 mg) was indicated and a good response was observed after the first infusion. Nowadays just over 200 cases were reported. Gorham-Stout disease was reported in different bones, at different age presentation and severe physical deformities, disabilities, and life-threatening complications can occur. Two cases of Gorham-Stout disease with good response to zoledronic acid was reported in this article.

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Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: a severe case of Gorham-Stout syndrome

Cited by 72 publications

References 11 publications

Gorham-Stout syndrome in mainland China: a case series of 67 patients and review of the literature

Gorham-Stout syndrome in mainland China: a case series of 67 patients and review of the literature

Intraosseous CSF Fistula in a Patient with Gorham Disease Resulting in Intracranial Hypotension

Two cases of Gorham-Stout disease with good response to zoledronic acid treatment

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