Benefit of a 17-year long-term bisphosphonate therapy in a patient with Gorham–Stout syndrome

Lehmann, G.; Pfeil, Alexander; Böttcher, Joachim; Kaiser, Werner A.; Füller, Jürgen; Hein, Gert; Wolf, Günter

doi:10.1007/s00402-008-0742-3

Cited by 43 publications

(46 citation statements)

References 21 publications

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“…Bisphosphonate is helpful because of its putative antiosteoclastic and anti-angiogenic activity. Lehmann et al (2009) reported that a case of GSS was successfully treated with bisphosphonates for more than 17 years. Hammer et al (2005) reported that mono-therapy with bisphosphonates (30 mg intravenous every three months) controlled the disorder during two years of follow-up.…”

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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“…Heyd et al reported a 77% to 80% success rate of radiotherapy, and recommended a total dose ranging from 36 to 45 Gy (Heyd et al 2011a). As for chemotherapy, bisphosphonates (Lehmann et al 2009) and interferon α-2b (Hagberg et al 1997) have most commonly been used. Other pharmaceuticals, including the anti-VEGF-A antibody (Grunewald et al 2010), propranolol (Nir et al 2014), low molecular weight heparin (Brodszki et al 2011), and rapamycin (Hammill et al 2011), have also been tested experimentally.…”

Section: Discussionmentioning

confidence: 99%

“…The precise etiology and pathophysiology of the disease remain poorly understood (Lehmann et al 2009;Dellinger et al 2014). Furthermore, due to uncertainty about the underlying cause, the appropriate treatment and prognosis currently remain uncertain.…”

Section: Introductionmentioning

confidence: 99%

Successful Management of Gorham-Stout Disease in the Cervical Spine by Combined Conservative and Surgical Treatments: A Case Report

Tateda

Aizawa

Hashimoto

et al. 2017

Tohoku J. Exp. Med.

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Gorham-Stout disease (GSD) is a rare condition characterized by intraosseous proliferation of endotheliallined vessels and progressive osteolysis. The precise etiology and pathophysiology of the disease remain poorly understood. Current therapeutic options for GSD include chemotherapy, radiotherapy, and surgical resection, but the surgical treatment of GSD is difficult, especially in the spinal lesion. The indication of wide-margined resection was limited because of anatomical features. Herein, we report a case of GSD of the cervical spine in which the lesions were successfully stabilized with combined conservative and surgical treatments. A 15-year-old male patient was admitted because of severe neck pain. The patient presented no neurological deficiency. However, the radiological findings revealed osteolytic lesions on the laminae and vertebrae between C1 to C5. An open biopsy confirmed an irregular, thin-walled vessel formation in the bone trabeculae, which was diagnosed as GSD. Conservative treatment was initiated with chemotherapy and radiotherapy. After one and a half year, the osteolytic condition had regressed. Spinal fusion surgery was then performed from C2 to C5 to prevent for progression of the cervical kyphotic changes, and spinal fusion was confirmed 7 months after the surgery. The patient showed no recurrence of GSD in the 5-year follow-up period after surgery. We were able to provide successful treatment by giving priority to the combined conservative treatments. If a patient has no severe deformity or progressive neurologic deficits, it might be better to prioritize conservative treatments and to perform the surgery after the osteolytic changes have stopped.

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“…Мнения о роли остеокластов в деструкции костной ткани при GSD противоречивы: ряд исследователей подтверждает их высокую активность [1,5,6,18,22,26,32,36,42,44,48,50], другие отмечают их отсутствие в оча-гах остеолиза [12,13,21,45,51]. При-чина такого противоречия, возмож-но, обусловлена проведением гисто-логического исследования на разных стадиях процесса [4].…”

Section: рисunclassified

A Rare Variant of Angiogenic Vertebral Body Destruction in a Child: Gorham’s Disease?

Мушкин¹,

Евсеев²,

Николаев³

et al. 2018

Hir. pozvonoč.

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Benefit of a 17-year long-term bisphosphonate therapy in a patient with Gorham–Stout syndrome

Cited by 43 publications

References 21 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

Successful Management of Gorham-Stout Disease in the Cervical Spine by Combined Conservative and Surgical Treatments: A Case Report

A Rare Variant of Angiogenic Vertebral Body Destruction in a Child: Gorham’s Disease?

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