Literature DB >> 28367856

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

Satoshi Tateda1, Toshimi Aizawa, Ko Hashimoto, Haruo Kanno, Susumu Ohtsu, Eiji Itoi, Hiroshi Ozawa.   

Abstract

Gorham-Stout disease (GSD) is a rare condition characterized by intraosseous proliferation of endothelial-lined 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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Year:  2017        PMID: 28367856     DOI: 10.1620/tjem.241.249

Source DB:  PubMed          Journal:  Tohoku J Exp Med        ISSN: 0040-8727            Impact factor:   1.848


  7 in total

1.  99mTc-SC lymphoscintigraphy and SPECT/CT findings in a case report of Gorham-Stout disease presenting with chylothorax and bone pain.

Authors:  Yuanyuan Jiang; Guozhu Hou; Wuying Cheng
Journal:  Medicine (Baltimore)       Date:  2019-04       Impact factor: 1.817

2.  Gorham-Stout disease of the spine presenting with intracranial hypotension and cerebrospinal fluid leak: A case report and review of the literature.

Authors:  Hana Yokoi; Vikram Chakravarthy; Benjamin Whiting; Scott E Kilpatrick; Tsulee Chen; Ajit Krishnaney
Journal:  Surg Neurol Int       Date:  2020-12-29

Review 3.  Current concepts from diagnosis to management in Gorham-Stout disease: a systematic narrative review of about 350 cases.

Authors:  Andrea Angelini; Nicolò Mosele; Elisa Pagliarini; Pietro Ruggieri
Journal:  EFORT Open Rev       Date:  2022-01-11

4.  Gorham-Stout disease: A multirod lumbar reconstruction with off-label suppression-remission therapy.

Authors:  Ajay Krishnan; Aditya Raj; Devanand Degulmadi; Shivanand Mayi; Raviranjan Rai; Shiv Kumar Bali; Vatsal Parmar; Prarthan Chirag Amin; Preety Krishnan; Mirant Dave; Bharat Dave
Journal:  Surg Neurol Int       Date:  2022-04-08

5.  Gorham-Stout disease in the rib and thoracic spine with spinal injury treated with radiotherapy, zoledronic acid, vitamin D, and propranolol: A case report and literature review.

Authors:  Kazutaka Koto; Kentarou Inui; Megumi Itoi; Kyoko Itoh
Journal:  Mol Clin Oncol       Date:  2019-10-07

6.  Utility of the spinal instability neoplastic score to identify patients with Gorham-Stout disease requiring spine surgery.

Authors:  Chloe Gui; Brett Rocos; Laura-Nanna Lohkamp; Angela Cheung; Robert Bleakney; Eric Massicotte
Journal:  Surg Neurol Int       Date:  2021-05-17

7.  Usefulness of 99mTc-ASC lymphoscintigraphy and SPECT/CT in the evaluation of rare lymphatic disorders: Gorham-Stout disease, lymphangioma, and lymphangioleiomyomatosis.

Authors:  Guozhu Hou; Yuanyuan Jiang; Hongli Jing; Wenshuai Xu; Kai-Feng Xu; Libo Chen; Fang Li; Wuying Cheng
Journal:  Medicine (Baltimore)       Date:  2020-09-25       Impact factor: 1.817

  7 in total

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