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Case Report Open Access

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
  • View Affiliations / Copyright

    Affiliations: Department of Orthopaedics, Meiji University of Integrative Medicine, Nantan, Kyoto 629‑0301, Japan, Department of Pathology and Applied Neurobiology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto 602‑0841, Japan
    Copyright: © Koto et al. This is an open access article distributed under the terms of Creative Commons Attribution License.
  • Pages: 551-556
    |
    Published online on: October 7, 2019
       https://doi.org/10.3892/mco.2019.1934
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Abstract

Gorham‑Stout disease (GSD) is a rare bone condition that is characterized by the spontaneous, idiopathic and progressive proliferation of blood or lymphatic vessels, which replace the bone and marrow space. The precise etiology and pathophysiology of GSD remain poorly understood. Therefore, there is no consensus on the treatment of this disease. In the current study, a rare case of GSD in the rib and thoracic spine with spinal injury that was treated with radiotherapy, zoledronic acid, vitamin D and propranolol, is reported. A 77‑year‑old man visited the hospital complaining of constipation for 10 days. Within a few days, the patient had recognized complete paralysis of the bilateral lower legs. Radiologically, large osteolytic lesions were confirmed in the 6, 7 and 8th right ribs, the 6 and 7th thoracic vertebrae and in the liver and spleen. The lesions were diagnosed as GSD based on clinical findings, imaging characteristics and needle biopsy results. The patient was treated with zoledronic acid, activated vitamin D, propranolol and radiotherapy to the thoracic vertebrae. However, approximately 5 months after the first treatment, the rapidly increasing hemorrhagic pleural effusion compressed the left lung and caused a mediastinal shift to the right thorax. Unfortunately, the patient succumbed to the disease 7 months after first admission.
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1 

Torg JS, DiGeorge AM, Kirkpatrick JA Jr and Trujillo MM: Hereditary multicentric osteolysis with recessive transmission: A new syndrome. J Pediatr. 75:243–252. 1969. View Article : Google Scholar : PubMed/NCBI

2 

Macpherson RI, Walker RD and Kowall MH: Essential osteolysis with nephropathy. J Can Assoc Radiol. 24:98–103. 1973.PubMed/NCBI

3 

Dellinger MT, Garg N and Olsen BR: Viewpoints on vessels and vanishing bones in Gorham-Stout disease. Bone. 63:47–52. 2014. View Article : Google Scholar : PubMed/NCBI

4 

Jackson JSB: A singular case of absorption of bone (a boneless arm). Boston Med Surg J. 18:368–369. 1838.

5 

Tolis K, Triantafyllopoulos IK, Tournis S and Papaioannou NA: Gorham-Stout disease of the pelvis: Seven years follow up with complete radiological evaluation. J Musculoskelet Neuronal Interact. 16:79–82. 2016.PubMed/NCBI

6 

Vinée P, Tanyü MO, Hauenstein KH, Sigmund G, Stöver B and Adler CP: CT and MRI of Gorham syndrome. J Comput Assist Tomogr. 18:985–989. 1994. View Article : Google Scholar : PubMed/NCBI

7 

Szabo C and Habre W: Gorham syndrome: Anaesthetic management. Anaesthesia. 55:157–159. 2000. View Article : Google Scholar : PubMed/NCBI

8 

García V, Alonso-Claudio G, Gómez-Hernández MT and Chamorro AJ: Sirolimus on Gorham-Stout disease. Case report. Colomb Med (Cali). 47:213–216. 2016.PubMed/NCBI

9 

Hagberg H, Lamberg K and Aström G: Alpha-2b interferon and oral clodronate for Gorham's disease. Lancet. 350:1822–1823. 1997. View Article : Google Scholar : PubMed/NCBI

10 

Avelar RL, Martins VB, Antunes AA, de Oliveira Neto PJ and Andrade ES: Use of zoledronic acid in the treatment of Gorham's disease. Int J Pediatr Otorhinolaryngol. 74:319–322. 2010. View Article : Google Scholar : PubMed/NCBI

11 

Yerganyan VV, Body JJ, De Saint Aubain N and Gebhart M: Gorham-Stout disease of the proximal fibula treated with radiotherapy and zoledronic acid. J Bone Oncol. 4:42–46. 2015. View Article : Google Scholar : PubMed/NCBI

12 

Gorham LW and Stout AP: Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); Its relation to hemangiomatosis. J Bone Joint Surg Am 37-A. 985–1004. 1955. View Article : Google Scholar

13 

Frankel HL, Hancock DO, Hyslop G, Melzak J, Michaelis LS, Ungar GH, Vernon JD and Walsh JJ: The value of postural reduction in the initial management of closed injuries of the spine with paraplegia and tetraplegia. I. Paraplegia. 7:179–192. 1969.PubMed/NCBI

14 

Brunner U, Rückl K, Konrads C, Rudert M and Plumhoff P: Gorham-Stout syndrome of the shoulder. SICOT J. 2:252016. View Article : Google Scholar : PubMed/NCBI

15 

Hagendoorn J, Yock TI, Borel Rinkes IH, Padera TP and Ebb DH: Novel molecular pathways in Gorham disease: Implications for treatment. Pediatr Blood Cancer. 61:401–406. 2014. View Article : Google Scholar : PubMed/NCBI

16 

Hagendoorn J, Padera TP, Yock TI, Nielsen GP, di Tomaso E, Duda DG, Delaney TF, Gaissert HA, Pearce J, Rosenberg AE, et al: Platelet-derived growth factor receptor-beta in Gorham's disease. Nat Clin Pract Oncol. 3:693–697. 2006. View Article : Google Scholar : PubMed/NCBI

17 

Colucci S, Taraboletti G, Primo L, Viale A, Roca C, Valdembri D, Geuna M, Pagano M, Grano M, Pogrel AM, et al: Gorham-Stout syndrome: A monocyte-mediated cytokine propelled disease. J Bone Miner Res. 21:207–218. 2006. View Article : Google Scholar : PubMed/NCBI

18 

Franchi A, Bertoni F, Bacchini P, Mourmouras V and Miracco C: CD105/endoglin expression in Gorham disease of bone. J Clin Pathol. 62:163–167. 2009. View Article : Google Scholar : PubMed/NCBI

19 

Devlin RD, Bone HG III and Roodman GD: Interleukin-6: A potential mediator of the massive osteolysis in patients with Gorham-Stout disease. J Clin Endocrinol Metab. 81:1893–1897. 1996. View Article : Google Scholar : PubMed/NCBI

20 

Rossler J, Saueressig U, Kayser G, von Winterfeld M and Klement GL: Personalized therapy for generalized lymphatic anomaly/Gorham-Stout disease with a combination of sunitinib and taxol. J Pediatr Hematol Oncol. 37:e481–e485. 2015. View Article : Google Scholar : PubMed/NCBI

21 

Patel DV: Gorham's disease or massive osteolysis. Clin Med Res. 3:65–74. 2005. View Article : Google Scholar : PubMed/NCBI

22 

Tateda S, Aizawa T, Hashimoto K, Kanno H, Ohtsu S, Itoi E and Ozawa H: Successful management of Gorham-Stout disease in the cervical spine by combined conservative and surgical treatments: A case report. Tohoku J Exp Med. 241:249–254. 2017. View Article : Google Scholar : PubMed/NCBI

23 

Srivastava SK, Aggarwal RA, Nemade PS and Bhoale SK: Vanishing bone disease of chest wall and spine with kyphoscoliosis and neurological deficit: A case report and review of literature. Indian J Orthop. 51:107–114. 2017. View Article : Google Scholar : PubMed/NCBI

24 

Takahashi A, Ogawa C, Kanazawa T, Watanabe H, Suzuki M, Suzuki N, Tsuchida Y, Morikawa A and Kuwano H: Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: A severe case of Gorham-Stout syndrome. J Pediatr Surg. 40:E47–E50. 2005. View Article : Google Scholar : PubMed/NCBI

25 

Ceroni D, De Coulon G, Regusci M and Kaelin A: Gorham-Stout disease of costo-vertebral localization: Radiographic, scintigraphic, computed tomography, and magnetic resonance imaging findings. Acta Radiol. 45:464–468. 2004. View Article : Google Scholar : PubMed/NCBI

26 

Quraishi NA, Rajagopal TS, Manoharan SR, Elsayed S, Edwards KL and Boszczyk BM: Effect of timing of surgery on neurological outcome and survival in metastatic spinal cord compression. Eur Spine J. 22:1383–1388. 2013. View Article : Google Scholar : PubMed/NCBI

27 

Ozeki M, Fukao T and Kondo N: Propranolol for intractable diffuse lymphangiomatosis. N Engl J Med. 364:1380–1382. 2011. View Article : Google Scholar : PubMed/NCBI

28 

Holroyd I, Dillon M and Roberts GJ: Gorham's disease: A case (including dental presentation) of vanishing bone disease. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 89:125–129. 2000. View Article : Google Scholar : PubMed/NCBI

29 

Koto K, Horie N, Kimura S, Murata H, Sakabe T, Matsui T, Watanabe M, Adachi S, Maekawa T, Fushiki S and Kubo T: Clinically relevant dose of zoledronic acid inhibits spontaneous lung metastasis in a murine osteosarcoma model. Cancer Lett. 274:271–278. 2009. View Article : Google Scholar : PubMed/NCBI

30 

Nir V, Guralnik L, Livnat G, Bar-Yoseph R, Hakim F, Ilivitzki A and Bentur L: Propranolol as a treatment option in Gorham-Stout syndrome: A case report. Pediatr Pulmonol. 49:417–419. 2014. View Article : Google Scholar : PubMed/NCBI

31 

Morimoto N, Ogiwara H, Miyazaki O, Kitamuara M, Nishina S, Nakazawa A, Maekawa T and Morota N: Gorham-Stout syndrome affecting the temporal bone with cerebrospinal fluid leakage. Int J Pediatr Otorhinolaryngol. 77:1596–1600. 2013. View Article : Google Scholar : PubMed/NCBI

32 

Baud J, Lomri A, Graber D and Bikfalvi A: The therapeutic response in Gorham's syndrome to the beta-blocking agent propranolol is correlated to VEGF-A, but not to VEGF-C or FLT1 expression. BMC Res Notes. 8:3332015. View Article : Google Scholar : PubMed/NCBI

33 

Heyd R, Micke O, Surholt C, Berger B, Martini C, Füller J, Schimpke T and Seegenschmiedt MH; German Cooperative Group on Radiotherapy for Benign Diseases (GCG-BD), : Radiation therapy for Gorham-Stout syndrome: Results of a national patterns-of-care study and literature review. Int J Radiat Oncol Biol Phys. 81:e179–e185. 2011. View Article : Google Scholar : PubMed/NCBI

34 

Dunbar SF, Rosenberg A, Mankin H, Rosenthal D and Suit HD: Gorham's massive osteolysis: The role of radiation therapy and a review of the literature. Int J Radiat Oncol Biol Phys. 26:491–497. 1993. View Article : Google Scholar : PubMed/NCBI

35 

Bode-Lesniewska B, von Hochstetter A, Exner GU and Hodler J: Gorham-Stout disease of the shoulder girdle and cervico-thoracic spine: Fatal course in a 65-year-old woman. Skeletal Radiol. 31:724–729. 2002. View Article : Google Scholar : PubMed/NCBI

36 

Rudert M, Gross W and Kirschner P: Gorham-Stout massive ostelysis. A case report. Unfallchirurg. 98:102–104. 1995.(In German). PubMed/NCBI

37 

Flörchinger A, Böttger E, Claass-Böttger F, Georgi M and Harms J: Gorham-Stout syndrome of the spine. Case report and review of the literature. Rofo. 168:68–76. 1998.(In German). View Article : Google Scholar : PubMed/NCBI

38 

Lee S, Finn L, Sze RW, Perkins JA and Sie KC: Gorham Stout syndrome (disappearing bone disease): Two additional case reports and a review of the literature. Arch Otolaryngol Head Neck Surg. 129:1340–1343. 2003. View Article : Google Scholar : PubMed/NCBI

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Copy and paste a formatted citation
Spandidos Publications style
Koto K, Inui K, Itoi M and Itoh K: 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. Mol Clin Oncol 11: 551-556, 2019.
APA
Koto, K., Inui, K., Itoi, M., & Itoh, K. (2019). 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. Molecular and Clinical Oncology, 11, 551-556. https://doi.org/10.3892/mco.2019.1934
MLA
Koto, K., Inui, K., Itoi, M., Itoh, K."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". Molecular and Clinical Oncology 11.6 (2019): 551-556.
Chicago
Koto, K., Inui, K., Itoi, M., Itoh, K."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". Molecular and Clinical Oncology 11, no. 6 (2019): 551-556. https://doi.org/10.3892/mco.2019.1934
Copy and paste a formatted citation
x
Spandidos Publications style
Koto K, Inui K, Itoi M and Itoh K: 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. Mol Clin Oncol 11: 551-556, 2019.
APA
Koto, K., Inui, K., Itoi, M., & Itoh, K. (2019). 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. Molecular and Clinical Oncology, 11, 551-556. https://doi.org/10.3892/mco.2019.1934
MLA
Koto, K., Inui, K., Itoi, M., Itoh, K."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". Molecular and Clinical Oncology 11.6 (2019): 551-556.
Chicago
Koto, K., Inui, K., Itoi, M., Itoh, K."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". Molecular and Clinical Oncology 11, no. 6 (2019): 551-556. https://doi.org/10.3892/mco.2019.1934
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