Gorham’s Disease that Gave Response to Radiotherapy

Sule Karabulut Gul; Ahmet Fatih Oruc; Duygu Gedik; Hakan Levent Gul; Atınc Aksu; Alpaslan Mayadagli

doi:doi:10.11648/j.jctr.20140205.11

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Gorham’s Disease that Gave Response to Radiotherapy

Sule Karabulut Gul, Ahmet Fatih Oruc, Duygu Gedik, Hakan Levent Gul, Atınc Aksu, Alpaslan Mayadagli

Published in Journal of Cancer Treatment and Research (Volume 2, Issue 5)

Received: 24 August 2014 Accepted: 9 September 2014 Published: 20 September 2014

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Abstract

Background: Gorham’s disease is a disorder that results as destruction and resorption of osseous matrix and is characterized by proliferation of vascular channels. We here reported a rare case of Gorham’s disease that gave response to radiotherapy. Our aim is to remind the clinicians about this rare entity, and to discuss the etiopathology, clinical presentation and treatment options for patients with Gorham’s disease. Case Report: 32 years old male patient had admitted to neurosurgery department for pain, weakness and limitation of motion at the head and neck. Osteolysis was detected at the bodies of 1, 2 and 3. vertebrae. After a number of examinations and excisional biopsy, the patient was diagnosed as Gorham’s disease. Cervical stabilization was made but after 3 years, the patient’s complaints had occurred again and a restabilization was made. 2 years after; with increased complaints, the patient was send to our clinic for consultation and radiotherapy (RT) was applied to the destructive parts of the bones. 2 months after RT, the patients’ complaints had a significant decrease both clinically and radiologically. Conclusion: Gorham’s disease can be seen in both men，women and at any age group, although most cases are diagnosed before the age of 40 years. No familial predisposition has been found. The disease may affect the appendicular or the axial skeleton. The exact nature of the disease process is unknown. The medical treatment for Gorham’s disease includes radiation therapy, anti-osteoclastic medications (bisphosphonates), and alpha-2b interferon. As laboratory studies are usually within normal limits in most cases, diagnosis can often be delayed. By the light of clinical findings with characteristic radiographic and histopathological findings, an early diagnosis can be possible.

Published in	Journal of Cancer Treatment and Research (Volume 2, Issue 5)
DOI	10.11648/j.jctr.20140205.11
Page(s)	45-47
Creative Commons	This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.
Copyright	Copyright © The Author(s), 2014. Published by Science Publishing Group

Keywords

Bone Disease, Neurosurgery, Osteolysis, Radiotherapy

References

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[2]	Gorham LW, Stout AP. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone): its relation to hemangiomatosis. J Bone Joint Surg [Am] 1955;37-A:985-1004.
[3]	Patel DV. Gorham's disease or massive osteolysis. Clin Med Res. 2005 May;3(2):65-74.
[4]	Dickson GR, Hamilton A, Hayes D, Carr KE, Davis R, Mollan RA. An investigation of vanishing bone disease. Bone 1990;11:205–210.
[5]	Devlin RD, Bone HG 3rd, Roodman GD. Interleukin-6: a potential mediator of the massive osteolysis in patients with Gorham-Stout disease. J Clin Endocrinol Metab 1996;81:1893–1897.
[6]	Pazzaglia UE, Andrini L, Bonato M, Leutner M. Pathology of disappearing bone disease: a case report with immunohistochemical study. Int Orthop 1997;21:303–307.
[7]	Spieth ME, Greenspan A, Forrester DM, Ansari AN, Kimura RL, Gleason-Jordan I. Gorham’s disease of the radius: radiographic, scintigraphic, and MRI findings with pathologic correlation. A case report and review of the literature. Skeletal Radiol 1997;26:659–663.
[8]	Halliday DR, Dahlin DC, Pugh DG, Young HH. Massive osteolysis and angiomatosis. Radiology 1964;82:637–644.
[9]	Ceroni D, De Coulon G, Regusci M, Kaelin A. Gorham-Stout disease of costo-vertebral localization: radiographic, scintigraphic, computed tomography, and magnetic resonance imaging findings. Acta Radiol 2004;45:464–468.
[10]	Assoun J, Richardi G, Railhac JJ, Le Guennec P, Caulier M, Dromer C, Sixou L, Fournie B, Mansat M, Durroux D. CT and MRI of massive osteolysis of Gorham. J Comput Assist Tomogr 1994;18:981–984.
[11]	Dominguez R, Washowich TL. Gorham’s disease or vanishing bone disease: plain film, CT, and MRI findings of two cases. Pediatr Radiol 1994;24:316–318.
[12]	Hagberg H, Lamberg K, Astrom G. Alpha-2b interferon and oral clodronate for Gorham’s disease. Lancet 1997;350(9094):1822–1823.
[13]	Fontanesi J. Radiation therapy in the treatment of Gorham disease. J Pediatr Hematol Oncol 2003;25:816–817.
[14]	Lee S, Finn L, Sze RW, Perkins JA, Sie KC. Gorham Stout syndrome (disappearing bone disease): two additional case reports and a review of the literature. Arch Otolaryngol Head Neck Surg 2003;129:1340–1343.
[15]	Handl-Zeller L, Hohenberg G. Radiotherapy of Morbus Gorham-Stout: the biological value of low irradiation dose. Br J Radiol 1990;63(747):206–208.
[16]	Dunbar SF, Rosenberg A, Mankin H, Rosenthal D, Suit HD. Gorham’s massive osteolysis: the role of radiation therapy and a review of the literature. Int J Radiat Oncol Biol Phys 1993;26:491–497.

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APA Style

Sule Karabulut Gul, Ahmet Fatih Oruc, Duygu Gedik, Hakan Levent Gul, Atınc Aksu, et al. (2014). Gorham’s Disease that Gave Response to Radiotherapy. Journal of Cancer Treatment and Research, 2(5), 45-47. https://doi.org/10.11648/j.jctr.20140205.11

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ACS Style

Sule Karabulut Gul; Ahmet Fatih Oruc; Duygu Gedik; Hakan Levent Gul; Atınc Aksu, et al. Gorham’s Disease that Gave Response to Radiotherapy. J. Cancer Treat. Res. 2014, 2(5), 45-47. doi: 10.11648/j.jctr.20140205.11

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AMA Style

Sule Karabulut Gul, Ahmet Fatih Oruc, Duygu Gedik, Hakan Levent Gul, Atınc Aksu, et al. Gorham’s Disease that Gave Response to Radiotherapy. J Cancer Treat Res. 2014;2(5):45-47. doi: 10.11648/j.jctr.20140205.11

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@article{10.11648/j.jctr.20140205.11,
  author = {Sule Karabulut Gul and Ahmet Fatih Oruc and Duygu Gedik and Hakan Levent Gul and Atınc Aksu and Alpaslan Mayadagli},
  title = {Gorham’s Disease that Gave Response to Radiotherapy},
  journal = {Journal of Cancer Treatment and Research},
  volume = {2},
  number = {5},
  pages = {45-47},
  doi = {10.11648/j.jctr.20140205.11},
  url = {https://doi.org/10.11648/j.jctr.20140205.11},
  eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.jctr.20140205.11},
  abstract = {Background: Gorham’s disease is a disorder that results as destruction and resorption of osseous matrix and is characterized by proliferation of vascular channels. We here reported a rare case of Gorham’s disease that gave response to radiotherapy. Our aim is to remind the clinicians about this rare entity, and to discuss the etiopathology, clinical presentation and treatment options for patients with Gorham’s disease. Case Report: 32 years old male patient had admitted to neurosurgery department for pain, weakness and limitation of motion at the head and neck. Osteolysis was detected at the bodies of 1, 2 and 3. vertebrae. After a number of examinations and excisional biopsy, the patient was diagnosed as Gorham’s disease. Cervical stabilization was made but after 3 years, the patient’s complaints had occurred again and a restabilization was made. 2 years after; with increased complaints, the patient was send to our clinic for consultation and radiotherapy (RT) was applied to the destructive parts of the bones. 2 months after RT, the patients’ complaints had a significant decrease both clinically and radiologically. Conclusion: Gorham’s disease can be seen in both men，women and at any age group, although most cases are diagnosed before the age of 40 years. No familial predisposition has been found. The disease may affect the appendicular or the axial skeleton. The exact nature of the disease process is unknown. The medical treatment for Gorham’s disease includes radiation therapy, anti-osteoclastic medications (bisphosphonates), and alpha-2b interferon. As laboratory studies are usually within normal limits in most cases, diagnosis can often be delayed. By the light of clinical findings with characteristic radiographic and histopathological findings, an early diagnosis can be possible.},
 year = {2014}
}

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TY - JOUR
T1 - Gorham’s Disease that Gave Response to Radiotherapy
AU - Sule Karabulut Gul
AU - Ahmet Fatih Oruc
AU - Duygu Gedik
AU - Hakan Levent Gul
AU - Atınc Aksu
AU - Alpaslan Mayadagli
Y1 - 2014/09/20
PY - 2014
N1 - https://doi.org/10.11648/j.jctr.20140205.11
DO - 10.11648/j.jctr.20140205.11
T2 - Journal of Cancer Treatment and Research
JF - Journal of Cancer Treatment and Research
JO - Journal of Cancer Treatment and Research
SP - 45
EP - 47
PB - Science Publishing Group
SN - 2376-7790
UR - https://doi.org/10.11648/j.jctr.20140205.11
AB - Background: Gorham’s disease is a disorder that results as destruction and resorption of osseous matrix and is characterized by proliferation of vascular channels. We here reported a rare case of Gorham’s disease that gave response to radiotherapy. Our aim is to remind the clinicians about this rare entity, and to discuss the etiopathology, clinical presentation and treatment options for patients with Gorham’s disease. Case Report: 32 years old male patient had admitted to neurosurgery department for pain, weakness and limitation of motion at the head and neck. Osteolysis was detected at the bodies of 1, 2 and 3. vertebrae. After a number of examinations and excisional biopsy, the patient was diagnosed as Gorham’s disease. Cervical stabilization was made but after 3 years, the patient’s complaints had occurred again and a restabilization was made. 2 years after; with increased complaints, the patient was send to our clinic for consultation and radiotherapy (RT) was applied to the destructive parts of the bones. 2 months after RT, the patients’ complaints had a significant decrease both clinically and radiologically. Conclusion: Gorham’s disease can be seen in both men，women and at any age group, although most cases are diagnosed before the age of 40 years. No familial predisposition has been found. The disease may affect the appendicular or the axial skeleton. The exact nature of the disease process is unknown. The medical treatment for Gorham’s disease includes radiation therapy, anti-osteoclastic medications (bisphosphonates), and alpha-2b interferon. As laboratory studies are usually within normal limits in most cases, diagnosis can often be delayed. By the light of clinical findings with characteristic radiographic and histopathological findings, an early diagnosis can be possible.
VL - 2
IS - 5
ER -

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Author Information

Sule Karabulut Gul

Kartal Education and Research Hospital, Department of Radiation Oncology, Istanbul, TR
Ahmet Fatih Oruc

Kartal Education and Research Hospital, Department of Radiation Oncology, Istanbul, TR
Duygu Gedik

Kartal Education and Research Hospital, Department of Radiation Oncology, Istanbul, TR
Hakan Levent Gul

Fatih Uninersity, Medical Faculty, Department of Neurology, Istanbul, TR
Atınc Aksu
Alpaslan Mayadagli

Bezmi Alem University, Medical Faculty, Department of Radiation Oncology, Istanbul, TR

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APA Style

Sule Karabulut Gul, Ahmet Fatih Oruc, Duygu Gedik, Hakan Levent Gul, Atınc Aksu, et al. (2014). Gorham’s Disease that Gave Response to Radiotherapy. Journal of Cancer Treatment and Research, 2(5), 45-47. https://doi.org/10.11648/j.jctr.20140205.11

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ACS Style

Sule Karabulut Gul; Ahmet Fatih Oruc; Duygu Gedik; Hakan Levent Gul; Atınc Aksu, et al. Gorham’s Disease that Gave Response to Radiotherapy. J. Cancer Treat. Res. 2014, 2(5), 45-47. doi: 10.11648/j.jctr.20140205.11

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AMA Style

Sule Karabulut Gul, Ahmet Fatih Oruc, Duygu Gedik, Hakan Levent Gul, Atınc Aksu, et al. Gorham’s Disease that Gave Response to Radiotherapy. J Cancer Treat Res. 2014;2(5):45-47. doi: 10.11648/j.jctr.20140205.11

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@article{10.11648/j.jctr.20140205.11,
  author = {Sule Karabulut Gul and Ahmet Fatih Oruc and Duygu Gedik and Hakan Levent Gul and Atınc Aksu and Alpaslan Mayadagli},
  title = {Gorham’s Disease that Gave Response to Radiotherapy},
  journal = {Journal of Cancer Treatment and Research},
  volume = {2},
  number = {5},
  pages = {45-47},
  doi = {10.11648/j.jctr.20140205.11},
  url = {https://doi.org/10.11648/j.jctr.20140205.11},
  eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.jctr.20140205.11},
  abstract = {Background: Gorham’s disease is a disorder that results as destruction and resorption of osseous matrix and is characterized by proliferation of vascular channels. We here reported a rare case of Gorham’s disease that gave response to radiotherapy. Our aim is to remind the clinicians about this rare entity, and to discuss the etiopathology, clinical presentation and treatment options for patients with Gorham’s disease. Case Report: 32 years old male patient had admitted to neurosurgery department for pain, weakness and limitation of motion at the head and neck. Osteolysis was detected at the bodies of 1, 2 and 3. vertebrae. After a number of examinations and excisional biopsy, the patient was diagnosed as Gorham’s disease. Cervical stabilization was made but after 3 years, the patient’s complaints had occurred again and a restabilization was made. 2 years after; with increased complaints, the patient was send to our clinic for consultation and radiotherapy (RT) was applied to the destructive parts of the bones. 2 months after RT, the patients’ complaints had a significant decrease both clinically and radiologically. Conclusion: Gorham’s disease can be seen in both men，women and at any age group, although most cases are diagnosed before the age of 40 years. No familial predisposition has been found. The disease may affect the appendicular or the axial skeleton. The exact nature of the disease process is unknown. The medical treatment for Gorham’s disease includes radiation therapy, anti-osteoclastic medications (bisphosphonates), and alpha-2b interferon. As laboratory studies are usually within normal limits in most cases, diagnosis can often be delayed. By the light of clinical findings with characteristic radiographic and histopathological findings, an early diagnosis can be possible.},
 year = {2014}
}

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