Skip to main content
SpringerLink
Log in

Gorham–Stout disease affecting both hands: stabilisation during biphosphonate treatment

  • Case Reports
  • Published:
HAND

Abstract

The Gorham–Stout syndrome is a rare condition in which spontaneous, progressive resorption of bone occurs. The aetiology is poorly understood. We report a patient with osteolysis of the metacarpal bones in both hands due to an increased number of stimulated osteoclasts. This suggests that early potent antiresorptive therapy with bisphosphonates may prevent local progressive osteolysis.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4

References

  1. Bode-Lesniewska B, von Hochstetter A, Exner GU, Hodler J. Gorham–Stout disease of the shoulder girdle and cervico-thoracic spine: fatal course in a 65-year-old woman. Skeletal Radiol. 2002;31:724–9. Epub 2002 Sep 21.

    Article  CAS  PubMed  Google Scholar 

  2. Carneiro RS, Steglich V. “Disappearing bone disease” in the hand. J Hand Surg. 1987;12A:629–34.

    Google Scholar 

  3. Chalidis BE, Dimitriou CG. Intercalary bone grafting for the reconstruction of phalangeal osteolysis in disappearing bone disease: case report. J Hand Surg. 2008;33A:1873–7.

    Google Scholar 

  4. Choma ND, Biscotti CV, Bauer TW, Mehta AC, Licata AA. Gorham’s syndrome: a case report and review of the literature. Am J Med. 1987;83(6):1151–6.

    Article  CAS  PubMed  Google Scholar 

  5. Devlin RD, Bone HG, Roodman GD. Interleukin-6: a potential mediator of the massive osteolysis in patient with Gorham–Stout disease. J Clin Endocrinol Metab. 1996;81(5):1893–7.

    Article  CAS  PubMed  Google Scholar 

  6. Dickson GR, Hamilton A, Hayes D, Carr KE, Davis R, Mollan RA. An investigation of vanishing bone disease. Bone. 1990;11:205–10.

    Article  CAS  PubMed  Google Scholar 

  7. Dickson GR, Mollan RAB, Carr KE. Cytochemical localization of alkaline and acid phosphatase in human vanishing bone disease. Histochemistry. 1987;87(6):569–72.

    Article  CAS  PubMed  Google Scholar 

  8. Fontanesi J. Radiation therapy in the treatment of Gorham disease. J Pediatr Hematol Oncol. 2003;25:816–7.

    Article  PubMed  Google Scholar 

  9. Fretz CJ, Jungi WF, Neuweiler J, Haertel M. The malignant degeneration of Gorham Stout disease? Rofo-Fortschr Geb Rontgenstr Neuen Bildgeb Verfahr. 1991;155:579–81.

    Article  CAS  PubMed  Google Scholar 

  10. Fujio K, Kanno R, Suzuki H, Nakamura N, Gotoh M. Chylothorax associated with massive osteolysis (Gorham’s syndrome). Ann Thorac Surg. 2002;73:1956–7.

    Article  Google Scholar 

  11. Gorham LW, Stout AP. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone): its relation to hemangiomatosis. J Bone Jt Surg [Am]. 1955;37A:985–1004.

    Google Scholar 

  12. Gorham LW, Wright AW, Schultz HH, Maxon FC. Disappearing bones: a rare form of massive osteolysis: report of 2 cases, 1 with autopsy findings. Am J Med. 1954;17(5):674–82.

    Article  CAS  PubMed  Google Scholar 

  13. Gratz KW, Prein J, Remagen W. A reconstruction attempt in progressive osteolysis (Gorham disease) of the mandible: a case report [in German]. Schweiz Monatsschr Zahnmed. 1987;97(8):980–4.

    CAS  PubMed  Google Scholar 

  14. Hagberg H, Lamberg K, Astrom G. Alpha-2b interferon and oral clodronate for Gorham’s disease. Lancet. 1997;350(9094):1822–3.

    Article  CAS  PubMed  Google Scholar 

  15. Hammer F, Kenn W, Wesselmann U, Hofbauer LC, Delling G, Allolio B, et al. Gorham–Stout disease—stabilization during bisphosphonate treatment. J Bone Miner Res. 2005;20:350–3. Epub 2004 Nov

    Article  PubMed  Google Scholar 

  16. Dunbar SF, Rosenberg, 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(3):491–7.

  17. Hanly JG, Walsh NM, Bresnihan B. Massive osteolysis in the hand and response to radiotherapy. J Rheumatol. 1985;12(3):580–2.

    CAS  PubMed  Google Scholar 

  18. Hardegger F, Simpson LA, Segmueller G. The syndrome of idiopathic osteolysis: classification, review and case report. J Bone Jt Surg [Br]. 1985;67-B:89–93.

    Google Scholar 

  19. Heffez L, Doku HC, Carter BL, Feeney JE. Perspectives of massive osteolysis: report of a case and review of the literature. Oral Surg Oral Med Oral Pathol. 1983;55(4):331–43.

    Article  CAS  PubMed  Google Scholar 

  20. Hejgaard N, Olsen PR. Massive Gorham osteolysis of the right hemipelvis complicated by chylothorax: report of a case in a 9-year-old boy successfully treated by pleurodesis. J Pediatr Orthop. 1987;7(1):96–9.

    CAS  PubMed  Google Scholar 

  21. Holroyd I, Dillon M, Roberts GJ. Gorham’s disease: a case (including dental presentation) of vanishing bone disease. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2000;89(1):125–9.

    Article  CAS  PubMed  Google Scholar 

  22. Hvos AG. Bone tumors, diagnosis, treatment and prognosis. 2nd ed. Philadelphia: Saunders; 1991.

    Google Scholar 

  23. Jackson JBS. A boneless arm. Boston Med Surg J. 1838;18:368–9.

    Google Scholar 

  24. Jackson JBS. Absorption of the humerus after fracture. Boston Med Surg J. 1872;10:245–7.

    Google Scholar 

  25. Johnson PM, McClure JG. Observations on massive osteolysis: a review of the literature and report of a case. Radiology. 1958;71(1):28–42.

    CAS  PubMed  Google Scholar 

  26. Kulenkampff HA, Richter GM, Hasse WE, Adler CP. Massive osteolysis in the Gorham–Stout syndrome. Int Orthop. 1990;14:361–6.

    Article  CAS  PubMed  Google Scholar 

  27. Lehnhardt M, Steinau HU, Homann HH, Steinstraesser L, Druecke D. Gorham–Stout disease: report of a case affecting the right hand with a follow-up of 24 years. Handchir Mikrochir Plast Chir. 2004;36:249–54.

    Article  CAS  PubMed  Google Scholar 

  28. Listewnik MJ, Marzecki Z, Kordowski J. Disappearing bone disease: massive osteolysis of the ribs treated with radiotherapy: report of a case. Strahlenther Onkol. 1986;162(5):338–41.

    CAS  PubMed  Google Scholar 

  29. Mawk JR, Obukhov SK, Nichols WD, Wynne TD, Odell JM, Urman SM. Successful conservative management of Gorham disease of the skull base and cervical spine. Childs Nerv Syst. 1997;13:622–5.

    Article  CAS  PubMed  Google Scholar 

  30. McNeil KD, Fong KM, Walker QJ, Jessup P, Zimmerman PV. Gorham’s syndrome: a usually fatal cause of pleural effusion treated successfully with radiotherapy. Thorax. 1996;51:1275–6.

    Article  CAS  PubMed  Google Scholar 

  31. Moller G, Priemel M, Amling M, Werner M, Kuhlmey AS, Delling G. The Gorham–Stout syndrome (Gorham’s massive osteolysis): a report of six cases with histopathological findings. J Bone Jt Surg Br. 1999;81(3):501–6.

    Article  CAS  Google Scholar 

  32. Naden BA. When bone disappears. RN. 1995;58(10):26–9.

    CAS  PubMed  Google Scholar 

  33. Picault C, Comtet JJ, Imbert JC, Boyer JM. Surgical repair of extensive idiopathic osteolysis of the pelvic girdle (Jackson–Gorham disease). J Bone Jt Surg Br. 1984;66:148–9.

    Google Scholar 

  34. Tunon JB, Gonzalez FP. Angiomatosis of the metacarpal skeleton. Hand. 1977;9:88–91.

    Article  CAS  PubMed  Google Scholar 

  35. Velez A, Herrera M, Del Rio E, Ruiz-Maldonado R. Gorham’s syndrome. Int J Dermatol. 1993;32(12):884–7.

    Article  CAS  PubMed  Google Scholar 

  36. Vinee P, Tanyu MO, Hauenstein KH, Sigmund G, Stover B, Adler CP. CT and MRI of Gorham syndrome. J Comput Assist Tomogr. 1994;18(6):985–9.

    Article  CAS  PubMed  Google Scholar 

Download references

Conflicts of interest

The author declare that they have no conflict of interest.

Author information

Authors and Affiliations

  1. Hospital de Los Valles, Av. Interoceanica Km. 12 1/2 y Av. Florencia, Cumbaya, PBX 6000 900/2977 900, Quito, Ecuador

    Santiago Silva

Authors
  1. Santiago Silva
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Santiago Silva.

About this article

Cite this article

Silva, S. Gorham–Stout disease affecting both hands: stabilisation during biphosphonate treatment. HAND 6, 85–89 (2011). https://doi.org/10.1007/s11552-010-9292-6

Download citation

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s11552-010-9292-6

Keywords

Search

Navigation