Abstract
This case report of a 61-year-old woman suffering from Gorham–Stout syndrome shows osteolyses of the left pelvis, proximal femur and lumbar spine. The therapeutic regime has included two courses of percutaneous radiotherapy and also continuous application of bisphosphonates over 17 years. Despite this antiresorptive therapy, elevated urinary excretion of desoxypyridinoline has indicated the persistence of increased bone destruction. The radiological progression following bisphosphonate treatment was only moderate. However, physical disability is reduced, but without soaring handicaps suggesting that long-term bisphophonate therapy is a therapeutical option for this rare syndrome.
Similar content being viewed by others
References
Jackson JBS (1883) A boneless arm. Boston Med Surg J 18:368–369
Gorham WL, Stout AP (1955) Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone): its relation to hemangiomatosis. J Bone Joint Surg 37A:985–10004
Mc Neil KD, Fong KM, Walker QJ, Jessup P, Zimmerman PV (1996) Gorham’s syndrome: a usually fatal course of pleural effusion treated successfully with radiotherapy. Thorax 51:1275–1276
Feigl D, Seidel L, Marmor A (1981) Gorham’s disease of the clavicle with bilateral pleural effusions. Chest 79:242–244
Yoo SY, Goo JM, Im JG (2002) Mediastinal lymphangioma and chylothorax. Thoracic involvement of Gorham’s disease. Korean J Radiol 3:130–132
Hammer F, Kenn W, Wesselmann U, Hofbauer LC, Delling G, Allolio B, Arlt W (2005) Gorham Stout disease—stabilization of the disease during bisphosphonate treatment. J Bone Miner Res 20:350–353
Moller G, Priemel M, Amling M, Werner M, Kuhlmey AS, Delling G (1999) The Gorham–Stout Syndrome (Gorham’s massive osteolysis). A report of six cases with histopathological findings. J Bone Joint Surg Br 81:501–506
Cannon SR (1986) Massive Osteolysis—a review of seven cases. J Bone Joint Surg Br 68:24–28
Schmitt O, Schmitt E, Biehl G (1982) Die essentielle Osteolyse Klinik und Verlauf eines seltenen Krankheitsbildes. Z Orthop 120:160–164
Kulenkampff HA, Richter GM, Haase WE, Adler CP (1990) Massive pelvic osteolysis in the Gorham Stout syndrome. Int Orthop (SICOT) 14:361–366
Kulenkampff HA, Richter GM, Adler C, P Haase WE (1989) Massive Osteolyse (Gorham Stout Syndrom) - Klinik, Diagnostik, Therapie und Prognose. Neuere Ergebnisse in der Osteologie. Springer, Heidelberg, pp 387–397
Hardegger F, Simpson LA, Segmueller G (1985) The syndrome of idiopathic osteolysis. Classification, review, and case report. J Bone Joint Surg Br 67:214–219
Paley MD, Lioyd CJ, Penfold CN (2004) Total mandibular reconstruction for massive osteolysis of the mandible (Gorham–Stout syndrome). Br J Oral Maxillofac Surg 3(2):166–168
Poirer H (1968) Massive osteolysis of the humerus treated by resection and prosthetic replacement. J Bone Joint Surg 50 B:158–160
Woodward HR, Chan DPK, Lee J (1981) Massive osteolysis of the cervical spine. A case report of bone graft failure. Spine 6:545–549
Lee S, Finn L, Sze RW, Perkins JA, Sie KC (2003) Gorham Stout syndrome (disappearing bone disease): two additional case reports and review of the literature. Arch Otolaryngol Head Neck Surg 129:1340–1343
Handl-Zeller L, Hohenberg G (1990) Radiotherapy of Morbus Gorham–Stout: the biological value of low irradiation dose. Br J Radiol 63:206–208
Dunbar SF, Rosenberg A, Mankin H, Rosenthal D, Siut HD (1993) Gorham`s massive osteolysis: the role of radiation therapy and a review of the literature. Int J Radiat Oncol Biol Phys 26:491–497
Hambach R, Pujman J, Mally V (1958) Massive osteolysis due to haemangiomatosis: report of a case of Gorham’s disease with autopsy. Radiology 71:43–47
Hagberg H, Lamberg K, Astrom G (1997) Alpha 2b-interferon and oral clodronate for Gorham’s disease. Lancet 350:1822–1823
Mignogna MD, Fedele S, Russo l Lo, Ciccarelli R (2005) Treatment of Gorham`s disease with zoledronic acid. Oral Oncol 41:747–750
Campbell J, Almond HG, Johnson R (1975) Massive osteolysis of the humerus with spontaneous recovery. J Bone Joint Surg Br 57:238–240
Boyer P, Bourgoeis P, Boyer O, Catonne Y, Saillant G (2005) Massive Gorham–Stout syndrome of the pelvis. Clin Rheumatol 24:551–555
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Lehmann, G., Pfeil, A., Böttcher, J. et al. Benefit of a 17-year long-term bisphosphonate therapy in a patient with Gorham–Stout syndrome. Arch Orthop Trauma Surg 129, 967–972 (2009). https://doi.org/10.1007/s00402-008-0742-3
Received:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00402-008-0742-3