Skip to main content
Erschienen in: Archives of Orthopaedic and Trauma Surgery 7/2009

01.07.2009 | Orthopaedic Outcome Assessment

Benefit of a 17-year long-term bisphosphonate therapy in a patient with Gorham–Stout syndrome

verfasst von: Gabriele Lehmann, Alexander Pfeil, Joachim Böttcher, Werner A. Kaiser, Jürgen Füller, Gert Hein, Gunter Wolf

Erschienen in: Archives of Orthopaedic and Trauma Surgery | Ausgabe 7/2009

Einloggen, um Zugang zu erhalten

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.
Literatur
1.
Zurück zum Zitat Jackson JBS (1883) A boneless arm. Boston Med Surg J 18:368–369 Jackson JBS (1883) A boneless arm. Boston Med Surg J 18:368–369
2.
Zurück zum Zitat 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 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
3.
Zurück zum Zitat 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 51:1275–1276CrossRef 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 51:1275–1276CrossRef
4.
Zurück zum Zitat Feigl D, Seidel L, Marmor A (1981) Gorham’s disease of the clavicle with bilateral pleural effusions. Chest 79:242–244PubMedCrossRef Feigl D, Seidel L, Marmor A (1981) Gorham’s disease of the clavicle with bilateral pleural effusions. Chest 79:242–244PubMedCrossRef
5.
Zurück zum Zitat Yoo SY, Goo JM, Im JG (2002) Mediastinal lymphangioma and chylothorax. Thoracic involvement of Gorham’s disease. Korean J Radiol 3:130–132PubMed Yoo SY, Goo JM, Im JG (2002) Mediastinal lymphangioma and chylothorax. Thoracic involvement of Gorham’s disease. Korean J Radiol 3:130–132PubMed
6.
Zurück zum Zitat 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 20:350–353PubMedCrossRef 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 20:350–353PubMedCrossRef
7.
Zurück zum Zitat 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 81:501–506PubMedCrossRef 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 81:501–506PubMedCrossRef
8.
Zurück zum Zitat Cannon SR (1986) Massive Osteolysis—a review of seven cases. J Bone Joint Surg Br 68:24–28PubMed Cannon SR (1986) Massive Osteolysis—a review of seven cases. J Bone Joint Surg Br 68:24–28PubMed
9.
Zurück zum Zitat Schmitt O, Schmitt E, Biehl G (1982) Die essentielle Osteolyse Klinik und Verlauf eines seltenen Krankheitsbildes. Z Orthop 120:160–164PubMedCrossRef Schmitt O, Schmitt E, Biehl G (1982) Die essentielle Osteolyse Klinik und Verlauf eines seltenen Krankheitsbildes. Z Orthop 120:160–164PubMedCrossRef
10.
Zurück zum Zitat Kulenkampff HA, Richter GM, Haase WE, Adler CP (1990) Massive pelvic osteolysis in the Gorham Stout syndrome. Int Orthop (SICOT) 14:361–366CrossRef Kulenkampff HA, Richter GM, Haase WE, Adler CP (1990) Massive pelvic osteolysis in the Gorham Stout syndrome. Int Orthop (SICOT) 14:361–366CrossRef
11.
Zurück zum Zitat 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 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
12.
Zurück zum Zitat 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 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
13.
Zurück zum Zitat 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 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
14.
Zurück zum Zitat Poirer H (1968) Massive osteolysis of the humerus treated by resection and prosthetic replacement. J Bone Joint Surg 50 B:158–160 Poirer H (1968) Massive osteolysis of the humerus treated by resection and prosthetic replacement. J Bone Joint Surg 50 B:158–160
15.
Zurück zum Zitat Woodward HR, Chan DPK, Lee J (1981) Massive osteolysis of the cervical spine. A case report of bone graft failure. Spine 6:545–549PubMedCrossRef Woodward HR, Chan DPK, Lee J (1981) Massive osteolysis of the cervical spine. A case report of bone graft failure. Spine 6:545–549PubMedCrossRef
16.
Zurück zum Zitat 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 129:1340–1343PubMedCrossRef 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 129:1340–1343PubMedCrossRef
17.
Zurück zum Zitat Handl-Zeller L, Hohenberg G (1990) Radiotherapy of Morbus Gorham–Stout: the biological value of low irradiation dose. Br J Radiol 63:206–208PubMed Handl-Zeller L, Hohenberg G (1990) Radiotherapy of Morbus Gorham–Stout: the biological value of low irradiation dose. Br J Radiol 63:206–208PubMed
18.
Zurück zum Zitat 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 26:491–497PubMed 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 26:491–497PubMed
19.
Zurück zum Zitat Hambach R, Pujman J, Mally V (1958) Massive osteolysis due to haemangiomatosis: report of a case of Gorham’s disease with autopsy. Radiology 71:43–47PubMed Hambach R, Pujman J, Mally V (1958) Massive osteolysis due to haemangiomatosis: report of a case of Gorham’s disease with autopsy. Radiology 71:43–47PubMed
20.
Zurück zum Zitat Hagberg H, Lamberg K, Astrom G (1997) Alpha 2b-interferon and oral clodronate for Gorham’s disease. Lancet 350:1822–1823PubMedCrossRef Hagberg H, Lamberg K, Astrom G (1997) Alpha 2b-interferon and oral clodronate for Gorham’s disease. Lancet 350:1822–1823PubMedCrossRef
21.
Zurück zum Zitat Mignogna MD, Fedele S, Russo l Lo, Ciccarelli R (2005) Treatment of Gorham`s disease with zoledronic acid. Oral Oncol 41:747–750PubMedCrossRef Mignogna MD, Fedele S, Russo l Lo, Ciccarelli R (2005) Treatment of Gorham`s disease with zoledronic acid. Oral Oncol 41:747–750PubMedCrossRef
22.
Zurück zum Zitat Campbell J, Almond HG, Johnson R (1975) Massive osteolysis of the humerus with spontaneous recovery. J Bone Joint Surg Br 57:238–240PubMed Campbell J, Almond HG, Johnson R (1975) Massive osteolysis of the humerus with spontaneous recovery. J Bone Joint Surg Br 57:238–240PubMed
23.
Zurück zum Zitat Boyer P, Bourgoeis P, Boyer O, Catonne Y, Saillant G (2005) Massive Gorham–Stout syndrome of the pelvis. Clin Rheumatol 24:551–555PubMedCrossRef Boyer P, Bourgoeis P, Boyer O, Catonne Y, Saillant G (2005) Massive Gorham–Stout syndrome of the pelvis. Clin Rheumatol 24:551–555PubMedCrossRef
Metadaten
Titel
Benefit of a 17-year long-term bisphosphonate therapy in a patient with Gorham–Stout syndrome
verfasst von
Gabriele Lehmann
Alexander Pfeil
Joachim Böttcher
Werner A. Kaiser
Jürgen Füller
Gert Hein
Gunter Wolf
Publikationsdatum
01.07.2009
Verlag
Springer-Verlag
Erschienen in
Archives of Orthopaedic and Trauma Surgery / Ausgabe 7/2009
Print ISSN: 0936-8051
Elektronische ISSN: 1434-3916
DOI
https://doi.org/10.1007/s00402-008-0742-3

Weitere Artikel der Ausgabe 7/2009

Archives of Orthopaedic and Trauma Surgery 7/2009 Zur Ausgabe

Arthropedia

Grundlagenwissen der Arthroskopie und Gelenkchirurgie. Erweitert durch Fallbeispiele, Videos und Abbildungen. 
» Jetzt entdecken

Update Orthopädie und Unfallchirurgie

Bestellen Sie unseren Fach-Newsletter und bleiben Sie gut informiert.