Bone loss in Gorham's disease: A case study

Chen,Bin; Lv,Xiaofeng; Wu,Jinxiao; Zhang,Xingguang; Jiao,Xiumin; Zhao,Jing; Cheng,Qianpeng; Cui,Can

doi:10.3892/etm.2013.940

Bone loss in Gorham's disease: A case study

Authors:
- Bin Chen
- Xiaofeng Lv
- Jinxiao Wu
- Xingguang Zhang
- Xiumin Jiao
- Jing Zhao
- Qianpeng Cheng
- Can Cui
View Affiliations

Affiliations: Department of Endocrinology and Metabolism, The Military General Hospital of Beijing PLA, Beijing 100700, P.R. China, Department of Endocrinology and Metabolism, The 2nd Affiliated Hospital, Harbin Medical University, Harbin 150080, P.R. China
Published online on: January 31, 2013 https://doi.org/10.3892/etm.2013.940
Pages: 1017-1018

Metrics: Total Views: 0 (Spandidos Publications: | PMC Statistics: )

Metrics: Total PDF Downloads: 0 (Spandidos Publications: | PMC Statistics: )

Cited By (CrossRef): 0 citations Loading Articles...

Abstract

In this study, a rare disease with characteristics of spontaneous osteolysis of the bone is reported. The patient was an eight-year-old male, who was admitted with shortness of breath. The absence of the right clavicle was identified by radiography. However, the change to the right clavicle was not malignant, as indicated by bone scanography. The biopsy of the right cervical rib revealed a number of vascular fibrous tissues and vessels distending and shunting together; however, no cell proliferation was observed. In addition, no acid-fast bacillia or malignant cells were detected in the sample of pleural effusion from the patient. Low hemoglobin (93 g/l) and a slight elevation of alkaline phosphatase levels (133 U/l) were observed; however, the other laboratory examination results were normal. The follow-up investigation and radiotherapy results indicated that the osteolysis of the skull and the other portion of bone had not worsened. Although it has been reported that >15% of patients succumb to this disease, the patient reported in the current study was in a relatively stable condition.

View Figures

View References

1	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. 37:985–1044. 1955.PubMed/NCBI
2	Choma ND, Biscotti CV, Bauer TW, et al: Gorham’s syndrome: a case report and review of the literature. Am J Med. 83:1151–1155. 1987.
3	Jackson J: A boneless arm. Boston Med Surg J. 18:368–369. 1838.
4	Zheng MW, Yang M, Qiu JX, et al: Gorham-Stout syndrome presenting in a 5-year-old girl with a successful bisphosphonate therapeutic effect. Exp Ther Med. 4:449–451. 2012.PubMed/NCBI
5	Al-Jamali J, Glaum R, Kassem A, Voss PJ, Schmelzeisen R and Schön R: Gorham-Stout syndrome of the facial bones: a review of pathogenesis and treatment modalities and report of a case with a rare cutaneous manifestations. Oral Surg Oral Med Oral Pathol Oral Radiol. 114:e23–e29. 2012. View Article : Google Scholar : PubMed/NCBI
6	Hardegger F, Simpson LA and Segmueller G: The syndrome of idiopathic osteolysis. Classification, review and case report. J Bone Joint Surg Br. 67:88–93. 1985.PubMed/NCBI
7	Bruch-Gerharz D, Gerharz CD, Stege H, et al: Cutaneous lymphatic malformations in disappearing bone (Gorham-Stout) disease: A novel clue to the pathogenesis to a rare syndrome. J Am Acad Dermatol. 56:S21–S25. 2007. View Article : Google Scholar : PubMed/NCBI
8	Heffez L, Doku HC, Carter BL, et al: Perspectives of massive osteolysis: report of a case and review of the literature. Oral Surg Oral Med Oral Pathol. 55:331–343. 1983. View Article : Google Scholar : PubMed/NCBI
9	Dickson GR, Mollan RAB and Carr KE: Cytochemical localization of alkaline and acid phosphatase in human vanishing bone disease. Histochemistry. 87:569–572. 1987. View Article : Google Scholar : PubMed/NCBI
10	Picault C, Comtet JJ, Imbert JC, et al: Surgical repair of extensive idiopathic osteolysis of the pelvic girdle (Jackson-Gorham disease). J Bone Jt Surg Br. 66:148–149. 1984.
11	Hejgaard N and 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. 7:96–99. 1987.
12	Fontanesi J: Radiation therapy in the treatment of Gorham disease. J Pediatr Hematol Oncol. 25:816–817. 2003. View Article : Google Scholar : PubMed/NCBI
13	Dunbar SF, Rosenberg A, Mankin H, et al: 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.
14	Hanly JG, Walsh NM and Bresnihan B: Massive osteolysis in the hand and response to radiotherapy. J Rheumatol. 12:580–582. 1985.PubMed/NCBI
15	Hagberg H, Lamberg K and Astrom G: Alpha-2b interferon and oral clodronate for Gorham’s disease. Lancet. 350:1822–1823. 1997.PubMed/NCBI
16	Hammer F, Kenn W, Wesselmann U, et al: Gorham-Stout disease - stabilization during bisphosphonate treatment. J Bone Miner Res. 20:350–353. 2005. View Article : Google Scholar : PubMed/NCBI
17	Silva S: Gorham-Stout disease affecting both hands: stabilization during biphosphonate treatment. Hand (NY). 6:85–89. 2011. View Article : Google Scholar : PubMed/NCBI
18	Bode-Lesniewska B, von Hochstetter A, Exner GU, et al: Gorham-Stout disease of the shoulder girdle and cervico-thoracic spine: fatal course in a 65-year-old woman. Skeletal Radio. 31:724–729. 2002.PubMed/NCBI