Case Report

Gorham Disease of Distal Humerus Successfully Treated by Slight Shortening and Pin and Plate Fixation

Abstract

Background: Gorham's disease (GD) is one of the scarce and idiopathic skeletal diseases which causes osteolysis following the proliferation of blood vessels. Progressive osteolysis GD of distal humerus with articular involvement and pathologic fracture has not been reported and our case is the first report of this disease involving distal humerus and its joints’ surfaces.

Case Report: A 9-year-old boy, case of nonunion of medial condyle of humerus and pathologic fracture of distal humerus after minor trauma, was referred to our clinic and treatment started by casting but due to displacement and nonunion, we decide to operate him. Intraoperative finding was in favour of aneurysmal bone cyst (ABC) near fracture site; therefore, wide resection and fixation by medial tension band wiring (TBW), lateral plating, and fibular allograft application was done (post-operation pathologic result did not show microscopical features of this tumor) and 6 weeks later, he developed stress riser fracture above lateral plate; thus, plate removal was done and severe bone resorption was revealed. Therefore, another specimen was sent for pathology that showed hamartomatous and hemangiomatous lesion of bone. All findings were in favour of GD. He was operated another time and fixed by Persian fixation with small pins and plate, and early plate removal was done for prevention of stress riser fracture.

Conclusion: In cases of GD of distal humerus and pathologic fracture, Persian fixation is a good option for fixation and we suggest early device removal for prevention of stress riser fracture.

1. Patel DV. Gorham's disease or massive osteolysis. Clin Med Res. 2005;3(2):65-74. doi: 10.3121/cmr.3.2.65. [PubMed: 16012123]. [PubMed Central: PMC1183435].
2. Bruch-Gerharz D, Gerharz CD, Stege H, Krutmann J, Pohl M, Koester R, et al. Cutaneous lymphatic malformations in disappearing bone (Gorham-Stout) disease: A novel clue to the pathogenesis of a rare syndrome. J Am Acad Dermatol. 2007;56(2 Suppl):S21-S25. doi: 10.1016/j.jaad.2006.01.063. [PubMed: 17097379].
3. Aizawa T, Sato T, Kokubun S. Gorham disease of the spine: A case report and treatment strategies for this enigmatic bone disease. Tohoku J Exp Med. 2005;205(2):187-96. doi: 10.1620/tjem.205.187. [PubMed: 15673977].
4. Rubel IF, Carrer A, Gilles JJ, Howard R, Cohen G. Progressive Gorham disease of the forearm. Orthopedics. 2008;31(3):284. doi: 10.3928/01477447-20080301-33. [PubMed: 19292225].
5. Campbell J, Almond HG, Johnson R. Massive osteolysis of the humerus with spontaneous recovery. J Bone Joint Surg Br. 1975;57(2):238-40. [PubMed: 1141289].
6. Tsang WM, Tong AC, Chow LT, Ng IO. Massive osteolysis (Gorham disease) of the maxillofacial skeleton: report of 2 cases. J Oral Maxillofac Surg. 2004;62(2):225-30. doi: 10.1016/j.joms.2003.05.008. [PubMed: 14762756].
7. Kamrani RS, Mehrpour SR, Aghamirsalim MR, Sorbi R, Zargar BR, Kaya A. Pin and plate fixation in complex distal humerus fractures: surgical technique and results. Int Orthop. 2012;36(4):839-44. doi: 10.1007/s00264-011-1343-2. [PubMed: 21881883]. [PubMed Central: PMC3311817].
8. Rao P, Kotwal PP, Goel S. Painless destruction of the shoulder joint: A case report. Clin Rheumatol. 2001;20(2):143-6. doi: 10.1007/pl00011194. [PubMed: 11346229].
9. Kai B, Ryan A, Munk PL, Dunlop P. Gorham disease of bone: Three cases and review of radiological features. Clin Radiol. 2006;61(12):1058-64. doi: 10.1016/j.crad.2006.04.014. [PubMed: 17097429].
10. Elluru RG, Balakrishnan K, Padua HM. Lymphatic malformations: diagnosis and management. Semin Pediatr Surg. 2014;23(4):178-85. doi: 10.1053/j.sempedsurg.2014.07.002. [PubMed: 25241095].
11. Ellati R, Attili A, Haddad H, Al-Hussaini M, Shehadeh A. Novel approach of treating Gorham-Stout disease in the humerus-- Case report and review of literature. Eur Rev Med Pharmacol Sci. 2016;20(3):426-32. [PubMed: 26914115].
12. Tavakoli DR, Sharifzadeh A, Bagherian LM, Farhang ZR. A rare case of gorham's disease: Primary ulnar involvement with secondary spread to the radius and elbow. Trauma Mon. 2013;18(1):41-5. doi: 10.5812/traumamon.9905. [PubMed: 24350149]. [PubMed Central: PMC3860655].
Files
IssueVol 8, No 4 (2022) QRcode
SectionCase Report
DOI https://doi.org/10.18502/jost.v8i4.10459
Keywords
Gorham Disease Bone Resorption Osteolysis Humerus
Rights and permissions
Creative Commons License This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
How to Cite
1.
Rahbari K, Zohrabi K, Kamrani RS. Gorham Disease of Distal Humerus Successfully Treated by Slight Shortening and Pin and Plate Fixation. J Orthop Spine Trauma. 2022;8(4):151-3.