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Oral Radiology
Published in: Oral Radiology 4/2020

01-10-2020 | Case Report

Gorham–Stout disease of the mandible: case report and review of literature of a rare type of osteolysis

Authors: Rohan Jagtap, Swati Gupta, Abdullah Lamfon, Axel Ruprecht, Benjamin Schlott, John Hardeman, Deeba Kashtwari

Published in: Oral Radiology | Issue 4/2020

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Abstract

Gorham–Stout disease (GSD) is a rare form of osteolysis, the aetiology and pathogenesis of which remains controversial to this date. Although more than 200 cases of GSD have been reported so far, this disease continues to go undiagnosed in the initial stages owing to its varied clinical presentations and rarity. Through this case report of GSD in a 3-year-old boy, we discuss the slow progression of the disease over a period of 13 years. The literature review is also done with an emphasis on the role of an oral and maxillofacial radiologist in understanding the disease at its incipient stage.
Literature
1.
Foster BL, Ramnitz MS, Gafni RI, Burke AB, Boyce AM, Lee JS, et al. Rare bone diseases and their dental, oral, and craniofacial manifestations. J Dent Res. 2014; 93(7_suppl):7S–19S.
2.
Jackson JB. Absorption of the humerus after fracture. Boston Med Surg J. 1872;10:245–7.
3.
Ricalde P, Ord RA, Sun CC. Vanishing bone disease in a five year old: report of a case and review of the literature. Int J Oral Maxillofac Surg. 2003;32(2):222–6.PubMedCrossRef
4.
Hardegger F, Simpson LA, Segmueller G. The syndrome of idiopathic osteolysis. Classification, review, and case report. J Bone Joint Surg Br. 1985;67(1):88–93.
5.
Maxie G. Jubb, Kennedy & Palmer's pathology of domestic animals-E-book. Oxford: Elsevier Health Sciences; 2015.
6.
Gorham LW, Wright AW, Shultz HH, Maxon FC Jr. Disappearing bones: a rare form of massive osteolysis; report of two cases, one with autopsy findings. Am J Med. 1954;17:674–82.PubMedCrossRef
7.
Gorham LW, Stout AP. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis. J Bone Joint Surg. 1955;37:985–1004.PubMedCrossRef
8.
Naqvi AA, Joshi SS, Bailey E. An unusual case of disappearing bone disease in the mandible and literature review. J Surg Case Rep 2017;2:1–2.
9.
Zhao SF, Wang YX, Yang XD, et al. Gorham disease of the mandible. J Craniofac Surg. 2015;26:e160–e162162.PubMedCrossRef
10.
Kai B, Ryan A, Munk PL, et al. Gorham disease of bone: three cases and review of radiological features. Clin Radiol. 2006;61:1058–64.PubMedCrossRef
11.
Gedam P, Dhamangaonkar AC, Parekh A. A rare case of progressive Gorham’s disease of right shoulder girdle and cervical spine in a child: 10 year follow-up and a review of literature. J Orthopaedic Case Rep. 2015;5(4):30–3.
12.
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.PubMedCrossRef
13.
Dellinger MT, Garg N, Olsen BR. Viewpoints on vessels and vanishing bones in Gorham–Stout disease. Bone. 2014;63:47–52.PubMedCrossRef
14.
Duffy BM, Manon R, Patel RR, Welsh JS. A case of Gorham’s disease with chylothorax treated curatively with radiation therapy. Clin Med Res. 2005;3(2):83–6.PubMedPubMedCentralCrossRef
15.
Zhang S, Wu D, Shi L, Zhang Y, Long K, Fan Y, et al. Gorham disease of the mandible: a report of two cases and a literature review. Oral Surg Oral Med Oral Pathol Oral Radiol. 2019;127(2):e71–e7676.PubMedCrossRef
16.
Jagtap R, Alansari R, Ruprecht A, Kashtwari D. Trichodentoosseous syndrome: a case report and review of literature. BJR Case Rep. 2019;5:20190039.PubMedPubMedCentral
17.
Ceroni D, De Coulon G, Regusci M, Kaelin A. Gorham-stout disease of costo-vertebral localization: radiographic, scintigraphic, computed tomography, and magnetic resonance imaging findings. Acta Radiol. 2004;45(4):464–8.PubMedCrossRef
18.
Johnson PM, McClure JG. Observations on massive osteolysis: a review of the literature and report of a case. Radiology. 1958;71(1):28–422.PubMedCrossRef
19.
Fisher KL, Pogrel MA. Gorham's syndrome (massive osteolysis): a case report. J Oral Maxillofac Surg. 1990;48(11):1222–5.PubMedCrossRef
20.
Auge B, Toussirot E, Kantelip B, Wendling D. From polycystic bone angiomatosis to Gorham's disease. Two case-reports and literature review. Revue du rhumatisme (English ed.). 1998;65(3):201–6.
21.
Mendez AA, Keret D, Robertson W, MacEwen GD. Massive osteolysis of the femur (Gorham's disease): a case report and review of the literature. J Pediatr Orthop. 1989;9(5):604–8.PubMedCrossRef
22.
Khan A, Fareed WM, Tandon P, Zafar MS. Distraction osteogenesis for correction of post ankylosis mandibular deformities. J Biomed Res. 2015;29(4):332.PubMedPubMedCentral
23.
Dunbar SF, Rosenberg A, 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.PubMedCrossRef
24.
Nikolaou VS, Chytas D, Korres D, Efstathopoulos N. Vanishing bone disease (Gorham–Stout syndrome): a review of a rare entity. World J Orthop. 2014;5(5):694.PubMedPubMedCentralCrossRef
25.
Devlin RD, Bone HG 3rd, Roodman GD. Interleukin-6: a potential mediator of the massive osteolysis in patients with Gorham–Stout disease. J Clin Endocrinol Metab. 1996;81(5):1893–7.PubMed
26.
Colucci S, Taraboletti G, Primo L, Viale A, Roca C, Valdembri D, et al. Gorham–Stout syndrome: a monocyte-mediated cytokine propelled disease. J Bone Miner Res. 2006;21(2):207–18.PubMedCrossRef
27.
Ruggieri P, Montalti M, Angelini A, Alberghini M, Mercuri M. Gorham-Stout disease: the experience of the Rizzoli Institute and review of the literature. Skelet Radiol. 2011;40(11):1391–7.CrossRef
Metadata
Title
Gorham–Stout disease of the mandible: case report and review of literature of a rare type of osteolysis
Authors
Rohan Jagtap
Swati Gupta
Abdullah Lamfon
Axel Ruprecht
Benjamin Schlott
John Hardeman
Deeba Kashtwari
Publication date
01-10-2020
Publisher
Springer Singapore
Published in
Oral Radiology / Issue 4/2020
Print ISSN: 0911-6028
Electronic ISSN: 1613-9674
DOI
https://doi.org/10.1007/s11282-019-00417-x

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