Article ID Journal Published Year Pages File Type
5732839 International Journal of Surgery Case Reports 2017 5 Pages PDF
Abstract

•Gorham-Stout disease (GSD) is a very rare idiopathic bone disorder characterised by spontaneous and progressive osteolysis, associated with angiomatous proliferation and soft tissue swelling without new bone formation.•The clinical presentation of GSD includes pain, functional impairment, and swelling, although a few asymptomatic cases have been reported, similar to our case.•We present this rare case of GSD with several localisations to emphasise that any procedure appearing to be simple can go catastrophically wrong and can result in the discovery of something that is unexpected and extremely rare.•We wish to emphasise the extreme rarity of the case, focusing on the large difference between the preoperative panoramic radiograph and whole-body CT and MRI images obtained on the day after the first surgical procedure.

IntroductionGorham-Stout disease (GSD), or vanishing bone disease, is a very rare condition of unknown aetiology. It is characterised by progressive osteolysis and angiomatosis.Case presentationWe report the discovery of this very rare disease following a trivial deciduous tooth extraction in a 14-year-old female. We focus initially on the difference between the preoperative orthopantomography and the whole-body computed tomography and magnetic resonance images obtained post-haemorrhage, and then on the improvement of strategies for the correct diagnosis and treatment of this disease.DiscussionBone loss and the proliferation of vascular structures can occur in a single bone or spread to soft tissue and adjacent bone; areas commonly affected by GSD include the ribs, spine, pelvis, skull, clavicle, and the maxillofacial area. The clinical presentation of GSD includes pain, functional impairment, and swelling, although a few asymptomatic cases have been reported, similar to our case.ConclusionWe report a very rare case of this multicentric disease in an asymptomatic child who presented for dental extraction, almost died, and was then diagnosed with and treated for GSD.

Related Topics
Health Sciences Medicine and Dentistry Surgery
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