Gorham’s disease: treatment with an autologous iliac bone graft and a reverse total shoulder arthroplasty

Abstract

Background Gorham’s disease (GSD) is a rare osteolytic disease with unclear etiology, and no known prevention or effective treatment. Here we report a new surgical treatment for a case of GSD in September 2017. Case presentation We report GSD in a 52-year-old woman. She had disappearance of her humeral head and a defect of the glenoid bone in her left shoulder joint, which were serious obstacles to joint function. We used an autologous iliac bone graft to repair the glenoid bone defect and a reverse total shoulder arthroplasty. After surgery, humeral osteolysis did not continue, and her shoulder function recovered well. Conclusions This case suggests that autologous bone grafting can still be used to treat GSD despite it being an osteolytic disease. The successful treatment suggests that this method could be used for GSD in other bones.

Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report

“…Jose R et al ( 31 ) reported a case of radiotherapy-induced sarcoma in a GSD patient in the left maxilla. Surgical treatment provides the best chance of cure for GSD, especially for patients with massive osteolysis ( 5 , 11 , 32 ). Surgical interventions include resection alone, biological reconstruction using autogenous or allogeneic bone after resection and artificial prosthesis reconstruction after resection ( 5 , 11 , 32 ).…”

“…Surgical treatment provides the best chance of cure for GSD, especially for patients with massive osteolysis ( 5 , 11 , 32 ). Surgical interventions include resection alone, biological reconstruction using autogenous or allogeneic bone after resection and artificial prosthesis reconstruction after resection ( 5 , 11 , 32 ). Regardless of the surgical approach, complete removal of the osteolytic lesion is a prerequisite to avoid recurrence of GSD.…”

“…Preoperative use of bisphosphonates to prevent disease progression can help control lesion size ( 2 , 11 ). In the literature, GSD patients with joint involvement treated with artificial prostheses have achieved good results with no recurrence ( 2 , 32 ). However, there are also risks of loosening, infection, and fractures around the prosthesis.…”

Gorham-Stout-Disease (GSD) of the proximal humerus: A rare cause of shoulder pain.

“…The basic pathogenesis includes inappropriate activation of osteoclasts via stimulation of interleukin-6 and the receptor activator of nuclear factor-kappa B ligand (RANKL) that leads to spontaneous progressive osteolysis. The destructed osseous matrix is replaced later by fibrous tissue [5].The diagnosis of Gorham's disease is based on clinical and radiologic features of bone loss with histological evidence of angiomatous proliferation [6].…”

Gorham Stout disease: 3 additional cases with 2 very rare polyostotic diseases

“…The diagnosis is made by exclusion. Initially, the most frequent symptom is pain in the affected bone, with or without fracture (5)(6)(7). The osteolysis can last for years and progress to bone resorption.…”