Two cases of Gorham-Stout disease with good response to zoledronic acid treatment

Abstract

SummaryGorham-Stout disease -also called vanishing bone syndrome -is a rare bone disease characterized by a progressive intra-osseous proliferation of non-neoplastic vascular tissue resulting in massive osteolysis. Here, we report two clinical cases of Gorham-Stout disease. Case 1: a 56-year-old woman with 20 years of history of pain and swell in elbows, ankles and wrist. Then she was diagnosed as systemic lupus erythematosus (SLE) with glomerulonephritis type III. After other pathologies were ruled out Gorham-Stout disease was diagnosed. Intravenous zoledronic acid (5 mg) was indicated and after third infusion a progressive improvement of pain, mobility and daily activities were observed. Case 2: a 70-years-old man with a history of pain and limited motion in the left shoulder without X-ray abnormality. Six months later pathological fracture in the left humerus occurred and after ruled out other pathologies Gorham-Stout disease was diagnosed. Intravenous zoledronic acid (5 mg) was indicated and a good response was observed after the first infusion. Nowadays just over 200 cases were reported. Gorham-Stout disease was reported in different bones, at different age presentation and severe physical deformities, disabilities, and life-threatening complications can occur. Two cases of Gorham-Stout disease with good response to zoledronic acid was reported in this article.

Successful treatment of Gorham–Stout syndrome in the spine by vertebroplasty with cement augmentation

“…So far, the treatment of GSS includes the following major categories: medication therapy, radiation therapy, surgery and targeted therapies. [ 6 – 16 , 27 – 30 ]…”

Gorham's disease of the maxilla – A rare case report with literature overview

“…A case of radiation induced sarcoma has been reported in a 53-year-old male patient by Rodriguez-Vazquez JR et al after treatment of GD of the maxilla. Use of various analgesics, chemotherapy, biphosphonate [28] , calcium, fluoride, alpha-2b interferon [29] , vitamin D, hormones, amino acids, adrenal extracts, UV radiation, somatotrophin, and transfusions of placental blood or blood from growing young children have been reported resulting in limited success though cases with spontaneous recovery have also been reported. [30] In our case, the maxillary bone was stabilized after the surgical intervention and was advised Vitamin D and calcium supplements.…”

Gorham–Stout disease: good results of bisphosphonate treatment in 6 of 7 patients

“…Due to disease progression and local symptoms, 6 of our 7 patients underwent bisphosphonate treatment, and 5 of them achieved radiologically stable disease, while treatment was only recently started in the remaining patient. Several case reports have demonstrated the clinical benefit from bisphosphonates (Mignogna et al 2005, Yerganyan et al 2015, Brance 2017, Ramaroli et al 2019. Hagberg et al (1997) reported the first successful treatment of GSD with bisphosphonates.…”