Benefit of a 17-year long-term bisphosphonate therapy in a patient with Gorham-Stout syndrome.

This case report of a 61-year-old woman suffering from Gorham-Stout syndrome shows osteolyses of the left pelvis, proximal femur and lumbar spine. The therapeutic regime has included two courses of percutaneous radiotherapy and also continuous application of bisphosphonates over 17 years. Despite this antiresorptive therapy, elevated urinary excretion of desoxypyridinoline has indicated the persistence of increased bone destruction. The radiological progression following bisphosphonate treatment was only moderate. However, physical disability is reduced, but without soaring handicaps suggesting that long-term bisphophonate therapy is a therapeutical option for this rare syndrome.