Gorham-Stout Syndrome of the Shoulder Girdle Successfully Controlled by Antiresorptive Agents: A Report of 2 Cases. Issue 2 (April 2019)

• Record Type: Journal Article
• Authors: Matsumoto, Takumi; Naito, Masashi; Hirose, Jun; Nakada, Izumi; Morikawa, Teppei; Tanaka, Sakae
• Is Part Of: JBJS case connector. Volume 9:Issue 2(2019)
• Publication Date: 2019-04
• Subjects: Orthopedic surgery -- Case studies -- Periodicals; Orthopedic surgery -- Periodicals; Orthopedics; Orthopedic Procedures; Orthopedic surgery; Periodicals; Electronic journals; Case studies; Periodicals; 616.7
• DOI: 10.2106/JBJS.CC.18.00285
• Languages: English
• ISSNs: 2160-3251

Abstract

Abstract : Case: Gorham-Stout disease (GSD) is a rare disorder characterized by massive bone destruction. Consensus is lacking on the effective treatment strategies for GSD. This report presents 2 cases of GSD, a 47-year-old man and a 72-year-old woman, involving the shoulder girdle which were successfully controlled by antiresorptive agents including bisphosphonates and denosumab, the antireceptor activator of nuclear factor-κB ligand antibody. Conclusions: These 2 cases suggest that antiresorptive agents targeting osteoclasts can be efficacious therapeutic options for GSD. This is the first case of GSD we are aware of which showed good response to denosumab treatment.