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Gorham-Stout Syndrome of the Shoulder Girdle Successfully Controlled by Antiresorptive Agents

A Report of 2 Cases

Matsumoto, Takumi MD, PhD1; Naito, Masashi MD, PhD1; Hirose, Jun MD, PhD1; Nakada, Izumi MD2; Morikawa, Teppei MD, PhD3; Tanaka, Sakae MD, PhD1

doi: 10.2106/JBJS.CC.18.00285
Case Reports

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.

1Department of Orthopaedic Surgery, Faculty of Medicine, The University of Tokyo, Bunkyo-ku, Tokyo, Japan

2Department of Orthopaedic Surgery, Tokyo Metropolitan Bokuto General Hospital, Sumida-ku, Tokyo, Japan

3Department of Pathology, Faculty of Medicine, The University of Tokyo, Bunkyo-ku, Tokyo, Japan

E-mail address for T. Matsumoto:

Investigation performed at The Department of Orthopaedic Surgery, The University of Tokyo, Tokyo, Japan

Disclosure: The Disclosure of Potential Conflicts of Interest forms are provided with the online version of the article (

Copyright © 2019 by The Journal of Bone and Joint Surgery, Incorporated
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