Gorham's disease of the proximal tibia successfully treated with local administration of OK-432, followed by reconstruction with distraction osteogenesis: a case report. Issue 1 (January 2016)

• Record Type: Journal Article
• Authors: Yamagishi, Eiki; Takeda, Akira; Konno, Shinichi; Takeda, Koichiro; Hagino, Seita; Hakozaki, Michiyuki
• Is Part Of: Journal of pediatric orthopedics. Volume 25:Issue 1(2016)
• Publication Date: 2016-01
• Subjects: distraction osteogenesis -- Gorham's disease -- Ilizarov method -- OK-432; Pediatric orthopedics -- Periodicals; 618.927005
• DOI: 10.1097/BPB.0000000000000218
• Languages: English
• ISSNs: 1060-152X

Abstract

Abstract : Gorham's disease (GD) is a rare and intractable disease characterized by marked progression of osteolysis associated with lymphangioma and/or hemangioma. Here, we describe a case of GD of the proximal tibia occurring in a 10-year-old boy. Although we could not correctly diagnose it at first, we finally diagnosed him as having GD. Progression of osteolysis of the tibia stopped 3 months after the local administration of OK-432. Thereafter, the huge bone defect with varus and extension deformity was reconstructed successfully by distraction osteogenesis using the Ilizarov method. The present case suggests that local administration of OK-432, followed by distraction osteogenesis is a treatment option for GD.