Novel case of Trevor's disease:Adult onset and later recurrence  

作　　者：Amrit S Khalsa Neil S Kumar Matthew A Chin Richard D Lackman 

机构地区：[1]the Department of Orthopaedic Surgery,Hahnemann University Hospital [2]the Department of Orthopaedic Surgery,Cooper University Hospital

出　　处：《World Journal of Orthopedics》2017年第1期77-81,共5页世界骨科杂志（英文版）

基　　金：Cooper University Hospital (Camden,NJ,United States) for supporting to this work

摘　　要：Dysplasia epiphysealis hemimelica(DEH),or Trevor’s disease,is an osteocartilaginous epiphyseal overgrowth typically occurring in children.The literature reports6 adult cases and none describe recurrence requiring additional procedures.We present a new-onset proximal tibial DEH in an adult recurring approximately 3 years after open excision.A 39-year-old female presented with a history of right knee pain,swelling,and instability.Physical examination revealed a firm proximal tibial mass.Computed tomography(CT)imaging showed an exophytic,lobulated,sclerotic mass involving the anterolateral margin of the lateral tibial plateau.Magnetic resonance imaging was suggestive of an osteochondroma.The patient underwent curettage of the lesion due to its periarticular location.Histology revealed benign and reactive bone and cartilage consistent with periosteal chondroma.Two and a half years later,the patient presented with a firm,palpable mass larger than the initial lesion.CT revealed a lateral tibial plateau sclerotic mass consistent with recurrent intraarticular DEH.A complete excision was performed and histology showed sclerotic bone with overlying cartilage consistent with exostosis.DEH is a rare epiphyseal osteocartilaginous outgrowth frequently occurring in the long bones of children less than 8 years old.DEH resembles an osteochondroma due to its pediatric presentation and similar histologic appearance.Adultonset cases comprise less than 1%of reported cases.Recurrence rate after surgical intervention is unknown.Only 1 such case,occurring in a child,has been described.Clinicians contemplating operative treatment for DEH should note the potential for recurrence and consider complete excision.A follow-up period of several years may be warranted to identify recurrent lesions.Dysplasia epiphysealis hemimelica(DEH), or Trevor's disease, is an osteocartilaginous epiphyseal overgrowth typically occurring in children. The literature reports 6 adult cases and none describe recurrence requiring additional procedures. We present a new-onset proximal tibial DEH in an adult recurring approximately 3 years after open excision. A 39-year-old female presented with a history of right knee pain, swelling, and instability. Physical examination revealed a firm proximal tibial mass. Computed tomography(CT) imaging showed an exophytic, lobulated, sclerotic mass involving the anterolateral margin of the lateral tibial plateau. Magnetic resonance imaging was suggestive of an osteochondroma. The patient underwent curettage of the lesion due to its periarticular location. Histology revealed benign and reactive bone and cartilage consistent with periosteal chondroma. Two and a half years later, the patient presented with a firm, palpable mass larger than the initial lesion. CT revealed a lateral tibial plateau sclerotic mass consistent with recurrent intraarticular DEH. A complete excision was performed and histology showed sclerotic bone with overlying cartilage consistent with exostosis. DEH is a rare epiphyseal osteocartilaginous outgrowth frequently occurring in the long bones of children less than 8 years old. DEH resembles an osteochondroma due to its pediatric presentation and similar histologic appearance. Adultonset cases comprise less than 1% of reported cases. Recurrence rate after surgical intervention is unknown. Only 1 such case, occurring in a child, has been described. Clinicians contemplating operative treatment for DEH should note the potential for recurrence and consider complete excision. A follow-up period of several years may be warranted to identify recurrent lesions.

关 键 词：Trevor’s disease DYSPLASIA epiphysealis hemimelica Adult RECURRENCE PROXIMAL TIBIA EXOSTOSIS 

分 类 号：R681.1[医药卫生—骨科学]