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肘部巨大孤立性滑膜骨软骨瘤病致尺神经病变:1例报告并文献复习

Giant solitary synovial osteochondromatosis of the elbow causing ulnar nerve neuropathy: a case report and review of literature.

作者信息

Al-Najjim Munnan, Mustafa Abubakar, Fenton Carl, Morapudi Syam, Waseem Mohammad

机构信息

Department of Orthopaedics, Macclesfield District General Hospital, Victoria road, Macclesfield, Cheshire, SK10 3BL, UK.

出版信息

J Brachial Plex Peripher Nerve Inj. 2013 Jan 25;8(1):1. doi: 10.1186/1749-7221-8-1.

Abstract

INTRODUCTION

Giant or solitary osteochondroma is part of a rare disorder known as synovial osteochondromatosis. It forms part of a spectrum of disease characterized by metaplastic changes within the joint synovium that are eventually extruded as loose bodies. It has been suggested that solitary synovial osteochondroma forms as progression of synovial osteochondromatosis through a process of either coalescence of multiple smaller bodies or the growth of a dominant synovial osteochondroma. Previous studies have shown that it occurs as a late phase of the disease. We report a rare case of giant synovial osteochondromatosis at the elbow causing ulnar nerve neuropathy and mechanical symptoms which has not been previously reported in the literature.

CASE REPORT

We report a case of a 56 year old Western European gentleman who presented with ulnar nerve neuropathy and swelling behind the elbow. The patient underwent MR imaging and subsequent biopsy that demonstrated synovial osteochondromatosis. Initially the patient declined surgery and opted for a watch and wait approach. Five years later he returned with worsening symptoms and underwent successful surgical resection of a giant solitary synovial osteochondroma.

CONCLUSION

The unique outcome in our patient despite the long interval between presentation and surgical treatment resulted in early full resolution of symptoms within a short period. It may suggest an improved prognosis as compared to multiple synovial osteochondromatosis in terms of mechanical and neurological outcomes.

摘要

引言

巨大或孤立性骨软骨瘤是一种罕见疾病——滑膜骨软骨瘤病的一部分。它是一系列疾病的一部分,其特征是关节滑膜内发生化生改变,最终这些改变以游离体的形式挤出。有人提出,孤立性滑膜骨软骨瘤是通过多个较小体的融合或优势滑膜骨软骨瘤的生长过程,由滑膜骨软骨瘤病进展而来。先前的研究表明,它作为该疾病的晚期阶段出现。我们报告了一例罕见的肘部巨大滑膜骨软骨瘤病病例,该病例导致尺神经病变和机械性症状,这在以前的文献中尚未有报道。

病例报告

我们报告了一例56岁的西欧男性患者,他表现为尺神经病变和肘部后方肿胀。患者接受了磁共振成像检查及后续活检,结果显示为滑膜骨软骨瘤病。最初,患者拒绝手术,选择了观察等待的方法。五年后,他因症状加重回来就诊,并成功接受了巨大孤立性滑膜骨软骨瘤的手术切除。

结论

尽管从出现症状到手术治疗间隔时间较长,但我们患者的独特结果导致症状在短时间内早期完全缓解。这可能表明,在机械和神经学结果方面,与多发性滑膜骨软骨瘤病相比,其预后有所改善。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fd4f/3599958/046b2aefac28/1749-7221-8-1-1.jpg

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