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临床上表现为腕管综合征的舟状骨非典型性软骨瘤:一例极其罕见病例报告并文献复习。

Atypical osteochondroma of the hamate that presented clinically as carpal tunnel syndrome: report of an extremely rare case and literature review.

机构信息

Department of Orthopaedic Surgery, Obihiro Kosei Hospital Hand Center|, Obihiro, 080-0024, Japan.

Department of Orthopaedic Surgery, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Sapporo, 060-8638, Japan.

出版信息

BMC Musculoskelet Disord. 2020 Apr 13;21(1):231. doi: 10.1186/s12891-020-03272-8.

Abstract

BACKGROUND

Osteochondroma is a benign tumor that occurs mainly at the metaphysis of long bones and seldom arises from carpal bones. We describe an extremely rare case of osteochondroma of the hamate without a typical cartilaginous cap and with a spiky bony protrusion in an elderly patient.

CASE PRESENTATION

A 78-year-old right-handed female housekeeper had a multilobed osteochondroma of the hamate, which caused carpal tunnel syndrome and irritation of the flexor tendons. Radiological examinations showed a morphological abnormality of the hamate comprising a spiky bony protrusion into the carpal tunnel and a free body proximal to the pisiform. Open carpal tunnel release and resection of the spiky bony protrusion on the hook of the hamate were performed. The flexor digitorum profundus tendons of the ring and little fingers displayed synovitis and partial laceration in the carpal tunnel. Histological examination also showed atypical findings: only a few regions of cartilaginous tissue were seen in the spiky bony protrusion, whereas the free body proximal to the pisiform contained thick cartilaginous tissue such as a cartilaginous cap typical of osteochondroma. We speculated that the bony protrusion to the carpal tunnel had been eroded by mechanical irritation caused by gliding of the flexor tendon and had resulted in the protruding spiky shape with less cartilaginous tissue. The fractured cartilaginous cap had moved into the cavity within the carpal tunnel proximal to the pisiform and had become a large free body.

CONCLUSIONS

Osteochondroma of the carpal bone may take various shapes because the carpal bone is surrounded by neighboring bones and tight ligaments, which can restrict tumor growth. This type of tumor is likely to present with various symptoms because of the close proximity of important structures including nerves, tendons, and joints. The diagnosis of osteochondroma of the carpal bone may be difficult because of its rarity and atypical radiological and histological findings, such as the lack of a round cartilaginous cap. We suggest that surgeons should have a detailed understanding of this condition and should make a definitive diagnosis based on the overall findings.

摘要

背景

骨软骨瘤是一种主要发生在长骨干骺端的良性肿瘤,很少发生在腕骨。我们描述了一例非常罕见的无典型软骨帽的手舟骨骨软骨瘤病例,其特征为骨棘状突起。

病例介绍

一位 78 岁的右利手女管家患有手舟骨多叶骨软骨瘤,导致腕管综合征和屈肌腱激惹。影像学检查显示手舟骨形态异常,包括向腕管内突出的棘状骨性突起和近豌豆骨的游离体。行腕管切开松解术和切除手舟骨钩突的骨棘状突起。环指和小指的屈指深肌腱在腕管内显示滑膜炎和部分撕裂。组织学检查也显示了非典型发现:在骨棘状突起中仅见少数软骨组织区域,而近豌豆骨的游离体则含有厚的软骨组织,如典型的骨软骨瘤的软骨帽。我们推测,向腕管突出的骨性突起是由屈肌腱滑动引起的机械性刺激侵蚀所致,导致突出的棘状形状和较少的软骨组织。骨折的软骨帽已移到手舟骨近侧的腕管腔内,并成为一个大的游离体。

结论

腕骨骨软骨瘤可能呈现各种形状,因为腕骨被相邻的骨骼和紧韧的韧带所环绕,这可以限制肿瘤的生长。由于靠近神经、肌腱和关节等重要结构,这种类型的肿瘤可能会出现各种症状。由于其罕见性和影像学及组织学表现不典型,如缺乏圆形软骨帽,腕骨骨软骨瘤的诊断可能较为困难。我们建议外科医生详细了解这种情况,并根据整体表现做出明确诊断。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9077/7155337/17266f0d51c5/12891_2020_3272_Fig1_HTML.jpg

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