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累及尺骨远端的奇异型骨旁骨软骨瘤样增生(诺拉病变):一例报告

Bizarre parosteal osteochondromatous proliferation (Nora's lesion) affecting the distal end of the ulna: a case report.

作者信息

Matsui Yuichiro, Funakoshi Tadanao, Kobayashi Hideyuki, Mitsuhashi Tomoko, Kamishima Tamotsu, Iwasaki Norimasa

机构信息

Department of Orthopaedic Surgery, Hokkaido University Graduate School of Medicine, Kita-15 Nishi-7, Kita-ku, Sapporo, 060-8638, Japan.

Department of Surgical Pathology, Hokkaido University Hospital, Kita-14 Nishi-5, Kita-ku, Sapporo, 060-8648, Japan.

出版信息

BMC Musculoskelet Disord. 2016 Mar 16;17:130. doi: 10.1186/s12891-016-0981-3.

Abstract

BACKGROUND

Bizarre parosteal osteochondromatous proliferation (BPOP), first described by Nora et al. in 1983 and therefore termed "Nora's lesion", is a rare lesion that occurs in the short bones of the hands and feet and eventually presents as a parosteal mass. Reports of BPOP in the long bones are very rare. A benign disease, BPOP does not become malignant, although a high rate of recurrence following surgical resection is reported. Because of its atypical imaging findings and histopathological appearance, a BPOP might be misdiagnosed as a malignant tumor such as an osteochondroma with malignant transformation, a parosteal osteosarcoma, or a periosteal osteosarcoma.

CASE PRESENTATION

A 58-year-old woman complained of left ulnar wrist pain at the time of her initial presentation. Plain x-rays showed ectopic calcifications in and around the distal radioulnar joint, which supported the diagnosis of subacute arthritis with hydroxyapatite crystal deposition. She was initially given a wrist brace and directed to follow-up, but her persistent pain required the administration of corticosteroid injections into the distal radioulnar joint. Increasing ulnar wrist joint pain and limited forearm pronation and wrist flexion necessitated computed tomography and contrast-enhanced magnetic resonance imaging. BPOP was diagnosed based on the preoperative imaging studies, and a resection of the lesion was performed along with the decortication of the underlying the cortical bone to reduce recurrence rates. The diagnosis of BPOP was confirmed by pathologic examination. Two years after surgery, the patient has no subsequent pain complaints and an improved range of motion.

CONCLUSIONS

BPOP affecting the distal end of the ulna is exceedingly rare. Because BPOP was diagnosed primarily based upon preoperative imaging findings in our patient, decortication of the underlying cortical bone was performed to reduce recurrence rates. Further careful follow-up in these patients is essential, despite the non-recurrence of the lesion.

摘要

背景

怪异型骨旁骨软骨瘤样增生(BPOP)于1983年由诺拉等人首次描述,因此被称为“诺拉病变”,是一种罕见的病变,发生于手足短骨,最终表现为骨旁肿块。长骨中BPOP的报道非常罕见。BPOP是一种良性疾病,不会恶变,尽管有报道称手术切除后复发率较高。由于其不典型的影像学表现和组织病理学外观,BPOP可能被误诊为恶性肿瘤,如伴有恶性转化的骨软骨瘤、骨旁骨肉瘤或骨膜骨肉瘤。

病例介绍

一名58岁女性初诊时主诉左尺侧腕部疼痛。X线平片显示桡尺远侧关节及其周围有异位钙化,支持羟基磷灰石晶体沉积所致亚急性关节炎的诊断。她最初佩戴腕部支具并接受随访,但持续的疼痛需要向桡尺远侧关节注射皮质类固醇。尺侧腕关节疼痛加剧以及前臂旋前和腕关节屈曲受限,需要进行计算机断层扫描和对比增强磁共振成像检查。根据术前影像学检查诊断为BPOP,并对病变进行切除,同时对下方皮质骨进行去皮质处理以降低复发率。病理检查证实了BPOP的诊断。术后两年,患者无后续疼痛主诉,活动范围有所改善。

结论

影响尺骨远端的BPOP极为罕见。由于我们的患者主要基于术前影像学检查结果诊断为BPOP,因此对下方皮质骨进行去皮质处理以降低复发率。尽管病变未复发,但对这些患者进行进一步仔细随访至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/aa5e/4793759/5b9b3f80e8be/12891_2016_981_Fig1_HTML.jpg

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