北京大学学报(医学版) ›› 2020, Vol. 52 ›› Issue (6): 1140-1145. doi: 10.19723/j.issn.1671-167X.2020.06.026

• 疑难/罕见病例分析 • 上一篇    下一篇

慢性多灶性骨髓炎1例及文献回顾

胡永玮1,刘蕊2,(),罗莉1,()   

  1. 1. 新疆医科大学第一附属医院风湿免疫科,乌鲁木齐 8300542
    2. 北京大学第三医院风湿免疫科,北京 100191
  • 收稿日期:2020-07-31 出版日期:2020-12-18 发布日期:2020-12-13
  • 通讯作者: 刘蕊,罗莉 E-mail:maryllr@163.com;luoli.6@163.com

Chronic multifocal osteomyelitis: A case report and literature review

Yong-wei HU1,Rui LIU2,(),Li LUO1,()   

  1. 1. Department of Rheumatology, the First Affiliated Hospital of Xinjiang Medical University, Urumchi 8300542, China
    2. Department of Rheumatology, Peking University Third Hospital, Beijing 100191, China
  • Received:2020-07-31 Online:2020-12-18 Published:2020-12-13
  • Contact: Rui LIU,Li LUO E-mail:maryllr@163.com;luoli.6@163.com

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摘要:

分析1例慢性多灶性骨髓炎病例的临床表现、血清学及影像学检查、诊断依据、治疗方案、出院后随访评估等,了解慢性复发性多灶性骨髓炎的发病机制、诊断、鉴别诊断、治疗,并进一步了解自身炎症性骨病的特点。本例患者青少年起病,伴有严重皮损,有进行性加重的脊柱关节疼痛,结合临床表现及辅助检查,符合慢性多灶性骨髓炎的诊断。经过抗炎、止痛效果不佳,后换用肿瘤坏死因子α(tumor necrosis factor α,TNF-α)抑制剂后疼痛缓解,炎症指标恢复正常,皮疹及影像学检查明显改善。慢性复发性多灶性骨髓炎属于自身炎症性骨病中多基因疾病的一种,又称慢性非细菌性骨髓炎,是一种罕见的非感染性炎性疾病,可引起多灶性溶骨性病变,以周期性加重和缓解为特征。该病发病率低,发病机制不清楚,可能与促炎及抗炎失衡有关,诊断无特异性指标及统一的诊断标准,常易与代谢性骨病、感染、肿瘤等疾病相混淆,临床表现为骨痛、发热、皮疹、骨折等,实验室检查可见炎症指标明显增高,影像学检查有溶骨性或硬化性改变,MRI在识别骨病变和组织水肿上更有效,而且比骨发射型计算机断层扫描(emission computed tomography,ECT)更准确。治疗用药方面,多数开始使用非甾体类抗炎药,但易复发并出现新发病灶,还可选择其他治疗方案,如糖皮质激素、TNF-α抑制剂及双磷酸盐、甲氨蝶呤等改善病情的抗风湿药物(disease-modifying anti-rheumatic drugs,DMARDs)。对慢性多灶性骨髓炎的早期诊断和治疗可以预防和减少疾病的并发症,改善患者预后。

关键词: 慢性多灶性骨髓炎, 自身炎症性骨病, 肿瘤坏死因子α抑制剂

Abstract:

A case of chronic multifocal osteomyelitis was described in terms of its clinical manifestations, serological and imaging examinations, diagnostic criteria, treatment options, and follow-up evaluation after discharge. The pathogenesis, diagnosis, differential diagnosis and treatment of chronic multifocal osteomyelitis were reviewed, and the characteristics of autoinflammatory osteopathy were reviewed. The patient with onset from youth had developed severe skin lesions, progressive arthralgia and rachialgia. The clinical manifestation and the auxiliary examination of the patient accorded with the diagnosis of chronic multifocal osteomyelitis. After poor anti-inflammatory and analgesic effects, the switch to tumor necrosis factor alpha (TNF-α) inhibitor resulted in pain relief, normalization of inflammation indexes, and significant improvement in rash and imaging examination. Chronic recurrent multifocal osteomyelitis was a kind of autoinflammatory bone disease of multiple genes in disease with low incidence, unknown mechanism and unified diagnostic criteria. It was also known as chronic nonbacterial osteomyelitis, which was a rare, noninfectious inflammatory disorder that caused multifocallytic bone lesions characterized by periodic exacerbations and remissions. The exact pathophysiology or mechanism of the sterile bone inflammation was poorly understood, although chronic nonbacterial osteomyelitis was probably an osteoclast-mediated disease. In addition, an imbalance between pro- and anti-inflammatory cytokines was suspected to play a role. The available data so far pointed to the interplay among genetics, environmental, and immunologic factors as the causes of chronic nonbacterial osteomyelitis. Infectious etiology did not seem to play a crucial role in the pathogenesis of chronic nonbacterial osteomyelitis. It was often confused with metabolic bone disease, infection, tumor and other diseases. Its clinical manifestations were bone pain, fever, rash, fracture and so on. Laboratory examination showed significant increase in inflammatory markers. Radiographic examination revealed osteolytic or sclerosing changes. Magnetic resonance imaging was very useful for identifying bone lesions and tissue edema and was more accurate than bone emission computed tomography (ECT). Most of the patients begin to use non-steroidal anti-inflammatory drugs (NSAIDs) for treatment, but they are prone to relapse and new lesions appear. Other treatment options can be selected, including glucocorticoids, TNF-α inhibitors, bisphosphonates, methotrexate and other disease-modifying anti-rheumatic drugs (DMARDs). Early diagnosis and treatment can prevent and reduce complications and improve prognosis.

Key words: Chronic multifocal osteomyelitis, Inflammatory osteopathy, Tumor necrosis factor-alpha inhibitors

中图分类号: 

  • R681.2

图1

治疗前后骶髂关节MRI"

图2

患者头面部、颈背部痤疮"

图3

骶髂关节CT"

图4

治疗前后脊柱MRI"

图5

骨ECT"

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