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一组慢性复发性多灶性骨髓炎(CRMO)患者的诊断与治疗见解

Diagnostic and therapeutic insights from a cohort of chronic recurrent multifocal osteomyelitis (CRMO) patients.

作者信息

Ashari Kosar Asna, Mousavi Mahdieh, Sharabian Fatemeh Tahghighi, Assari Raheleh, Raeeskarami Seyyed Reza, Ziaee Vahid

机构信息

Children's Medical Center, Pediatrics Center of Excellence, Tehran, Iran.

Department of Pediatrics, Tehran University of Medical Sciences, Tehran, Iran.

出版信息

BMC Rheumatol. 2025 Jul 1;9(1):70. doi: 10.1186/s41927-025-00537-4.

Abstract

BACKGROUND

Chronic recurrent multifocal osteomyelitis (CRMO) is a sporadic form of autoinflammatory bone disorders (ABDs) presenting with sterile chronic and/or recurrent and multiple sites of bone involvement. We aimed to describe our 10-year cohort of CRMO patients and analyze the characteristics and treatment approaches.

METHODS

We retrospectively analyzed the data on patients with bone diseases at Children's Medical Center, Tehran University of Medical Sciences, Iran in the previous 10 years. The criteria for inclusion of patients as CNO/CRMO were mono-, oligo- or multifocal inflammatory bone lesions (osteomyelitis, osteitis, osteosclerosis) by imaging techniques; duration of complaints for > 6 weeks; and onset before 18 years of age.

RESULTS

Thirty-nine patients were enrolled. Diagnosis of five patients were found compatible with monogenic ABDs. There were four sites of bone involvement per patient. The most common sites were vertebrae, pelvis, and tibia. Eight patients (23%) had dermatologic manifestations, including three psoriasis cases and one palmar pustulosis. All patients received NSAIDs, and 85% received it as first-line. Treatment was escalated to methotrexate or prednisolone when response was suboptimal. Out of 17 patients primarily treated only with NSAIDs, 47% remitted. In general, 84% of our patients are in remission: 36% without medication and 48% with medication.

CONCLUSION

Our CRMO patients showed an acceptable remission response to the current treatment regimen. Results of bone scintigraphy in suspected CRMO patients should be interpreted cautiously as an adjunct to clinical investigations. Special attention should be paid to extraosseous manifestations in suspected CRMO patients to avoid overlooking monogenic ABDs.

CLINICAL TRIAL NUMBER

Not applicable.

摘要

背景

慢性复发性多灶性骨髓炎(CRMO)是自身炎症性骨病(ABD)的一种散发形式,表现为无菌性慢性和/或复发性多部位骨受累。我们旨在描述我们10年的CRMO患者队列,并分析其特征和治疗方法。

方法

我们回顾性分析了伊朗德黑兰医科大学儿童医学中心过去10年中骨病患者的数据。将患者纳入CNO/CRMO的标准为通过影像学技术发现单灶、少灶或多灶性炎症性骨病变(骨髓炎、骨炎、骨硬化);主诉持续时间>6周;发病年龄在18岁之前。

结果

共纳入39例患者。发现5例患者的诊断与单基因ABD相符。每位患者有4个骨受累部位。最常见的部位是椎骨、骨盆和胫骨。8例患者(23%)有皮肤表现,包括3例银屑病和1例掌跖脓疱病。所有患者均接受非甾体抗炎药治疗,85%将其作为一线治疗药物。当反应欠佳时,治疗升级为甲氨蝶呤或泼尼松龙。在17例仅接受非甾体抗炎药初始治疗的患者中,47%病情缓解。总体而言,我们84%的患者病情缓解:36%无需药物治疗,48%需要药物治疗。

结论

我们的CRMO患者对当前治疗方案显示出可接受的缓解反应。对于疑似CRMO患者,骨闪烁显像结果作为临床检查的辅助手段应谨慎解读。应特别关注疑似CRMO患者的骨外表现,以避免漏诊单基因ABD。

临床试验编号

不适用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c531/12218946/12678ca057cb/41927_2025_537_Fig1_HTML.jpg

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