Incidence of chronic recurrent multifocal osteomyelitis in children and adolescents in the UK and Republic of Ireland.

IF 4.7 2区 医学 Q1 RHEUMATOLOGY
Daphne Theresa Chia, Andoni Paul Toms, Anish Sanghrajka, Athimalaipet V Ramanan, Orla G Killeen, Cristina Ilea, Kamran Mahmood, Sandrine Compeyrot-Lacassagne, Kathryn Bailey, Neil Martin, Kate Armon, Chenqu Suo
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Abstract

Objectives: Chronic recurrent multifocal osteomyelitis (CRMO), also known as chronic non-bacterial osteomyelitis (CNO), is a rare autoinflammatory condition affecting the bones in children and teenagers. The actual incidence of CRMO remains uncertain. The objective of this study was to identify the incidence of CRMO in children and young people under the age of 16 years in the United Kingdom (UK) and Republic of Ireland (ROI). We also aimed to delineate the demographics, clinical presentation, investigations, initial management and healthcare needs for children and adolescents with CRMO.

Methods: We conducted monthly surveys among all paediatric consultants and paediatric orthopaedic surgeons to identify patients newly diagnosed with CRMO between October 2020 and November 2022. A standardized questionnaire was sent to reporting clinicians to collect further information.

Results: Over the surveillance period, 288 patients were reported, among which, 165 confirmed and 20 probable cases were included in the analysis. The highest incidences were among 8-10 year-olds. A two-to-one female-to-male difference in incidence was observed, and male patients were more likely to present with multifocal disease. A negative correlation was observed between reporting clavicular and leg pain. Investigation-wise, 80.0% of patients were reported to have undergone whole-body MRI and 51.1% had bone biopsies. The most common initial treatments were NSAIDs (93.9%) and bisphosphonates (44.8%).

Conclusion: This study estimates an average annual CRMO incidence of 0.65 cases per 100 000 children and adolescents in the UK and ROI. These findings establish a crucial baseline for ongoing research and improvement in the care of individuals with CRMO.

英国和爱尔兰共和国儿童和青少年慢性复发性多灶性骨髓炎的发病率。
目的:慢性复发性多灶性骨髓炎(CRMO)又称慢性非细菌性骨髓炎(CNO),是一种罕见的影响儿童和青少年骨骼的自身炎症。CRMO 的实际发病率仍不确定。本研究旨在确定英国(UK)和爱尔兰共和国(ROI)16 岁以下儿童和青少年中 CRMO 的发病率。我们还希望了解患有 CRMO 的儿童和青少年的人口统计学、临床表现、检查、初始管理和医疗保健需求:我们每月对所有儿科顾问和儿科矫形外科医生进行调查,以确定 2020 年 10 月至 2022 年 11 月期间新诊断出的 CRMO 患者。我们向报告的临床医生发送了一份标准化问卷,以收集更多信息:在监测期间,共报告了288名患者,其中165例确诊病例和20例疑似病例被纳入分析。8-10岁儿童的发病率最高。女性与男性的发病率相差2倍,男性患者更有可能出现多灶性疾病。报告锁骨痛和腿痛之间呈负相关。据报告,80.0%的患者接受过全身核磁共振成像检查,51.1%的患者接受过骨活检。最常见的初始治疗为非甾体抗炎药(93.9%)和双磷酸盐(44.8%):这项研究估计,在英国和爱尔兰共和国,CRMO 的年平均发病率为每 10 万名儿童和青少年中 0.65 例。这些发现为持续研究和改善CRMO患者的护理工作奠定了重要的基础。
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来源期刊
Rheumatology
Rheumatology 医学-风湿病学
CiteScore
9.40
自引率
7.30%
发文量
1091
审稿时长
2 months
期刊介绍: Rheumatology strives to support research and discovery by publishing the highest quality original scientific papers with a focus on basic, clinical and translational research. The journal’s subject areas cover a wide range of paediatric and adult rheumatological conditions from an international perspective. It is an official journal of the British Society for Rheumatology, published by Oxford University Press. Rheumatology publishes original articles, reviews, editorials, guidelines, concise reports, meta-analyses, original case reports, clinical vignettes, letters and matters arising from published material. The journal takes pride in serving the global rheumatology community, with a focus on high societal impact in the form of podcasts, videos and extended social media presence, and utilizing metrics such as Altmetric. Keep up to date by following the journal on Twitter @RheumJnl.
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