Evolution of bone lesions in adults with chronic nonbacterial osteitis (CNO): A long-term follow-up study

IF 4.6 2区医学 Q1 RHEUMATOLOGY

Seminars in arthritis and rheumatism Pub Date : 2025-02-09 DOI:10.1016/j.semarthrit.2025.152658

Anne T. Leerling , Christophe C.J. Weizenbach , Ana Navas-Cañete , O.M. Dekkers , E.M. Winter

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Abstract

Objectives

Chronic nonbacterial osteitis (CNO) is a rare disease characterised by sterile bone inflammation. Little is known about the evolution of bone lesions, especially for the adult variant of the disease (adult CNO). We therefore aimed to characterize the radiologic course of adult CNO.

Methods

We conducted a cohort study among confirmed adults with CNO, treated at the Dutch national CNO referral centre between 1992 and 2023. Imaging reports from the first-performed radiological scan (baseline) to the last available scan (end of follow-up) were systematically reviewed for lesion location and radiologic features (sclerosis, hyperostosis, erosions, ankylosis). Incidence rates (IRs) for new lesions, progression, and regression of existing lesions were estimated using the Poisson method. Kaplan-Meier curves were used to visualize cumulative incidence, and Poisson regression models assessed associations between patient characteristics and the outcomes.

Results

The study included 182 adult CNO patients with a mean follow-up of 6.1 ± 5.2 years, treated with nonsteroidal anti-inflammatory drugs or cyclooxygenase-inhibitors and/or intravenous bisphosphonates or tumour necrosis factor alpha inhibitors. The most common pattern was sole involvement of the anterior chest wall (84 %). IRs per 100 person-years were 4 (95 % CI 3–5) (new lesions), 7 (6–9) (progression), and 1 (0.3.-1) (regression). Among patients with anterior chest wall involvement only (n = 147), one person developed a lesion outside this area (IR 0.3 (0.06–1)). At 2 years, cumulative incidence of new lesion development and progression were 2 % (0–5) and 7 % (3–10), increasing to 11 % (5–17) and 29 % (20–36) at 5 years, and 36 % (23–48) and 56 % (43–64) at 10 years. No associations were found between clinical characteristics at baseline and these outcomes.

Conclusions

The development of new bone lesions in treated adult CNO patients is typically confined to previously affected regions, primarily the anterior chest wall. Progression of structural changes occurs in the majority of patients after longer follow-up. These findings can be used for prognostic counselling, and suggest that routine whole-body imaging may not be necessary for most patients during follow-up.

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成人慢性非细菌性骨炎（CNO）骨病变的演变：一项长期随访研究

目的慢性非细菌性骨炎（CNO）是一种以无菌性骨炎症为特征的罕见疾病。对骨病变的演变知之甚少，特别是对成人变异的疾病（成人CNO）。因此，我们的目的是表征成人CNO的放射学过程。方法：我们对1992年至2023年间在荷兰国家CNO转诊中心治疗的确诊成人CNO进行了一项队列研究。系统回顾从首次放射扫描（基线）到最后一次可用扫描（随访结束）的影像学报告，以确定病变位置和放射学特征（硬化、骨质增生、糜烂、强直）。使用泊松方法估计新病变、进展和现有病变消退的发病率（IRs）。Kaplan-Meier曲线用于可视化累积发病率，泊松回归模型评估患者特征与结果之间的关联。结果研究纳入182例成年CNO患者，平均随访6.1±5.2年，接受非甾体类抗炎药或环氧化酶抑制剂和/或静脉注射双膦酸盐或肿瘤坏死因子α抑制剂治疗。最常见的模式是单纯累及前胸壁（84%）。每100人年的ir分别为4 (95% CI 3-5)（新病灶）、7(6-9)（进展）和1(0.3 -1)（消退）。在仅累及前胸壁的患者中（n = 147）， 1人在该区域外出现病变（IR 0.3(0.06-1)）。2年时，新病变发生和进展的累积发生率分别为2%（0-5）和7%(3-10)，5年时增加到11%（5 - 17）和29%(20-36)，10年时增加到36%（23-48）和56%（43-64）。在基线时的临床特征和这些结果之间没有发现关联。结论成年CNO患者在治疗后发生的新骨病变通常局限于先前的病变区域，主要是前胸壁。大多数患者在较长时间的随访后发生结构改变的进展。这些发现可用于预后咨询，并提示大多数患者在随访期间可能不需要常规全身成像。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Seminars in arthritis and rheumatism 医学-风湿病学

CiteScore

9.20

自引率

4.00%

发文量

176

审稿时长

46 days

期刊介绍： Seminars in Arthritis and Rheumatism provides access to the highest-quality clinical, therapeutic and translational research about arthritis, rheumatology and musculoskeletal disorders that affect the joints and connective tissue. Each bimonthly issue includes articles giving you the latest diagnostic criteria, consensus statements, systematic reviews and meta-analyses as well as clinical and translational research studies. Read this journal for the latest groundbreaking research and to gain insights from scientists and clinicians on the management and treatment of musculoskeletal and autoimmune rheumatologic diseases. The journal is of interest to rheumatologists, orthopedic surgeons, internal medicine physicians, immunologists and specialists in bone and mineral metabolism.