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基于传统磁共振成像的模型用于预测脑室周围白质损伤的婴儿(6至24个月)脑瘫的开发与验证:一项多中心回顾性队列研究

Development and validation of a conventional MRI-based model to predict cerebral palsy in infants (aged 6-24 months) with periventricular white matter injury: a multicentre, retrospective cohort study.

作者信息

Huang Tingting, Zheng Jie, Liu Heng, Jiang Haoxiang, Jin Chao, Li Xianjun, Wu Liang, Zhang Lei, Liu Congcong, Bian Yitong, Wang Miaomiao, Wu Fan, Zhao Xin, Shi Shengli, Wang Fei, Li Mengxuan, Zhu Linlin, Feng Yuying, Zhang Gang, Yang Jian

机构信息

Department of Radiology, The First Affiliated Hospital of Xi'an Jiaotong University, Xi'an, China.

Department of MRI, The First Affiliated Hospital of Henan University of Chinese Medicine, Zhengzhou, China.

出版信息

EClinicalMedicine. 2025 Jul 30;86:103364. doi: 10.1016/j.eclinm.2025.103364. eCollection 2025 Aug.

DOI:10.1016/j.eclinm.2025.103364
PMID:40791895
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12336658/
Abstract

BACKGROUND

Periventricular white matter injury (PVWMI) is the most common form of brain injury and the leading cause of cerebral palsy (CP). Early prediction of CP within the first 2 years of life is crucial for timely and effective intervention. Early CP prediction tools for infants with PVWMI are lacking. This study aimed to develop and validate a conventional Magnetic Resonance Imaging (MRI)-based model to predict CP in infants with PVWMI.

METHODS

In this multicentre retrospective cohort study in China, infants with PVWMI who underwent conventional MRI between 6 and 24 months of corrected age (CA) were included from five hospitals and confirmed to have CP or non-CP by 5 years of age. Between April 2013 and September 2018, a multivariable regression logistic model was developed and internally validated using data from one hospital to identify significant independent MRI features associated with CP, followed by external validation across four other hospitals. A visual nomogram was constructed based on these factors. Predictive performance was evaluated via the area under the receiver operating characteristic curve (AUC), calibration curves, and decision curves. Between October 2018 and January 2021, data from one hospital was included in a multiple readers test cohort (nine radiologists and two paediatric neurologists with varying experience) to assess the diagnostic performance and generalisability of the model. Subgroup analyses were conducted by age and sex.

FINDINGS

Across the two recruitment periods, 383 infants (65% male) with MRI-diagnosed PVWMI were included: 191 infants (122 with CP) in the derivation cohort, 115 (75 with CP) in the external validation cohort, and 77 (46 with CP) in the multiple readers test cohort. Five MRI features were associated with CP: abnormal signals in the posterior limb of the internal capsule (odds ratio [OR] 16.52; 95% confidence interval (CI) 5.78-52.67; < 0.001), corticospinal tract in centrum semiovale (13.01; 3.49-62.30; < 0.001), and cerebral peduncle (5.54; 1.20-32.15; = 0.04), abnormal signals or atrophy in the thalamus (4.76; 1.41-19.32; = 0.02) and lenticular nucleus (4.58; 1.24-21.35; = 0.03). The model yielded an AUC of 0.94 (95% CI 0.91-0.98) in the derivation cohort. Similar AUCs were achieved in the internal (0.96 [0.93-0.99]) and external (0.92 [0.86-0.97]) validation cohorts. In the multiple readers test cohort, the average AUC, average sensitivity, and average specificity of 11 readers were 0.96 (95% CI 0.93-0.99), 0.90 (0.84-0.96), and 0.88 (0.77-0.98), respectively. Subgroup analyses were robust, yielding similar AUCs.

INTERPRETATION

The conventional MRI-based model showed good performance for predicting CP in infants aged 6-24 months with PVWMI and also had good diagnostic performance and generalisability, which may assist in identifying high-risk infants of CP and facilitating timely interventions. Future work with external validation in diverse countries and socioeconomic contexts are needed.

FUNDING

National Natural Science Foundation of China, Key R&D Program of Shanxi Province, National Medical Centre Project of the First Affiliated Hospital of Xi'an Jiaotong University, Henan Provincial Health Commission National Traditional Chinese Medicine Clinical Research Base Scientific Research Special Fund, and Clinical Research Award of the First Affiliated Hospital of Xi'an Jiaotong University.

摘要

背景

脑室周围白质损伤(PVWMI)是最常见的脑损伤形式,也是脑瘫(CP)的主要原因。在生命的头2年内早期预测脑瘫对于及时有效的干预至关重要。目前缺乏针对PVWMI婴儿的早期脑瘫预测工具。本研究旨在开发并验证一种基于传统磁共振成像(MRI)的模型,以预测PVWMI婴儿的脑瘫情况。

方法

在中国这项多中心回顾性队列研究中,纳入了5家医院中在矫正年龄(CA)6至24个月期间接受传统MRI检查的PVWMI婴儿,并在5岁时确诊为脑瘫或非脑瘫。在2013年4月至2018年9月期间,使用一家医院的数据开发了多变量回归逻辑模型并进行内部验证,以识别与脑瘫相关的显著独立MRI特征,随后在其他四家医院进行外部验证。基于这些因素构建了视觉列线图。通过受试者操作特征曲线(AUC)下面积、校准曲线和决策曲线评估预测性能。在2018年10月至2021年1月期间,将一家医院的数据纳入多读者测试队列(9名放射科医生和2名经验各异的儿科神经科医生),以评估该模型的诊断性能和通用性。按年龄和性别进行亚组分析。

结果

在两个招募阶段,共纳入383例经MRI诊断为PVWMI的婴儿(65%为男性):推导队列中有191例婴儿(122例患有脑瘫),外部验证队列中有115例(75例患有脑瘫),多读者测试队列中有77例(46例患有脑瘫)。五个MRI特征与脑瘫相关:内囊后肢信号异常(优势比[OR]16.52;95%置信区间[CI]5.78 - 52.67;<0.001)、半卵圆中心的皮质脊髓束(13.01;3.49 - 62.30;<0.001)和大脑脚(5.54;1.20 - 32.15;=0.04)、丘脑信号异常或萎缩(4.76;1.41 - 19.32;=0.02)以及豆状核(4.58;1.24 - 21.35;=0.03)。该模型在推导队列中的AUC为0.94(95%CI 0.91 - 0.98)。内部(0.96[0.93 - 0.99])和外部(0.92[0.86 - 0.97])验证队列也获得了类似的AUC。在多读者测试队列中,11名读者的平均AUC、平均敏感性和平均特异性分别为0.96(95%CI 0.93 - 0.99)、0.90(0.84 - 0.96)和0.88(0.77 - 0.98)。亚组分析结果稳健,AUC相似。

解读

基于传统MRI的模型在预测6至24个月PVWMI婴儿的脑瘫方面表现良好,并且具有良好的诊断性能和通用性,这可能有助于识别脑瘫高危婴儿并促进及时干预。未来需要在不同国家和社会经济背景下进行外部验证的研究。

资助

中国国家自然科学基金、山西省重点研发计划、西安交通大学第一附属医院国家医学中心项目、河南省卫生健康委员会国家中医临床研究基地科研专项基金以及西安交通大学第一附属医院临床研究奖。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/17c8/12336658/318ddced47c3/gr6.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/17c8/12336658/318ddced47c3/gr6.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/17c8/12336658/faffbcf35fff/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/17c8/12336658/9dcb7a30d5ea/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/17c8/12336658/ced15d5f2863/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/17c8/12336658/cefd64c48ae1/gr4.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/17c8/12336658/318ddced47c3/gr6.jpg

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