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威尔逊病诊断中的挑战与最新进展

Challenges and Recent Advances in Diagnosing Wilson Disease.

作者信息

Aboalam Hani S, Hassan Marwa K, El-Domiaty Nada, Ibrahim Nagat F, Ali Anwar M, Hassan Wesam, Abu El Wafa Esam G, Elsaghier Ashraf, Hetta Helal F, Elbadry Mohamed, El-Kassas Mohamed

机构信息

Tropical Medicine and Gastroenterology Department, Assiut Liver Center, Assiut, Egypt.

Endemic Medicine Department, Faculty of Medicine, Helwan University, Cairo, Egypt.

出版信息

J Clin Exp Hepatol. 2025 Jul-Aug;15(4):102531. doi: 10.1016/j.jceh.2025.102531. Epub 2025 Feb 22.

Abstract

Wilson disease (WD) is a rare autosomal recessive disorder caused by ATP7B gene mutations, leading to pathological copper accumulation that primarily affects the liver, brain, and eyes. Diagnosing WD remains a significant challenge due to its highly variable clinical presentation, which ranges from asymptomatic biochemical abnormalities to acute liver failure and severe neuropsychiatric manifestations. Traditional diagnostic markers, such as serum ceruloplasmin, urinary copper excretion, and liver biopsy, lack sufficient specificity and sensitivity, often leading to delays in diagnosis and misclassification. Additionally, the absence of a single gold-standard test and the overlap with other hepatic and neurological disorders further complicate early detection. Recent advances in diagnostic techniques offer promising solutions to overcome these limitations. Novel biomarkers, including relative exchangeable copper (REC) and ATP7B protein quantification in dried blood spots have demonstrated improved accuracy in distinguishing WD from other conditions. Advanced imaging modalities, such as anterior segment optical coherence tomography (AS-OCT), quantitative susceptibility mapping (QSM), and copper-64 positron emission tomography imaging provide noninvasive tools for detecting early disease-related changes. Furthermore, next-generation sequencing (NGS) enhances genetic screening, facilitating earlier diagnosis, and family screening. A comprehensive approach integrating conventional and emerging diagnostic methodologies is essential for improving early detection and patient outcomes. Greater awareness of the limitations of traditional tests and the incorporation of novel biomarkers and imaging techniques into clinical practice can help refine diagnostic accuracy, reduce delays, and optimize treatment strategies for WD.

摘要

威尔逊病(WD)是一种由ATP7B基因突变引起的罕见常染色体隐性疾病,导致病理性铜蓄积,主要影响肝脏、大脑和眼睛。由于其临床表现高度可变,从无症状的生化异常到急性肝衰竭和严重的神经精神表现,WD的诊断仍然是一项重大挑战。传统的诊断标志物,如血清铜蓝蛋白、尿铜排泄和肝活检,缺乏足够的特异性和敏感性,常常导致诊断延迟和误诊。此外,缺乏单一的金标准检测方法以及与其他肝脏和神经系统疾病的重叠,进一步使早期检测复杂化。诊断技术的最新进展为克服这些局限性提供了有前景的解决方案。新型生物标志物,包括干血斑中的相对可交换铜(REC)和ATP7B蛋白定量,在区分WD与其他疾病方面已显示出更高的准确性。先进的成像方式,如眼前节光学相干断层扫描(AS-OCT)、定量磁化率成像(QSM)和铜-64正电子发射断层扫描成像,为检测早期疾病相关变化提供了非侵入性工具。此外,下一代测序(NGS)增强了基因筛查,有助于早期诊断和家族筛查。整合传统和新兴诊断方法的综合方法对于改善早期检测和患者预后至关重要。提高对传统检测局限性的认识,并将新型生物标志物和成像技术纳入临床实践,有助于提高诊断准确性,减少延迟,并优化WD的治疗策略。

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