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糖原贮积病:分子检测后临床诊断的专家观点再评价。

Glycogen Storage Disease: Expert Opinion on Clinical Diagnosis Revisited after Molecular Testing.

机构信息

Genética Médica e Medicina Genômica, Departamento de Medicina Translacional, Faculdade de Ciências Médicas, Universidade Estadual de Campinas (Unicamp), Campinas 13083-970, SP, Brazil.

Departamento de Genética, Universidade Federal do Rio Grande do Sul (UFRGS), Porto Alegre 90010-150, RS, Brazil.

出版信息

Genes (Basel). 2023 Dec 15;14(12):2219. doi: 10.3390/genes14122219.

Abstract

This study sought to analyze whether an accurate diagnosis of the type and subtype of hepatic Glycogen Storage Diseases (GSDs) could be performed based on general clinical and biochemical aspects via comparing the proposed diagnostic hypotheses with the molecular results. Twelve physicians with experience in hepatic GSDs reviewed 45 real cases comprising a standardized summary of clinical and laboratory data. There was no relation between the hit rate and the time since graduation, the time of experience in GSD, and the number of patients treated during their careers. The average assertiveness was 47%, with GSD Ia and Ib being the best-identified types, while no expert correctly identified GSD IXc. Underage investigation for later manifestations, incomplete clinical description, and complementary analysis, the overvaluation of a specific clinical finding ("false positive") or the discarding of the diagnosis in the absence of it ("false negative"), as well as the lack of knowledge of the rarest GSD types, may have impacted the accuracy of the assessment. This study emphasized that characteristics considered as determinants in identifying the specific types or subtypes of GSD are not exclusive, thus becoming factors that may have induced the evaluators to misdiagnose.

摘要

本研究旨在通过将提出的诊断假设与分子结果进行比较,分析是否可以基于一般临床和生化方面准确诊断肝糖原贮积病(GSDs)的类型和亚型。12 名具有肝 GSD 经验的医生回顾了 45 个真实病例,这些病例包含了临床和实验室数据的标准化摘要。命中率与毕业时间、GSD 经验时间以及职业生涯中治疗的患者数量之间没有关系。平均断言率为 47%,其中 GSD Ia 和 Ib 是最容易识别的类型,而没有专家能够正确识别 GSD IXc。对后期表现的未成年调查、不完全的临床描述和补充分析、对特定临床发现的高估(“假阳性”)或在缺乏该发现时放弃诊断(“假阴性”),以及对最罕见的 GSD 类型缺乏了解,可能会影响评估的准确性。本研究强调,被认为是确定 GSD 具体类型或亚型的决定因素的特征并非排他性的,因此成为可能导致评估者误诊的因素。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2b1f/10743078/d260902c6ed5/genes-14-02219-g001.jpg

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