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一种新策略,即在先天性 N-糖基化障碍(CDG)的诊断中实施质谱法。

A new strategy implementing mass spectrometry in the diagnosis of congenital disorders of N-glycosylation (CDG).

机构信息

Newborn Screening, Biochemistry and Pharmacology Laboratory, Pediatric Neurology Unit and Laboratories, Meyer Children's University Hospital, Florence, Italy.

Division of Metabolism, Department of Pediatric Specialties, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.

出版信息

Clin Chem Lab Med. 2020 Aug 10;59(1):165-171. doi: 10.1515/cclm-2020-0650.

Abstract

OBJECTIVES

Congenital disorders of N-glycosylation (CDG) are a large group of rare metabolic disorders caused by defects in the most common post-translational modification of proteins. CDGs are often difficult to diagnose as they are manifested with non-specific symptoms and signs. Analysis of serum transferrin (TRF) isoforms, as the classical procedure used to identify a CDG patient, enables to predict pathological steps in the N-linked glycosylation process.

METHODS

We devised a new strategy based on liquid chromatography-mass spectrometry (LC-MS) for the analysis of TRF isoforms by combining a simple and fast sample preparation with a specific chromatographic cleanup/separation step followed by mass-spectrometric measurement. Single TRF isoform masses were obtained through reconstruction of multiply charged electrospray data collected by quadrupole-MS technology. Hereby, we report the first analyzed serum samples obtained from 20 CDG patients and 100 controls.

RESULTS

The ratio of desialylated isoforms to total TRF was calculated for patients and controls. CDG-Type I patients showed higher amounts of bi-sialo isoform (range: 6.7-29.6%) compared to controls (<5.5%, mean percentage 3.9%). CDG-Type II pattern showed an increased peak of tri-sialo isoforms. The mean percentage of tri-sialo-TRF was 9.3% (range: 2.9-12.9%) in controls, which was lower than that obtained from two patients with COG5-CDG and MAN1B1-CDG (18.5 and 24.5%). Intraday and between-day imprecisions were less than 9 and 16%, respectively, for bi-sialo- and less than 3 and 6% for tri-sialo-TRF.

CONCLUSIONS

This LC-MS-based approach provides a simple, sensitive and fast analytical tool for characterizing CDG disorders in a routine clinical biochemistry while improving diagnostic accuracy and speeding clinical decision-making.

摘要

目的

先天性 N-糖基化缺陷(CDG)是一大类由蛋白质最常见的翻译后修饰缺陷引起的罕见代谢疾病。由于 CDG 表现出非特异性症状和体征,因此通常难以诊断。分析血清转铁蛋白(TRF)同工型作为识别 CDG 患者的经典方法,可以预测 N 连接糖基化过程中的病理步骤。

方法

我们设计了一种基于液相色谱-质谱(LC-MS)的新策略,通过结合简单快速的样品制备与特定的色谱净化/分离步骤,以及质谱测量,对 TRF 同工型进行分析。通过对四极杆-MS 技术收集的多电荷电喷雾数据进行重建,获得了单个 TRF 同工型的质量。在此,我们报告了从 20 名 CDG 患者和 100 名对照者获得的第一批分析的血清样本。

结果

计算了患者和对照组去唾液酸同工型与总 TRF 的比值。与对照组(<5.5%,平均值 3.9%)相比,CDG-Type I 患者的双唾液酸同工型(范围:6.7-29.6%)含量更高。CDG-Type II 型表现出三唾液酸同工型峰的增加。对照组三唾液酸-TRF 的平均值百分比为 9.3%(范围:2.9-12.9%),低于 COG5-CDG 和 MAN1B1-CDG 两名患者(18.5%和 24.5%)获得的数值。双唾液酸-TRF 的日内和日间精密度分别小于 9%和 16%,三唾液酸-TRF 的精密度分别小于 3%和 6%。

结论

这种基于 LC-MS 的方法为常规临床生物化学中 CDG 疾病的特征提供了一种简单、灵敏和快速的分析工具,提高了诊断准确性并加快了临床决策。

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