MedElevator Войти в исследования
← Поиск публикаций

DOI 10.1111/cge.13936

Медицинская библиотека · Clinical genetics · 2021

Accuracy diagnosis improvement of Fabry disease from dried blood spots: Enzyme activity, lyso-Gb3 accumulation and GLA gene sequencing.

Delarosa-Rodríguez Rocío, Santotoribio José D, Paula Hernández-Arévalo, González-Meneses Antonio, García-Morillo Salvador, Jiménez-Arriscado Pilar, Guerrero Juan M, Macher Hada C

СтатьяНаблюдательное исследованиеПеревод пока не подготовлен

Аннотация

The purpose of this study was to examine the applicability of the use of samples in dried blood spot (DBS) for the definitive diagnosis of Fabry disease (FD) in males and females and to compare the diagnostic role of α-galactosidase A activity (α-Gal A), levels of lyso-Gb3 and sequencing of the GLA gene in screening patients with suspected FD. Measurement of α-Gal A activity in suspected FD patients in DBS was made followed by lyso-Gb3 determination and GLA gene sequencing. Of the 2381 subjects analyzed, FD was confirmed in 24 patients. Thirteen different variants were considered like pathogenic, five of which had not been previously described (c.143A > G; c.455A > C; c.487G > T; c.554delA; c.1045_1046insA). None of the patients with normal enzyme activity had FD confirmation. The DBS measurement of α-Gal A was more sensitive than lyso-Gb3 levels in both men and women. Definitive diagnosis of FD from a single DBS is possible, allowing samples to be easily sent from anywhere to the reference laboratory.

Для цитирования: Delarosa-Rodríguez Rocío, Santotoribio José D, Paula Hernández-Arévalo, González-Meneses Antonio, García-Morillo Salvador, Jiménez-Arriscado Pilar, Guerrero Juan M, Macher Hada C. Accuracy diagnosis improvement of Fabry disease from dried blood spots: Enzyme activity, lyso-Gb3 accumulation and GLA gene sequencing.. Clinical genetics. 2021. DOI: 10.1111/cge.13936.

Предметные рубрики MeSH

Подготовка и перевод публикации
Обработка статьи · этапы, ошибки и управление
Загрузка состояния…

История · было → стало

Состояние обновляется каждые 5 секунд, пока блок открыт. Процент показывается только там, где известен объём работы. Пауза останавливает продолжение этапа; текущий запрос к источнику может завершиться.

Тип публикации указан по данным источника. Уровень доказательности автоматически не присваивается.