MedElevator Войти в исследования
← Поиск публикаций

DOI 10.1097/jcma.0000000000000379

Медицинская библиотека · Journal of the Chinese Medical Association : JCMA · 2020

Cardiac manifestations in patients with classical or cardiac subtype of Fabry disease.

Wang Wei-Ting, Sung Shih-Hsien, Liao Jo-Nan, Hsu Ting-Rong, Niu Dau-Ming, Yu Wen-Chung

СтатьяПеревод пока не подготовлен

Аннотация

BACKGROUND

Fabry disease (FD) is an X-linked lysosomal storage disorder engendered by a deficiency of the enzyme α-galactosidase A, leading to systemic accumulation of glycolipids. Studies have reported that the cardiac subtype of FD has a later onset and minimal extracardiac involvement. However, whether the severity of cardiac involvement differs between the classic and cardiac subtypes of FD remains unclear.

METHODS

We enrolled consecutive patients with classic FD (n = 22; median age [25th-75th percentile], 47.0 [32.75-56.25] years; men, 72.7%) as well as age- and sex-matched patients with a later-onset cardiac subtype of FD who were selected from our cohort of patients with IVS4 919G>A mutation. FD was diagnosed on the basis of clinical symptoms/signs and pedigree screening of index case, plasma α-galactosidase activity, and molecular analysis. Data on clinical manifestations, laboratory findings, and echocardiogram findings were collected before enzyme replacement treatment. Disease severity was evaluated using the Mainz Severity Score Index score.

RESULTS

All female patients demonstrated heterozygous mutations, with five, one, and four of them showing normal α-galactosidase activity, classic FD, and cardiac subtype of FD, respectively. The distributions of left ventricular performance indices and comorbidities, including hypertension, diabetes mellitus, and dyslipidemia, were similar between the two groups. Moreover, MSSI cardiovascular scores did not differ significantly between the groups (classic vs cardiac subtype, 10.0 [2.0-12.5] vs 10.5 [9.0-15.25]; p = 0.277).

CONCLUSION

Cardiac manifestations are similar between patients with classic and cardiac subtype of FD.

Для цитирования: Wang Wei-Ting, Sung Shih-Hsien, Liao Jo-Nan, Hsu Ting-Rong, Niu Dau-Ming, Yu Wen-Chung. Cardiac manifestations in patients with classical or cardiac subtype of Fabry disease.. Journal of the Chinese Medical Association : JCMA. 2020. DOI: 10.1097/jcma.0000000000000379.

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.