MedElevator Войти в исследования
← Поиск публикаций

DOI 10.1016/j.nmd.2021.05.009

Медицинская библиотека · Neuromuscular disorders : NMD · 2021

The phenotypic and genotypic features of a Scottish cohort with McArdle disease.

Gandhi Sacha E, Longman Cheryl, Petty Richard K H, Brennan Kathryn M, Stewart Willie, Kinch Kevin, Töpf Ana, Straub Volker, Quinlivan Rosaline, Farrugia Maria Elena

СтатьяПеревод в очереди

Аннотация

This retrospective study evaluated the phenotypic and genotypic features of 14 patients with McArdle disease attending the West of Scotland adult muscle clinic. Although all patients experienced exercise-induced cramps, exercise intolerance and hyperCKaemia, only 71% (n = 10) experienced the second wind phenomenon, rhabdomyolysis and/or myoglobinuria. We observed a high rate of fixed muscle weakness (50%; n = 7), coronary artery disease (36%; n = 5), and psychological comorbidity (50%; n = 7). Although 79% had symptom onset in the first decade of life, the mean age at presentation and at genetic diagnosis was 43.8 years and 47.7 years, respectively. 93% had at least one copy of the common PYGM pathogenic variant, c.148C > T, p.(Arg50*), with 50% (n = 7) of the cohort being homozygous. Our cohort highlights the phenotypic variability seen in McArdle disease and underscores the potential for late-onset presentations. It emphasises the need for improved awareness and recognition of this condition amongst neurologists, rheumatologists and general physicians. A history of exercise intolerance and second wind phenomenon may not always be volunteered by the patient, underscoring the need to ask specific questions in clinic to extrapolate the relevant symptoms in this patient cohort.

Для цитирования: Gandhi Sacha E, Longman Cheryl, Petty Richard K H, Brennan Kathryn M, Stewart Willie, Kinch Kevin, Töpf Ana, Straub Volker, Quinlivan Rosaline, Farrugia Maria Elena. The phenotypic and genotypic features of a Scottish cohort with McArdle disease.. Neuromuscular disorders : NMD. 2021. DOI: 10.1016/j.nmd.2021.05.009.

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.