MedElevator Войти в исследования
← Поиск публикаций

DOI 10.1038/s41598-022-23666-z

Медицинская библиотека · Scientific reports · 2022

Prediction of the disease course in Friedreich ataxia.

Hohenfeld Christian, Terstiege Ulrich, Dogan Imis, Giunti Paola, Parkinson Michael H, Mariotti Caterina, Nanetti Lorenzo, Fichera Mario, Durr Alexandra, Ewenczyk Claire, Boesch Sylvia, Nachbauer Wolfgang, Klopstock Thomas, Stendel Claudia, Rodríguez de Rivera Garrido Francisco Javier, Schöls Ludger, Hayer Stefanie N, Klockgether Thomas, Giordano Ilaria, Didszun Claire, Rai Myriam, Pandolfo Massimo, Rauhut Holger, Schulz Jörg B, Reetz Kathrin

СтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

We explored whether disease severity of Friedreich ataxia can be predicted using data from clinical examinations. From the database of the European Friedreich Ataxia Consortium for Translational Studies (EFACTS) data from up to five examinations of 602 patients with genetically confirmed FRDA was included. Clinical instruments and important symptoms of FRDA were identified as targets for prediction, while variables such as genetics, age of disease onset and first symptom of the disease were used as predictors. We used modelling techniques including generalised linear models, support-vector-machines and decision trees. The scale for rating and assessment of ataxia (SARA) and the activities of daily living (ADL) could be predicted with predictive errors quantified by root-mean-squared-errors (RMSE) of 6.49 and 5.83, respectively. Also, we were able to achieve reasonable performance for loss of ambulation (ROC-AUC score of 0.83). However, predictions for the SCA functional assessment (SCAFI) and presence of cardiological symptoms were difficult. In conclusion, we demonstrate that some clinical features of FRDA can be predicted with reasonable error; being a first step towards future clinical applications of predictive modelling. In contrast, targets where predictions were difficult raise the question whether there are yet unknown variables driving the clinical phenotype of FRDA.

Для цитирования: Hohenfeld Christian, Terstiege Ulrich, Dogan Imis, Giunti Paola, Parkinson Michael H, Mariotti Caterina, Nanetti Lorenzo, Fichera Mario, Durr Alexandra, Ewenczyk Claire, Boesch Sylvia, Nachbauer Wolfgang, Klopstock Thomas, Stendel Claudia, Rodríguez de Rivera Garrido Francisco Javier, Schöls Ludger, Hayer Stefanie N, Klockgether Thomas, Giordano Ilaria, Didszun Claire, Rai Myriam, Pandolfo Massimo, Rauhut Holger, Schulz Jörg B, Reetz Kathrin. Prediction of the disease course in Friedreich ataxia.. Scientific reports. 2022. DOI: 10.1038/s41598-022-23666-z.

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.