MedElevator Войти в исследования
← Поиск публикаций

DOI 10.1016/j.nmd.2023.10.001

Медицинская библиотека · Neuromuscular disorders : NMD · 2023

Description of clinical and genetic features of 122 patients included in the Spanish Pompe registry.

Martinez-Marin Rafael Jenaro, Reyes-Leiva David, Nascimento Andrés, Muelas Nuria, Dominguez-González C, Paradas Carmen, Olivé Montse, García-Romero Mar, Pascual-Pascual Samuel Ignacio, Grau Josep Maria, Barba-Romero Miguel Angel, Gomez-Caravaca Maria Teresa, de Las Heras Javier, Casquero Pilar, Mendoza Maria Dolores, de León Juan Carlos, Gutierrez Antonio, Morís Germán, Blanco-Lago Raquel, Ramos-Fransi Alba, Pintós Guillem, García-Antelo Maria José, Rabasa Maria, Morgado Yolanda, Usón Mercedes, Miralles Francisco Javier, Bárcena-Llona Jose Eulalio, Gómez-Belda Ana Belén, Pedraza-Hueso Maria Isabel, Hortelano Miryam, Colomé Antoni, Garcia-Martin Guillermina, Lopez de Munain Adolfo, Jericó Ivonne, Galán-Dávila Lucía, Pardo Julio, Salgueiro-Origlia Giorgina, Alonso-Pérez Jorge, Pla-Junca Francesc, Schiava Marianela, Segovia-Simón Sonia, Díaz-Manera Jordi

СтатьяПеревод пока не подготовлен

Аннотация

Pompe disease is a rare genetic disorder with an estimated prevalence of 1:60.000. The two main phenotypes are Infantile Onset Pompe Disease (IOPD) and Late Onset Pompe Disease (LOPD). There is no published data from Spain regarding the existing number of cases, regional distribution, clinical features or, access and response to the treatment. We created a registry to collect all these data from patients with Pompe in Spain. Here, we report the data of the 122 patients registered including nine IOPD and 113 LOPD patients. There was a high variability in how the diagnosis was obtained and how the follow-up was performed among different centres. Seven IOPD patients were still alive being all treated with enzymatic replacement therapy (ERT) at last visit. Ninety four of the 113 LOPD patients had muscle weakness of which 81 were receiving ERT. We observed a progressive decline in the results of muscle function tests during follow-up. Overall, the Spanish Pompe Registry is a valuable resource for understanding the demographics, patient's journey and clinical characteristics of patients in Spain. Our data supports the development of agreed guidelines to ensure that the care provided to the patients is standardized across the country.

Для цитирования: Martinez-Marin Rafael Jenaro, Reyes-Leiva David, Nascimento Andrés, Muelas Nuria, Dominguez-González C, Paradas Carmen, Olivé Montse, García-Romero Mar, Pascual-Pascual Samuel Ignacio, Grau Josep Maria, Barba-Romero Miguel Angel, Gomez-Caravaca Maria Teresa, de Las Heras Javier, Casquero Pilar, Mendoza Maria Dolores, de León Juan Carlos, Gutierrez Antonio, Morís Germán, Blanco-Lago Raquel, Ramos-Fransi Alba, Pintós Guillem, García-Antelo Maria José, Rabasa Maria, Morgado Yolanda, Usón Mercedes, Miralles Francisco Javier, Bárcena-Llona Jose Eulalio, Gómez-Belda Ana Belén, Pedraza-Hueso Maria Isabel, Hortelano Miryam, Colomé Antoni, Garcia-Martin Guillermina, Lopez de Munain Adolfo, Jericó Ivonne, Galán-Dávila Lucía, Pardo Julio, Salgueiro-Origlia Giorgina, Alonso-Pérez Jorge, Pla-Junca Francesc, Schiava Marianela, Segovia-Simón Sonia, Díaz-Manera Jordi. Description of clinical and genetic features of 122 patients included in the Spanish Pompe registry.. Neuromuscular disorders : NMD. 2023. DOI: 10.1016/j.nmd.2023.10.001.

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.