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DOI 10.1007/s00415-013-7154-1

Медицинская библиотека · Journal of neurology · 2013

Mapping the differences in care for 5,000 spinal muscular atrophy patients, a survey of 24 national registries in North America, Australasia and Europe.

Bladen Catherine L, Thompson Rachel, Jackson Jacqueline M, Garland Connie, Wegel Claire, Ambrosini Anna, Pisano Paolo, Walter Maggie C, Schreiber Olivia, Lusakowska Anna, Jedrzejowska Maria, Kostera-Pruszczyk Anna, van der Pol Ludo, Wadman Renske I, Gredal Ole, Karaduman Ayse, Topaloglu Haluk, Yilmaz Oznur, Matyushenko Vitaliy, Rasic Vedrana Milic, Kosac Ana, Karcagi Veronika, Garami Marta, Herczegfalvi Agnes, Monges Soledad, Moresco Angelica, Chertkoff Lilien, Chamova Teodora, Guergueltcheva Velina, Butoianu Niculina, Craiu Dana, Korngut Lawrence, Campbell Craig, Haberlova Jana, Strenkova Jana, Alejandro Moises, Jimenez Alatorre, Ortiz Genaro Gabriel, Enriquez Gracia Viviana Gonzalez, Rodrigues Miriam, Roxburgh Richard, Dawkins Hugh, Youngs Leanne, Lahdetie Jaana, Angelkova Natalija, Saugier-Veber Pascal, Cuisset Jean-Marie, Bloetzer Clemens, Jeannet Pierre-Yves, Klein Andrea, Nascimento Andres, Tizzano Eduardo, Salgado David, Mercuri Eugenio, Sejersen Thomas, Kirschner Jan, Rafferty Karen, Straub Volker, Bushby Kate, Verschuuren Jan, Beroud Christophe, Lochmüller Hanns

СтатьяПоддержка исследования: вне правительства СШАПеревод пока не подготовлен

Аннотация

Spinal muscular atrophy (SMA) is an autosomal recessive genetic disorder characterised by the degeneration of motor neurons and progressive muscle weakness. It is caused by homozygous deletions in the survival motor neuron gene on chromosome 5. SMA shows a wide range of clinical severity, with SMA type I patients often dying before 2 years of age, whereas type III patients experience less severe clinical manifestations and can have a normal life span. Here, we describe the design, setup and utilisation of the TREAT-NMD national SMA patient registries characterised by a small, but fully standardised set of registry items and by genetic confirmation in all patients. We analyse a selection of clinical items from the SMA registries in order to provide a snapshot of the clinical data stratified by SMA subtype, and compare these results with published recommendations on standards of care. Our study included 5,068 SMA patients in 25 countries. A total of 615 patients were ventilated, either invasively (178) or non-invasively (437), 439 received tube feeding and 455 had had scoliosis surgery. Some of these interventions were not available to patients in all countries, but differences were also noted among high-income countries with comparable wealth and health care systems. This study provides the basis for further research, such as quality of life in ventilated SMA patients, and will inform clinical trial planning.

Для цитирования: Bladen Catherine L, Thompson Rachel, Jackson Jacqueline M, Garland Connie, Wegel Claire, Ambrosini Anna, Pisano Paolo, Walter Maggie C, Schreiber Olivia, Lusakowska Anna, Jedrzejowska Maria, Kostera-Pruszczyk Anna, van der Pol Ludo, Wadman Renske I, Gredal Ole, Karaduman Ayse, Topaloglu Haluk, Yilmaz Oznur, Matyushenko Vitaliy, Rasic Vedrana Milic, Kosac Ana, Karcagi Veronika, Garami Marta, Herczegfalvi Agnes, Monges Soledad, Moresco Angelica, Chertkoff Lilien, Chamova Teodora, Guergueltcheva Velina, Butoianu Niculina, Craiu Dana, Korngut Lawrence, Campbell Craig, Haberlova Jana, Strenkova Jana, Alejandro Moises, Jimenez Alatorre, Ortiz Genaro Gabriel, Enriquez Gracia Viviana Gonzalez, Rodrigues Miriam, Roxburgh Richard, Dawkins Hugh, Youngs Leanne, Lahdetie Jaana, Angelkova Natalija, Saugier-Veber Pascal, Cuisset Jean-Marie, Bloetzer Clemens, Jeannet Pierre-Yves, Klein Andrea, Nascimento Andres, Tizzano Eduardo, Salgado David, Mercuri Eugenio, Sejersen Thomas, Kirschner Jan, Rafferty Karen, Straub Volker, Bushby Kate, Verschuuren Jan, Beroud Christophe, Lochmüller Hanns. Mapping the differences in care for 5,000 spinal muscular atrophy patients, a survey of 24 national registries in North America, Australasia and Europe.. Journal of neurology. 2013. DOI: 10.1007/s00415-013-7154-1.

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