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DOI 10.1016/j.jns.2015.10.030

Медицинская библиотека · Journal of the neurological sciences · 2015

Molecular spectrum of the SPAST, ATL1 and REEP1 gene mutations associated with the most common hereditary spastic paraplegias in a group of Polish patients.

Elert-Dobkowska Ewelina, Stepniak Iwona, Krysa Wioletta, Rajkiewicz Marta, Rakowicz Maria, Sobanska Anna, Rudzinska Monika, Wasielewska Anna, Pilch Jacek, Kubalska Jolanta, Lipczynska-Lojkowska Wanda, Kulczycki Jerzy, Kurdziel Katarzyna, Sikorska Agata, Beetz Christian, Zaremba Jacek, Sulek Anna

СтатьяПоддержка исследования: вне правительства СШАПеревод пока не подготовлен

Аннотация

Hereditary spastic paraplegias (HSPs) consist of a heterogeneous group of genetically determined neurodegenerative disorders. Progressive lower extremity weakness and spasticity are the prominent features of HSPs resulting from retrograde axonal degeneration of the corticospinal tracts. Three genetic types, SPG3 (ATL1), SPG4 (SPAST) and SPG31 (REEP1), appear predominantly and may account for up to 50% of autosomal dominant hereditary spastic paraplegias (AD-HSPs). Here, we present the results of genetic testing of the three mentioned SPG genetic types in a group of 216 unrelated Polish patients affected with spastic paraplegia. Molecular evaluation was performed by multiplex ligation-dependent probe amplification (MLPA) and DNA sequencing. Nineteen novel mutations: 13 in SPAST, 4 in ATL1 and 2 in REEP1, were identified among overall 50 different mutations detected in 57 families. Genetic analysis resulted in the identification of molecular defects in 54% of familial and 8.4% of isolated cases. Our research expanded the causative mutations spectrum of the three most common genetic forms of HSPs found in a large cohort of probands originating from the Central Europe.

Для цитирования: Elert-Dobkowska Ewelina, Stepniak Iwona, Krysa Wioletta, Rajkiewicz Marta, Rakowicz Maria, Sobanska Anna, Rudzinska Monika, Wasielewska Anna, Pilch Jacek, Kubalska Jolanta, Lipczynska-Lojkowska Wanda, Kulczycki Jerzy, Kurdziel Katarzyna, Sikorska Agata, Beetz Christian, Zaremba Jacek, Sulek Anna. Molecular spectrum of the SPAST, ATL1 and REEP1 gene mutations associated with the most common hereditary spastic paraplegias in a group of Polish patients.. Journal of the neurological sciences. 2015. DOI: 10.1016/j.jns.2015.10.030.

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