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DOI 10.1016/j.ajhg.2015.11.012

Медицинская библиотека · American journal of human genetics · 2015

Spell Checking Nature: Versatility of CRISPR/Cas9 for Developing Treatments for Inherited Disorders.

Wojtal Daria, Kemaladewi Dwi U, Malam Zeenat, Abdullah Sarah, Wong Tatianna W Y, Hyatt Elzbieta, Baghestani Zahra, Pereira Sergio, Stavropoulos James, Mouly Vincent, Mamchaoui Kamel, Muntoni Francesco, Voit Thomas, Gonorazky Hernan D, Dowling James J, Wilson Michael D, Mendoza-Londono Roberto, Ivakine Evgueni A, Cohn Ronald D

СтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

Clustered regularly interspaced short palindromic repeat (CRISPR) has arisen as a frontrunner for efficient genome engineering. However, the potentially broad therapeutic implications are largely unexplored. Here, to investigate the therapeutic potential of CRISPR/Cas9 in a diverse set of genetic disorders, we establish a pipeline that uses readily obtainable cells from affected individuals. We show that an adapted version of CRISPR/Cas9 increases the amount of utrophin, a known disease modifier in Duchenne muscular dystrophy (DMD). Furthermore, we demonstrate preferential elimination of the dominant-negative FGFR3 c.1138G>A allele in fibroblasts of an individual affected by achondroplasia. Using a previously undescribed approach involving single guide RNA, we successfully removed large genome rearrangement in primary cells of an individual with an X chromosome duplication including MECP2. Moreover, removal of a duplication of DMD exons 18-30 in myotubes of an individual affected by DMD produced full-length dystrophin. Our findings establish the far-reaching therapeutic utility of CRISPR/Cas9, which can be tailored to target numerous inherited disorders.

Для цитирования: Wojtal Daria, Kemaladewi Dwi U, Malam Zeenat, Abdullah Sarah, Wong Tatianna W Y, Hyatt Elzbieta, Baghestani Zahra, Pereira Sergio, Stavropoulos James, Mouly Vincent, Mamchaoui Kamel, Muntoni Francesco, Voit Thomas, Gonorazky Hernan D, Dowling James J, Wilson Michael D, Mendoza-Londono Roberto, Ivakine Evgueni A, Cohn Ronald D. Spell Checking Nature: Versatility of CRISPR/Cas9 for Developing Treatments for Inherited Disorders.. American journal of human genetics. 2015. DOI: 10.1016/j.ajhg.2015.11.012.

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