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DOI 10.1016/j.ajhg.2019.02.003

Медицинская библиотека · American journal of human genetics · 2019

Systemic mRNA Therapy for the Treatment of Fabry Disease: Preclinical Studies in Wild-Type Mice, Fabry Mouse Model, and Wild-Type Non-human Primates.

Zhu Xuling, Yin Ling, Theisen Matt, Zhuo Jenny, Siddiqui Summar, Levy Becca, Presnyak Vladimir, Frassetto Andrea, Milton Jaclyn, Salerno Timothy, Benenato Kerry E, Milano Joe, Lynn Andy, Sabnis Staci, Burke Kristine, Besin Gilles, Lukacs Christine M, Guey Lin T, Finn Patrick F, Martini Paolo G V

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Аннотация

Fabry disease is an X-linked lysosomal storage disease caused by loss of alpha galactosidase A (α-Gal A) activity and is characterized by progressive accumulation of globotriaosylceramide and its analogs in all cells and tissues. Although enzyme replacement therapy (ERT) is considered standard of care, the long-term effects of ERT on renal and cardiac manifestations remain uncertain and thus novel therapies are desirable. We herein report preclinical studies evaluating systemic messenger RNA (mRNA) encoding human α-Gal A in wild-type (WT) mice, α-Gal A-deficient mice, and WT non-human primates (NHPs). The pharmacokinetics and distribution of h-α-Gal A mRNA encoded protein in WT mice demonstrated prolonged half-lives of α-Gal A in tissues and plasma. Single intravenous administration of h-α-Gal A mRNA to Gla-deficient mice showed dose-dependent protein activity and substrate reduction. Moreover, long duration (up to 6 weeks) of substrate reductions in tissues and plasma were observed after a single injection. Furthermore, repeat i.v. administration of h-α-Gal A mRNA showed a sustained pharmacodynamic response and efficacy in Fabry mice model. Lastly, multiple administrations to non-human primates confirmed safety and translatability. Taken together, these studies across species demonstrate preclinical proof-of-concept of systemic mRNA therapy for the treatment of Fabry disease and this approach may be useful for other lysosomal storage disorders.

Для цитирования: Zhu Xuling, Yin Ling, Theisen Matt, Zhuo Jenny, Siddiqui Summar, Levy Becca, Presnyak Vladimir, Frassetto Andrea, Milton Jaclyn, Salerno Timothy, Benenato Kerry E, Milano Joe, Lynn Andy, Sabnis Staci, Burke Kristine, Besin Gilles, Lukacs Christine M, Guey Lin T, Finn Patrick F, Martini Paolo G V. Systemic mRNA Therapy for the Treatment of Fabry Disease: Preclinical Studies in Wild-Type Mice, Fabry Mouse Model, and Wild-Type Non-human Primates.. American journal of human genetics. 2019. DOI: 10.1016/j.ajhg.2019.02.003.

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