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Review
. 2019 Feb;64(2):139-143.
doi: 10.1038/s10038-018-0537-5. Epub 2018 Nov 29.

Gene therapy for lysosomal storage diseases and peroxisomal diseases

Affiliations
Review

Gene therapy for lysosomal storage diseases and peroxisomal diseases

Toya Ohashi. J Hum Genet. 2019 Feb.

Abstract

Gene therapies for lysosomal storage diseases (LSD) and peroxisomal diseases (PD) are rapidly advancing. Most LSDs and PDs are characterized by brain involvement, prompting the development of therapies targeting the brain. There are two types of gene therapy for brain involvement in LSD and PD, i.e., the direct transfer of a therapeutic gene into brain cells and hematopoietic stem cell-targeted gene therapy. The rationale for the latter approach is that brain microglia are derived from hematopoietic cells. Thus, gene-corrected hematopoietic cells migrate into the brain and differentiate into microglial cells. These gene-corrected microglial cells correct the metabolic defects associated with LSD and reduce inflammation in PD and LSD, leading to a clinical benefit. Gene editing technology has recently been applied in this area and a trial focused on LSD is currently ongoing. Although these approaches are still under investigation, very encouraging results have been obtained. This review provides an overview of recently developed gene therapies for various LSDs and PDs, including the results of clinical trials, with an emphasis on the benefits of this approach for these diseases.

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References

    1. Science. 2003 Oct 17;302(5644):415-9 - PubMed
    1. Science. 2009 Nov 6;326(5954):818-23 - PubMed
    1. Hum Gene Ther. 2013 Jun;24(6):630-40 - PubMed
    1. Science. 2013 Aug 23;341(6148):1233158 - PubMed
    1. Hum Gene Ther. 2014 Jun;25(6):506-16 - PubMed

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