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Everolimus rescues multiple cellular defects in laminopathy-patient fibroblasts.
DuBose, Amanda J; Lichtenstein, Stephen T; Petrash, Noreen M; Erdos, Michael R; Gordon, Leslie B; Collins, Francis S.
Afiliação
  • DuBose AJ; Genome Technology Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD 20892.
  • Lichtenstein ST; Genome Technology Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD 20892.
  • Petrash NM; Genome Technology Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD 20892.
  • Erdos MR; Genome Technology Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD 20892.
  • Gordon LB; Department of Pediatrics, Hasbro Children's Hospital and Warren Alpert Medical School of Brown University, Providence, RI 02903.
  • Collins FS; Department of Anesthesia, Boston Children's Hospital and Harvard Medical School, Boston, MA 02115.
Proc Natl Acad Sci U S A ; 115(16): 4206-4211, 2018 04 17.
Article em En | MEDLINE | ID: mdl-29581305
LMNA encodes the A-type lamins that are part of the nuclear scaffold. Mutations in LMNA can cause a variety of disorders called laminopathies, including Hutchinson-Gilford progeria syndrome (HGPS), atypical Werner syndrome, and Emery-Dreifuss muscular dystrophy. Previous work has shown that treatment of HGPS cells with the mTOR inhibitor rapamycin or with the rapamycin analog everolimus corrects several of the phenotypes seen at the cellular level-at least in part by increasing autophagy and reducing the amount of progerin, the toxic form of lamin A that is overproduced in HGPS patients. Since other laminopathies also result in production of abnormal and potentially toxic lamin proteins, we hypothesized that everolimus would also be beneficial in those disorders. To test this, we applied everolimus to fibroblast cell lines from six laminopathy patients, each with a different mutation in LMNA Everolimus treatment increased proliferative ability and delayed senescence in all cell lines. In several cell lines, we observed that with treatment, there is a significant improvement in nuclear blebbing, which is a cellular hallmark of HGPS and other lamin disorders. These preclinical results suggest that everolimus might have clinical benefit for multiple laminopathy syndromes.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Progéria / Síndrome de Werner / Distrofia Muscular de Emery-Dreifuss / Lamina Tipo A / Serina-Treonina Quinases TOR / Fibroblastos / Everolimo Limite: Humans Idioma: En Ano de publicação: 2018 Tipo de documento: Article

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Progéria / Síndrome de Werner / Distrofia Muscular de Emery-Dreifuss / Lamina Tipo A / Serina-Treonina Quinases TOR / Fibroblastos / Everolimo Limite: Humans Idioma: En Ano de publicação: 2018 Tipo de documento: Article