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Reversal of phenotypes in MECP2 duplication mice using genetic rescue or antisense oligonucleotides

Author

Listed:
  • Yehezkel Sztainberg

    (Baylor College of Medicine
    Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital)

  • Hong-mei Chen

    (The Cain Foundation Laboratories, Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital
    Baylor College of Medicine
    Baylor College of Medicine)

  • John W. Swann

    (The Cain Foundation Laboratories, Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital
    Baylor College of Medicine
    Baylor College of Medicine)

  • Shuang Hao

    (Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital
    Baylor College of Medicine)

  • Bin Tang

    (Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital
    Baylor College of Medicine)

  • Zhenyu Wu

    (Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital
    Baylor College of Medicine)

  • Jianrong Tang

    (Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital
    Baylor College of Medicine)

  • Ying-Wooi Wan

    (Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital
    Baylor College of Medicine)

  • Zhandong Liu

    (Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital
    Baylor College of Medicine)

  • Frank Rigo

    (Isis Pharmaceuticals)

  • Huda Y. Zoghbi

    (Baylor College of Medicine
    Jan and Dan Duncan Neurological Research Institute at Texas Children’s Hospital
    Baylor College of Medicine
    Howard Hughes Medical Institute, Baylor College of Medicine)

Abstract

Genetic correction of MeCP2 levels largely reversed the behavioural, molecular and physiological deficits associated with MECP2 duplication syndrome in a transgenic mouse model; similarly, reduction of MeCP2 levels using an antisense oligonucleotide strategy resulted in phenotypic rescue in adult transgenic mice, and dose-dependently corrected MeCP2 levels in cells from patients with MECP2 duplication.

Suggested Citation

  • Yehezkel Sztainberg & Hong-mei Chen & John W. Swann & Shuang Hao & Bin Tang & Zhenyu Wu & Jianrong Tang & Ying-Wooi Wan & Zhandong Liu & Frank Rigo & Huda Y. Zoghbi, 2015. "Reversal of phenotypes in MECP2 duplication mice using genetic rescue or antisense oligonucleotides," Nature, Nature, vol. 528(7580), pages 123-126, December.
    • Handle: RePEc:nat:nature:v:528:y:2015:i:7580:d:10.1038_nature16159
    DOI: 10.1038/nature16159
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    Cited by:

    1. Takeo Kubota & Kazuki Mochizuki, 2016. "Epigenetic Effect of Environmental Factors on Autism Spectrum Disorders," IJERPH, MDPI, vol. 13(5), pages 1-12, May.
    2. James Okoh & Jacqunae Mays & Alexandre Bacq & Juan A. Oses-Prieto & Stefka Tyanova & Chien-Ju Chen & Khalel Imanbeyev & Marion Doladilhe & Hongyi Zhou & Paymaan Jafar-Nejad & Alma Burlingame & Jeffrey, 2023. "Targeted suppression of mTORC2 reduces seizures across models of epilepsy," Nature Communications, Nature, vol. 14(1), pages 1-13, December.

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