TY - JOUR
T1 - Tau silencing by siRNA in the P301S mouse model of tauopathy
AU - Xu, Hong
AU - Rösler, Thomas W.
AU - Carlsson, Thomas
AU - de Andrade, Anderson
AU - Fiala, Ondrej
AU - Höllerhage, Matthias
AU - Oertel, Wolfgang H.
AU - Goedert, Michel
AU - Aigner, Achim
AU - Höglinger, Günter U.
N1 - Publisher Copyright:
© 2014 Bentham Science Publishers.
PY - 2014/3/1
Y1 - 2014/3/1
N2 - Suppression of tau protein expression has been shown to improve behavioral deficits in mouse models of tauopathies, offering an attractive therapeutic approach. Experimentally this had been achieved by switching off the promoters controlling the transgenic human tau gene (MAPT), which is not possible in human patients. The aim of the present study was therefore to evaluate the effectiveness of small interfering RNAs (siRNAs) and their cerebral delivery to suppress human tau expression in vivo, which might be a therapeutic option for human tauopathies. We used primary cortical neurons of transgenic mice expressing P301S-mutated human tau and Lund human mesencephalic (LUHMES) cells to validate the suppressive effect of siRNA in vitro. For measuring the effect in vivo, we stereotactically injected siRNA into the brains of P301S mice to reveal the suppression of tau by immunochemistry (AT180, MC1, and CP13 antibodies). We found that the Accell™ SMART pool siRNA against MAPT can effectively suppress tau expression in vitro and in vivo without a specific delivery agent. The siRNA showed a moderate distribution in the hippocampus of mice after single injection. NeuN, GFAP, Iba-1, MHC II immunoreactivities and the terminal deoxynucleotidyl transferase dUTP nick end labeling (TUNEL) assay showed neither signs of neurotoxicity or neuroinflammation nor apoptosis when MAPT siRNA is present in the hippocampus. Our data suggest that siRNA against MAPT can serve as a potential tool for gene therapy in tauopathies.
AB - Suppression of tau protein expression has been shown to improve behavioral deficits in mouse models of tauopathies, offering an attractive therapeutic approach. Experimentally this had been achieved by switching off the promoters controlling the transgenic human tau gene (MAPT), which is not possible in human patients. The aim of the present study was therefore to evaluate the effectiveness of small interfering RNAs (siRNAs) and their cerebral delivery to suppress human tau expression in vivo, which might be a therapeutic option for human tauopathies. We used primary cortical neurons of transgenic mice expressing P301S-mutated human tau and Lund human mesencephalic (LUHMES) cells to validate the suppressive effect of siRNA in vitro. For measuring the effect in vivo, we stereotactically injected siRNA into the brains of P301S mice to reveal the suppression of tau by immunochemistry (AT180, MC1, and CP13 antibodies). We found that the Accell™ SMART pool siRNA against MAPT can effectively suppress tau expression in vitro and in vivo without a specific delivery agent. The siRNA showed a moderate distribution in the hippocampus of mice after single injection. NeuN, GFAP, Iba-1, MHC II immunoreactivities and the terminal deoxynucleotidyl transferase dUTP nick end labeling (TUNEL) assay showed neither signs of neurotoxicity or neuroinflammation nor apoptosis when MAPT siRNA is present in the hippocampus. Our data suggest that siRNA against MAPT can serve as a potential tool for gene therapy in tauopathies.
KW - FTDP-17-tau
KW - In vitro
KW - In vivo
KW - P301S MAPT transgenic mouse
KW - RNAi
KW - siRNA tauopathy
UR - http://www.scopus.com/inward/record.url?scp=84911493266&partnerID=8YFLogxK
U2 - 10.2174/156652321405140926160602
DO - 10.2174/156652321405140926160602
M3 - Article
C2 - 25687501
AN - SCOPUS:84911493266
SN - 1566-5232
VL - 14
SP - 343
EP - 351
JO - Current gene therapy
JF - Current gene therapy
IS - 5
ER -