Activation of XBP1s attenuates disease severity in models of proteotoxic Charcot-Marie-Tooth type 1B.

In addition, neurophysiological parameters were improved in adult S63del animals overexpressing XBP1s, with a significant increase in nerve conduction velocities and a strong trend towards reduction in F-wave latencies ( Fig. 6F and G ).

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Remarkably, however, in the S63del model XBP1s overexpression appeared to ameliorate the hypomyelination defects, with a clear trend towards reduced g-ratio values ( Fig. 6B and C ).

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