Wild-type huntingtin protects from apoptosis upstream of caspase-3

D Rigamonti, JH Bauer, C De-Fraja, L Conti… - Journal of …, 2000 - Soc Neuroscience
D Rigamonti, JH Bauer, C De-Fraja, L Conti, S Sipione, C Sciorati, E Clementi, A Hackam…
Journal of Neuroscience, 2000Soc Neuroscience
Expansion of a polyglutamine sequence in the N terminus of huntingtin is the gain-of-
function event that causes Huntington's disease. This mutation affects primarily the medium-
size spiny neurons of the striatum. Huntingtin is expressed in many neuronal and non-
neuronal cell types, implying a more general function for the wild-type protein. Here we
report that wild-type huntingtin acts by protecting CNS cells from a variety of apoptotic
stimuli, including serum withdrawal, death receptors, and pro-apoptotic Bcl-2 homologs. This …
Expansion of a polyglutamine sequence in the N terminus of huntingtin is the gain-of-function event that causes Huntington's disease. This mutation affects primarily the medium-size spiny neurons of the striatum. Huntingtin is expressed in many neuronal and non-neuronal cell types, implying a more general function for the wild-type protein. Here we report that wild-type huntingtin acts by protecting CNS cells from a variety of apoptotic stimuli, including serum withdrawal, death receptors, and pro-apoptotic Bcl-2 homologs. This protection may take place at the level of caspase-9 activation. The full-length protein also modulates the toxicity of the poly-Q expansion. Cells expressing full-length mutant protein are susceptible to fewer death stimuli than cells expressing truncated mutant huntingtin.
Soc Neuroscience