Article abstract


Nature Structural & Molecular Biology 16, 380 - 389 (2009)
Published online: 8 March 2009 | doi:10.1038/nsmb.1570

Polyglutamine disruption of the huntingtin exon 1 N terminus triggers a complex aggregation mechanism

Ashwani K Thakur1,2,4, Murali Jayaraman1,2,4, Rakesh Mishra1,2, Monika Thakur1,2, Veronique M Chellgren3, In-Ja L Byeon1, Dalaver H Anjum1, Ravindra Kodali1,2, Trevor P Creamer3, James F Conway1, Angela M Gronenborn1 & Ronald Wetzel1,2


Simple polyglutamine (polyQ) peptides aggregate in vitro via a nucleated growth pathway directly yielding amyloid-like aggregates. We show here that the 17-amino-acid flanking sequence (HTTNT) N-terminal to the polyQ in the toxic huntingtin exon 1 fragment imparts onto this peptide a complex alternative aggregation mechanism. In isolation, the HTTNT peptide is a compact coil that resists aggregation. When polyQ is fused to this sequence, it induces in HTTNT, in a repeat-length dependent fashion, a more extended conformation that greatly enhances its aggregation into globular oligomers with HTTNT cores and exposed polyQ. In a second step, a new, amyloid-like aggregate is formed with a core composed of both HTTNT and polyQ. The results indicate unprecedented complexity in how primary sequence controls aggregation within a substantially disordered peptide and have implications for the molecular mechanism of Huntington's disease.

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  1. Department of Structural Biology.
  2. Pittsburgh Institute for Neurodegenerative Diseases, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania 15260, USA.
  3. Department of Molecular and Cellular Biochemistry, University of Kentucky, Lexington, Kentucky 40536, USA.
  4. These authors contributed equally to this work.

Correspondence to: Ronald Wetzel1,2 e-mail: rwetzel@pitt.edu



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