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The schizophrenia associated protein DISC1 forms a multivalent tetrameric hub via conserved UVR dimers

Abstract:
DISC1 is a pleiotropic protein with essential roles in neuronal proliferation and migration, intracellular signalling and cargo transport. It associates with a diverse array of partner molecules in these contexts. Mutations at the DISC1 locus are strongly associated with a spectrum of mental illnesses such as schizophrenia and depression. Despite its clinical relevance, the molecular architecture and function of DISC1 have remained largely elusive. We present a cryo-EM structure of the entire conserved core region of DISC1. The structure reveals an intricate homotetrameric assembly that harbours conserved bacteria-derived UVR domains. Four of these domains, one from each monomer, mediate extensive contacts forming two asymmetric dimer units. The dimers in turn interface with each other at two distinct coiled coil domains to achieve a two-fold symmetric tetramer. Mutational analysis shows that this tetrameric architecture enables DISC1 to simultaneously bind multiple copies of NDE1 client protein. Importantly, tetramerization and partner binding are structurally independent functions of DISC1. Altogether, our study provides a compelling molecular model of an ancient bacteria protein fold participating in the assembly of a multivalent mammalian scaffold hub that can coordinate multiple partner molecules.
Publication status:
Published
Peer review status:
Peer reviewed

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Publisher copy:
10.1038/s41467-026-71838-6

Authors

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Institution:
University of Oxford
Division:
MSD
Department:
Biochemistry
Role:
Author
ORCID:
0000-0001-5716-5170
More by this author
Institution:
University of Oxford
Division:
MPLS
Department:
Chemistry
Sub department:
Sub-Department of Physical and Theoretical Chemistry
Role:
Author
ORCID:
0000-0003-1282-1562
More by this author
Institution:
University of Oxford
Oxford college:
Somerville College
Role:
Author
ORCID:
0000-0003-3506-6045
More by this author
Institution:
University of Oxford
Division:
MPLS
Department:
Chemistry
Sub department:
Sub-Department of Physical and Theoretical Chemistry
Oxford college:
Exeter College
Role:
Author
ORCID:
0000-0003-0136-7704


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Funder identifier:
https://ror.org/029chgv08
Grant:
202827/Z/16/Z
226647/Z/22/Z
More from this funder
Funder identifier:
https://ror.org/001aqnf71
Grant:
EP/X025713/1
More from this funder
Funder identifier:
https://ror.org/0439y7842
Grant:
EP/T03419X/1


Publisher:
Nature Research
Journal:
Nature Communications More from this journal
Place of publication:
England
Publication date:
2026-04-17
Acceptance date:
2026-03-30
DOI:
EISSN:
2041-1723
Pmid:
41997921


Language:
English
Pubs id:
2408281
Local pid:
pubs:2408281
Source identifiers:
W7154597193
Deposit date:
2026-05-11
ARK identifier:

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