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Record W4409967622 · doi:10.1093/nar/gkaf347

PRMT5 promotes full-length <i>HTT</i> expression by repressing multiple proximal intronic polyadenylation sites

2025· article· en· W4409967622 on OpenAlexafffund
Manisha Yadav, Mona Alqazzaz, Felipe E. Ciamponi, Jolene Caifeng Ho, Maxim I. Maron, Aiden M. Sababi, Graham MacLeod, Moloud Ahmadi, Garrett Bullivant, Vincent Tano, Sarah R. Langley, María Sánchez‐Osuna, Patty Sachamitr, Michelle Kushida, Costanza Ferrari Bardile, Mahmoud A. Pouladi, Rebecca Kurtz, Laura M. Richards, Trevor J. Pugh, Mike Tyers, Stéphane Angers, Peter Dirks, Gary D. Bader, Ray Truant, Katlin B. Massirer, Dalia Baršytė-Lovejoy, David Shechter, Rachel Harding, C.H. Arrowsmith, Panagiotis Prinos

Bibliographic record

VenueNucleic Acids Research · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicCancer-related gene regulation
Canadian institutionsUniversity Health NetworkOntario Institute for Cancer ResearchStructural Genomics ConsortiumMcMaster UniversityHospital for Sick ChildrenUniversité de MontréalBC Children's HospitalUniversity of TorontoInstitute for Research in Immunology and CancerSickKids FoundationUniversity of British ColumbiaLunenfeld-Tanenbaum Research InstitutePrincess Margaret Cancer Centre
FundersNational Institute of General Medical SciencesJanssen BiotechCanadian Institutes of Health ResearchBristol-Myers Squibb CanadaNational Institutes of HealthMitacsOntario Genomics InstituteStand Up To Cancer CanadaGenentechGovernment of OntarioOntario Institute for Cancer ResearchStructural Genomics ConsortiumMerck KGaAGenome CanadaIrma T. Hirschl TrustAmerican Association for Cancer ResearchBayerTakeda Pharmaceuticals U.S.A.PfizerEntertainment Industry FoundationBoehringer IngelheimGenentech Foundation
KeywordsHuntingtinPolyadenylationBiologyRNA splicingExonIntronTrinucleotide repeat expansionAlternative splicingRNA-binding proteinMessenger RNAMolecular biologyPrecursor mRNACell biologyGeneticsRNAGeneAllele

Abstract

fetched live from OpenAlex

Expansion of the CAG trinucleotide repeat tract in exon 1 of the Huntingtin (HTT) gene causes Huntington's disease (HD) through the expression of a polyglutamine-expanded form of the HTT protein. This mutation triggers cellular and biochemical pathologies, leading to cognitive, motor, and psychiatric symptoms in HD patients. Targeting HTT splicing with small molecule drugs is a compelling approach to lowering HTT protein levels to treat HD, and splice modulators are currently being tested in the clinic. Here, we identify PRMT5 as a novel regulator of HTT messenger RNA (mRNA) splicing and alternative polyadenylation. PRMT5 inhibition disrupts the splicing of HTT introns 9 and 10, leading to the activation of multiple proximal intronic polyadenylation sites within these introns and promoting premature termination, cleavage, and polyadenylation of the HTT mRNA. This suggests that HTT protein levels may be lowered due to this mechanism. We also detected increasing levels of these truncated HTT transcripts across a series of neuronal differentiation samples, which correlated with lower PRMT5 expression. Notably, PRMT5 inhibition in glioblastoma stem cells potently induced neuronal differentiation. We posit that PRMT5-mediated regulation of intronic polyadenylation, premature termination, and cleavage of the HTT mRNA modulates HTT expression and plays an important role during neuronal differentiation.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.017
Threshold uncertainty score0.785

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.018
GPT teacher head0.321
Teacher spread0.303 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designBench or experimental
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations3
Published2025
Admission routes2
Has abstractyes

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