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Record W2763599687 · doi:10.1073/pnas.1713625114

WD40-repeat 47, a microtubule-associated protein, is essential for brain development and autophagy

2017· article· en· W2763599687 on OpenAlexfundno aff
Meghna Kannan, Efil Bayam, Christel Wagner, Bruno Rinaldi, Perrine F. Kretz, Peggy Tilly, Marna Roos, L. McGillewie, Séverine Bär, Shilpi Minocha, Claire Chevalier, Chrystelle Po, Jamel Chelly, Jean‐Louis Mandel, Renato Borgatti, Amélie Piton, Craig Kinnear, Ben Loos, David J. Adams, Yann Hérault, Stephan C. Collins, Sylvie Friant, Juliette D. Godin, Binnaz Yalcin, Valerie E. Vancollie, Lauren F. E. Anthony, Simon A. Maguire, David Lafont, Selina Pearson, Amy S. Gates, Mark Sanderson, Carl Shannon, Maksymilian T. Sumowski, Robbie S. B. McLaren-Jones, Agnieszka Świątkowska, Christopher Isherwood, Emma L. Cambridge, Heather Wilson, Susana Caetano, Anna Karin B. Maguire, Antonella Galli, Anneliese O. Speak, Joshua Dench, Elizabeth Tuck, Jeanne Estabel, Angela Green, Catherine Tudor, Emma Siragher, Monika Dabrowska, Cecilia Mazzeo, Yvette Hooks, Fiona Kussy, Mark Griffiths, David Gannon, Brendan Doe, Katharina Boroviak, Hannah Wardle‐Jones, Nicola Griggs, Joanna Bottomley, Edward J. Ryder, Diane Gleeson, Jacqueline K. White, Ramiro Ramírez‐Solis, Christopher J. Lelliott

Bibliographic record

VenueProceedings of the National Academy of Sciences · 2017
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicEpigenetics and DNA Methylation
Canadian institutionsnot available
FundersUniversiteit UtrechtÉcole Polytechnique Fédérale de LausanneInstitute of GeneticsInstitut National de la Santé et de la Recherche MédicaleFondation FyssenAgence Nationale de la RechercheMedical Research CouncilCentre National de la Recherche ScientifiqueSouth African Medical Research Council
KeywordsBiologyLissencephalyMicrocephalyCell biologyNeurogenesisAutophagyMicrotubule-associated proteinNeural developmentMicrotubuleNeurosciencePhenotypeNeural stem cellGeneGeneticsStem cell

Abstract

fetched live from OpenAlex

showed lethality, extensive fiber defects, microcephaly, thinner cortices, and sensory motor gating abnormalities. We showed that WDR47 shares functional characteristics with LIS1 and participates in key microtubule-mediated processes, including neural stem cell proliferation, radial migration, and growth cone dynamics. In absence of WDR47, the exhaustion of late cortical progenitors and the consequent decrease of neurogenesis together with the impaired survival of late-born neurons are likely yielding to the worsening of the microcephaly phenotype postnatally. Interestingly, the WDR47-specific C-terminal to LisH (CTLH) domain was associated with functions in autophagy described in mammals. Silencing WDR47 in hypothalamic GT1-7 neuronal cells and yeast models independently recapitulated these findings, showing conserved mechanisms. Finally, our data identified superior cervical ganglion-10 (SCG10) as an interacting partner of WDR47. Taken together, these results provide a starting point for studying the implications of WDR proteins in neuronal regulation of microtubules and autophagy.

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 score0.044
Threshold uncertainty score0.306

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 head0.031
GPT teacher head0.319
Teacher spread0.288 · 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

Citations99
Published2017
Admission routes1
Has abstractyes

Explore more

Same venueProceedings of the National Academy of SciencesSame topicEpigenetics and DNA MethylationFrench-language works237,207