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Record W4414650730 · doi:10.1038/s41467-025-63752-0

Molecular cartography of the human down syndrome and trisomic mouse brain

2025· article· en· W4414650730 on OpenAlexaff
Min Yi Feng, Wuxinhao Cao, Nareh Tahmasian, Bharti Kukreja, Gen Li, Bianca Rusu, Ji‐Young Youn, Brian T. Kalish

Bibliographic record

VenueNature Communications · 2025
Typearticle
Languageen
FieldMedicine
TopicDown syndrome and intellectual disability research
Canadian institutionsSickKids FoundationMental Health Research CanadaHospital for Sick ChildrenUniversity of Toronto
Fundersnot available
KeywordsCorticogenesisTranscriptomeDown syndromeTrisomyNeurogenesisMulticellular organismHuman brainProteomicsChromosome 21

Abstract

fetched live from OpenAlex

Down syndrome (DS, or Trisomy 21) is one of the most common genetic causes of intellectual disability. DS results in both abnormal neurodevelopment and accelerated neurodegeneration, but the molecular mechanisms underlying abnormal corticogenesis are incompletely understood. To gain molecular insight into the prenatal neurobiology of DS, we performed single-nucleus sequencing, spatial transcriptomics, and proteomics on mid-gestational prenatal human cortex. We captured altered expression dynamics of lineage commitment genes and de-repression of transposable elements in DS neural progenitor cells, which suggest changes to the fate and functionality of neuronal and glial cells. Given the importance of linking human and model system pathobiology, we also performed highly multiplexed RNA in situ spatial transcriptomics on a well-established trisomic mouse model (Ts65Dn) to study the cellular landscape of the trisomic brain during early development and maturation. We profiled the spatial transcriptome of > 240,000 cells in the mouse brain and identified trisomy-associated gene expression patterns in the molecular control of neurogenesis and gliogenesis. Together, our study provides an extensive resource for understanding of the complex multicellular processes underlying DS neurodevelopment. Down Syndrome is a common genetic cause of intellectual disability and is associated with altered fetal neurodevelopment. Here, the authors identify altered expression dynamics of lineage commitment genes and de-repression of transposable elements in DS neural progenitor cells.

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.004
Threshold uncertainty score0.013

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0040.001

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 head0.021
GPT teacher head0.359
Teacher spread0.338 · 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 source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
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

Citations5
Published2025
Admission routes1
Has abstractyes

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