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Record W4313545337 · doi:10.1126/sciadv.abn9793

In vitro cellular reprogramming to model gonad development and its disorders

2023· article· en· W4313545337 on OpenAlexfundno aff
Nitzan Gonen, Caroline Eozénou, Richard Mitter, Maëva Elzaïat, Isabelle Stévant, Rona Aviram, Andreia S. Bernardo, Almira Chervova, Somboon Wankanit, Emmanuel Frachon, Pierre‐Henri Commère, Sylvie Brailly‐Tabard, Léo Valon, Laura Barrio, Romain Levayer, Inas Mazen, Samy Gobaa, James C. Smith, Kenneth McElreavey, Robin Lovell‐Badge, Anu Bashamboo

Bibliographic record

VenueScience Advances · 2023
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetic and Clinical Aspects of Sex Determination and Chromosomal Abnormalities
Canadian institutionsnot available
FundersBiotechnology and Biological Sciences Research CouncilMedical Research CouncilInstitute of GeneticsInstitut National de la Recherche AgronomiqueAgence Nationale de la RechercheWellcome TrustFrancis Crick InstituteBritish Heart FoundationBar-Ilan UniversityCancer Research UKUniversity of Minnesota
KeywordsBiologyReprogrammingGonadEmbryonic stem cellTranscriptomeInduced pluripotent stem cellCell biologyGonadal ridgeDisorders of sex developmentSertoli cellSexual differentiationProgenitor cellSomatic cellGeneticsGeneStem cellEndocrinologyGene expressionSpermatogenesis

Abstract

fetched live from OpenAlex

During embryonic development, mutually antagonistic signaling cascades determine gonadal fate toward a testicular or ovarian identity. Errors in this process result in disorders of sex development (DSDs), characterized by discordance between chromosomal, gonadal, and anatomical sex. The absence of an appropriate, accessible in vitro system is a major obstacle in understanding mechanisms of sex-determination/DSDs. Here, we describe protocols for differentiation of mouse and human pluripotent cells toward gonadal progenitors. Transcriptomic analysis reveals that the in vitro–derived murine gonadal cells are equivalent to embryonic day 11.5 in vivo progenitors. Using similar conditions, Sertoli-like cells derived from 46,XY human induced pluripotent stem cells (hiPSCs) exhibit sustained expression of testis-specific genes, secrete anti-Müllerian hormone, migrate, and form tubular structures. Cells derived from 46,XY DSD female hiPSCs, carrying an NR5A1 variant, show aberrant gene expression and absence of tubule formation. CRISPR-Cas9–mediated variant correction rescued the phenotype. This is a robust tool to understand mechanisms of sex determination and model DSDs.

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)0.000
metaresearch head score (Gemma)0.000
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 score0.091
Threshold uncertainty score0.239

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
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 head0.014
GPT teacher head0.291
Teacher spread0.277 · 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

Citations42
Published2023
Admission routes1
Has abstractyes

Explore more

Same venueScience AdvancesSame topicGenetic and Clinical Aspects of Sex Determination and Chromosomal AbnormalitiesFrench-language works237,207