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Record W2296292121 · doi:10.1681/asn.2015060621

Developmental Origins for Kidney Disease Due to Shroom3 Deficiency

2016· article· en· W2296292121 on OpenAlexafffund
Hadiseh Khalili, Alexandra Sull, Sanjay Sarin, Felix Boivin, Rami Halabi, Bruno Svajger, Aihua Li, Valerie Wenche Cui, Thomas A. Drysdale, Darren Bridgewater

Bibliographic record

VenueJournal of the American Society of Nephrology · 2016
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicRenal and related cancers
Canadian institutionsSt. Joseph’s Healthcare HamiltonWestern UniversityMcMaster University
FundersNational Research Council CanadaKidney Foundation of CanadaMcMaster University
KeywordsPodocyteBiologyKidney developmentKidneyKidney diseaseGlomerulosclerosisMesenchymeFocal segmental glomerulosclerosisZebrafishEndocrinologyGlomerulonephritisInternal medicineCell biologyPathologyGeneticsProteinuriaGeneMedicineMesenchymal stem cellEmbryonic stem cell

Abstract

fetched live from OpenAlex

CKD is a significant health concern with an underlying genetic component. Multiple genome–wide association studies (GWASs) strongly associated CKD with the shroom family member 3 ( SHROOM3 ) gene, which encodes an actin-associated protein important in epithelial morphogenesis. However, the role of SHROOM3 in kidney development and function is virtually unknown. Studies in zebrafish and rat showed that alterations in Shroom3 can result in glomerular dysfunction. Furthermore, human SHROOM3 variants can induce impaired kidney function in animal models. Here, we examined the temporal and spatial expression of Shroom3 in the mammalian kidney. We detected Shroom3 expression in the condensing mesenchyme, Bowman’s capsule, and developing and mature podocytes in mice. Shroom3 null ( Shroom3Gt/Gt ) mice showed marked glomerular abnormalities, including cystic and collapsing/degenerating glomeruli, and marked disruptions in podocyte arrangement and morphology. These podocyte-specific abnormalities are associated with altered Rho–kinase/myosin II signaling and loss of apically distributed actin. Additionally, Shroom3 heterozygous ( Shroom3Gt/+ ) mice showed developmental irregularities that manifested as adult-onset glomerulosclerosis and proteinuria. Taken together, our results establish the significance of Shroom3 in mammalian kidney development and progression of kidney disease. Specifically, Shroom3 maintains normal podocyte architecture in mice via modulation of the actomyosin network, which is essential for podocyte function. Furthermore, our findings strongly support the GWASs that suggest a role for SHROOM3 in human kidney disease.

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: Not applicable · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.787
Threshold uncertainty score0.188

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 head0.008
GPT teacher head0.250
Teacher spread0.242 · 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 designNot applicable
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

Citations62
Published2016
Admission routes2
Has abstractyes

Explore more

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