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Record W4247918749 · doi:10.1159/000332071

Alagille Syndrome

2012· book-chapter· en· W4247918749 on OpenAlexaff
Marialena Mouzaki, Binita M. Kamath

Bibliographic record

VenuePediatric and adolescent medicine · 2012
Typebook-chapter
Languageen
FieldMedicine
TopicPediatric Hepatobiliary Diseases and Treatments
Canadian institutionsUniversity of TorontoHospital for Sick ChildrenSickKids Foundation
Fundersnot available
KeywordsAlagille syndromeJAG1PenetranceMedicineLiver diseasePathologyInternal medicineGastroenterologyPhenotypeCholestasisBiologyNotch signaling pathwayGenetics

Abstract

fetched live from OpenAlex

Alagille syndrome is a multisystem, autosomal-dominant condition with variable penetrance, caused by defects in the NOTCH signaling pathway. JAG1 mutations are identified in the majority of patients and a small percentage carry NOTCH2 mutations. There are no clear genotype-phenotype correlations. It is classically described as the constellation of characteristic facies (triangular face, broad forehead, pointed chin), cholestatic liver disease (with paucity of intrahepatic bile ducts on liver biopsy), cardiovascular anomalies (most commonly peripheral pulmonic stenosis), ocular abnormalities (usually posterior embryotoxon) and vertebral anomalies (commonly butterfly vertebrae). It is now clear that Alagille syndrome also frequently affects the kidneys and vascular network. Morbidity and mortality are usually determined by the severity of the hepatic or cardiovascular involvement. Despite significant advances in the understanding of the genetics of this condition, these data have not made significant impact on management. The treatment of Alagille syndrome remains largely supportive.

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: Not applicable · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: none
Teacher disagreement score0.031
Threshold uncertainty score0.104

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0010.000
Scholarly communication0.0010.001
Open science0.0010.001
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0310.012

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 head0.022
GPT teacher head0.249
Teacher spread0.227 · 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 designNot applicable
Domainnot available
GenreReview

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

Citations1
Published2012
Admission routes1
Has abstractyes

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