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MG-108 Agenesis of the corpus callosum and autism associated with zeb1 gene deletion – a case report

2015· article· en· W2277718162 on OpenAlexaff
Michal Inbar‐Feigenberg, Brian HY Chung, Christian R. Marshall, Daniele Merico, Dimitri J. Stavropoulos, David Chitayat

Bibliographic record

Venuenot available
Typearticle
Languageen
FieldMedicine
TopicFetal and Pediatric Neurological Disorders
Canadian institutionsOntario Power GenerationSickKids FoundationHospital for Sick Children
Fundersnot available
KeywordsAgenesis of the corpus callosumCorpus callosumAutismGeneticsBiologyGenetic heterogeneityChromosomeMedicineGenePhenotypeAnatomy

Abstract

fetched live from OpenAlex

Background Agenesis of the corpus callosum (ACC) is a rare congenital anomaly presenting with partial or complete absence of the corpus callosum (CC). ACC could present as an isolated finding or a part of a genetic syndrome. A recent publication reported two cases of mal-development of CC in two unrelated individuals with posterior polymorphous corneal dystrophy (PPCD), an autosomal dominant inherited disorder of the corneal endothelium. Both cases presented novel deletions of the ZEB1 gene. *ZEB1 mutations are reported in approximately one third of PPCD patients. Objective To report a case of a child with ACC and de novo chromosome 10 deletion involving ZEB1 gene. Case report: a 12-year-old girl presented with facial dysmorphism, bilateral renal reflux, developmental delay, autism and ACC. She presented with normal growth parameters, continued to acquire new developmental skills with no regression episodes or seizures. Ophthalmological exam showed thinning of the retinal nerve fibre layer in the nasal aspect around the discs with relative sparing of the macular areas bilaterally. The rest of the ocular examination, including corneal exam was normal. Results Whole genome sequencing revealed heterozygous de novo deletion in chromosome 10p11.23- p11.22 (del:chr10:30814000–32892000) overlapping ZEB1 gene. Conclusion This is the third case reported of ZEB1 gene deletion in a patient with CC anomalies, and the first patient with no corneal anomalies but with autism. This finding might imply a broader phenotypic range for ZEB1 deletion than previously thought and add it to the possible causes of autism.

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)0.001
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.004
Threshold uncertainty score0.008

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0030.001
Science and technology studies0.0020.002
Scholarly communication0.0010.001
Open science0.0010.002
Research integrity0.0040.002
Insufficient payload (model declined to judge)0.0020.001

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 head0.030
GPT teacher head0.235
Teacher spread0.205 · 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 designCase report
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".

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Citations0
Published2015
Admission routes1
Has abstractyes

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