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MG-140 17Q21.31 microdeletion syndrome: A description of two cases

2015· article· en· W2413084409 on OpenAlexaff
Kurston Doonanco, Cherise Klotz, Judy Chernos, Maisa Yoshimoto, Shailly Jain

Bibliographic record

Venuenot available
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomic variations and chromosomal abnormalities
Canadian institutionsAlberta Children's Hospital
Fundersnot available
KeywordsHypotoniaMicrodeletion syndromeComparative genomic hybridizationIntellectual disabilityPediatricsMedicineGeneticsCraniofacial abnormalityDysplasiaLaryngomalaciaMedical geneticsGenetic counselingCraniofacialPhenotypeBiologyPathologyGeneSurgeryGenome

Abstract

fetched live from OpenAlex

17q21.31 microdeletion syndrome, also known as Koolen-De Vries syndrome, commonly involves a 500- to 650-kb heterozygous deletion and has a possible incidence of 1 in 16,000. The most frequent clinical features include distinctive facial features, significant speech delays with mild global developmental delays (GDD), childhood hypotonia, congenital malformations, and a friendly disposition. Here we report two patients with 17q21.31 microdeletion syndrome referred for evaluation by metabolic genetics. Both patients were referred at approximately two and a half years of age for evaluation of lysosomal storage disease in view of presumed plateauing in speech development, GDD, and coarse and dysmorphic facial features. Patient one also had seizures and laryngomalacia. She was found to carry a 987.4 kb deletion, the largest described in the literature to our knowledge, extending distally to partially include the gene NSF (Figure 1). Patient two also had hip dysplasia and mild hypotonia. She was found to carry a 695.5 kb deletion (Figure 2). By presenting these two cases, we hope to add to the phenotype of 17q21.31 microdeletion syndrome and to outline the importance of considering comparative genomic hybridization in patients initially suspected to have inborn errors of metabolism.

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)0.003
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 score0.003
Threshold uncertainty score0.010

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.003
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0030.002
Science and technology studies0.0020.002
Scholarly communication0.0010.001
Open science0.0010.002
Research integrity0.0030.001
Insufficient payload (model declined to judge)0.0030.001

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 head0.246
Teacher spread0.216 · 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".

Quick stats

Citations0
Published2015
Admission routes1
Has abstractyes

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