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Exon Sequencing for the Presence of Late Onset Tay‐Sachs

2018· article· en· W3176543765 on OpenAlexaboutno aff
Joshua Nathan Slostad, Jodi E. Goldberg

Bibliographic record

VenueThe FASEB Journal · 2018
Typearticle
Languageen
FieldNeuroscience
TopicAutism Spectrum Disorder Research
Canadian institutionsnot available
Fundersnot available
KeywordsGeneticsTay-Sachs diseasePopulationDNA sequencingAlleleMutationExonBiologyGeneGenealogyDiseaseMedicineHistory

Abstract

fetched live from OpenAlex

Tay‐Sachs is a disease that causes rapid developmental stagnation and death in infants. It primarily affects people of French Canadian, Cajun, and Ashkenazi Jewish descent. In recent years an alternate form of the disease, called late onset Tay‐Sachs (LOTS), has been described. LOTS is caused by the inheritance of one null allele and a pseudodeficiency allele of the hexosaminidase A ( HEXA) gene. The mutation rate of the pseudodeficiency is not well known. The aim of this experiment was to examine the DNA of the descendants of an Ashkenazi Jewish individual who was believed to have undiagnosed LOTS. The purpose of this case study is to attempt to confirm this suspected diagnosis by identifying potential mutations in the family members. DNA of the HEXA gene from three family members of the suspected LOTS patient (wife and 2 daughters) and one control set (unrelated female) was sequenced. The sequences were aligned and compared both by hand and using the NCBI Blast database and the Benchling tool. No known mutations or confirmed novel mutations have been found in all fourteen exons sequenced. Currently re‐sequencing of some regions that showed potential single nucleotide polymorphisms (SNPs) as well as sequencing the promoter region of the gene is underway. If a mutation is confirmed this research could help pave the way for further genetic testing in the broader population in order to fully grasp the prevalence of LOTS pseudodeficiency alleles within the Ashkenazi population. Support or Funding Information Hamline Undergraduate Collaborative Research Ridgeway Forum Fund This abstract is from the Experimental Biology 2018 Meeting. There is no full text article associated with this abstract published in The FASEB Journal .

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: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.009

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.000
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 head0.076
GPT teacher head0.330
Teacher spread0.254 · 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 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

Citations0
Published2018
Admission routes1
Has abstractyes

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