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Record W2535770099 · doi:10.1002/cphg.24

High‐Risk Screening of Fabry Disease: Analysis of Fifteen Urinary Methylated and Non‐Methylated Gb<sub>3</sub> Isoforms Using Tandem Mass Spectrometry

2016· article· en· W2535770099 on OpenAlexafffund
Mona Abaoui, Michel Boutin, Paméla Lavoie, Christiane Auray‐Blais

Bibliographic record

VenueCurrent Protocols in Human Genetics · 2016
Typearticle
Languageen
FieldMedicine
TopicLysosomal Storage Disorders Research
Canadian institutionsUniversité de Sherbrooke
FundersCanadian Institutes of Health Research
KeywordsGlobotriaosylceramideFabry diseaseGlycosphingolipidGene isoformUrineChemistryTandem mass spectrometryElectrospray ionizationMass spectrometryLactosylceramideChromatographyUrinary systemBiochemistryInternal medicineDiseaseEnzymeMedicineGene

Abstract

fetched live from OpenAlex

Abstract Fabry disease is a multisystemic, X‐linked lysosomal storage disorder caused by mutations in the GLA gene, leading to α‐galactosidase A deficiency and resulting in the accumulation of glycosphingolipids in different tissues and biological fluids. Glycosphingolipid biomarkers, such as globotriaosylceramide (Gb3) isoforms, globotriaosylsphingosine (lyso‐Gb3) and related analogs, and galabiosylceramide (Ga2) isoforms and analogs, are found to be abnormally increased in urine and in plasma of Fabry patients and have the potential to be used as specific biomarkers of the disease. This unit presents a protocol for the relative quantification of fifteen urinary isoforms of Gb3 analyzed simultaneously with creatinine by ultra‐performance liquid chromatography (UPLC) coupled to tandem mass spectrometry (MS/MS). In order to purify urine samples, a liquid‐liquid extraction is performed and samples are analyzed by MS/MS in positive electrospray ionization mode. These biomarkers are useful for screening, diagnosis, and long‐term monitoring of Fabry disease patients. We have shown that the methylated Gb3 isoforms are particularly useful for screening Fabry patients who present with late‐onset cardiac variant mutations. © 2016 by John Wiley & Sons, Inc.

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)0.001
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: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.001
Threshold uncertainty score0.003

Distilled classifier scores by category (both heads)

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

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 head0.051
GPT teacher head0.369
Teacher spread0.318 · 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 designObservational
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

Citations3
Published2016
Admission routes2
Has abstractyes

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