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Record W2036374990 · doi:10.1002/pd.1735

Prenatal diagnosis of Smith‐Lemli‐Opitz syndrome (SLOS) by <i>DHCR7</i> mutation analysis

2007· article· en· W2036374990 on OpenAlexaff
John S. Waye, Barry Eng, Małgorzata J.M. Nowaczyk

Bibliographic record

VenuePrenatal Diagnosis · 2007
Typearticle
Languageen
FieldMedicine
TopicCholesterol and Lipid Metabolism
Canadian institutionsMcMaster UniversityHamilton Health SciencesHamilton Regional Laboratory Medicine Program
Fundersnot available
KeywordsSmith–Lemli–Opitz syndromeAmniotic fluidPrenatal diagnosisChorionic villiChorionic villus samplingMutationFetusPregnancyBiologyMedicineGene mutationObstetricsReductaseGeneticsGene7-Dehydrocholesterol reductasePhysiologyEnzymeBiochemistry

Abstract

fetched live from OpenAlex

OBJECTIVES: We review our experience using mutation analysis of the DHCR7 gene for prenatal diagnosis of Smith-Lemli-Opitz syndrome (SLOS), an autosomal recessive disorder of endogenous cholesterol biosynthesis caused by deficiency of 7-dehydrocholesterol reductase (DHCR7). METHODS AND RESULTS: Prenatal diagnosis of SLOS was conducted for 21 pregnancies involving 15 families. DNA was isolated directly from amniotic fluid cells or chorionic villus samples (CVS), and the DHCR7 gene was screened for the parental mutations using PCR-ARMS and nucleotide sequencing. Sixteen of the pregnancies were unaffected and five were affected. There have been no incidences of misdiagnosed pregnancies. CONCLUSIONS: DHCR7 mutation is a rapid and reliable method for prenatal diagnosis of SLOS, and provides an alternative to specialized biochemical tests for elevated 7DHC in amniotic fluid or CVS.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.180
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0010.002
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0010.000

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 head0.009
GPT teacher head0.259
Teacher spread0.251 · 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 teacher head, not a consensus.

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

Citations24
Published2007
Admission routes1
Has abstractyes

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