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Record W4409956483 · doi:10.1002/cpt.3693

Impaired Drug Metabolism Caused by Compound Heterozygous Loss‐of‐Function Variants in <i>CYP3A4</i>: A Case Report

2025· article· en· W4409956483 on OpenAlexafffund
Han Byul Kang, Iris Cohn, Sanjay Mahant, Gregory Costain, Shinya Ito, Ruud H J Verstegen

Bibliographic record

VenueClinical Pharmacology & Therapeutics · 2025
Typearticle
Languageen
FieldPharmacology, Toxicology and Pharmaceutics
TopicPharmacogenetics and Drug Metabolism
Canadian institutionsSickKids FoundationInstitute for Clinical Evaluative SciencesPublic Health OntarioUniversity of TorontoHospital for Sick Children
FundersHospital for Sick ChildrenUniversity of Toronto
KeywordsCYP3A4PharmacogeneticsMedicineDrugPharmacologyPopulationDrug metabolismInternal medicineMetabolismBiologyGeneticsGenotypeGene

Abstract

fetched live from OpenAlex

Recent consensus guidelines recommend including *22 as the only CYP3A4 variant in pharmacogenetic test panels, given the presence of sufficient functional data as well as a relatively high minor allele frequency. We here describe the clinical presentation of a child with an extremely high sirolimus blood concentration (88.4 μg/L; therapeutic range at trough: 5-15 μg/L) and prolonged elimination half-life of 121 hours (mean 14 hours in the pediatric population) following only eight standard-dose administrations. Due to the clinical suspicion of impaired CYP3A4 metabolism, in the absence of drug-drug or drug-food interactions, clinical sequencing of CYP3A4 was performed. This revealed that the patient is compound heterozygous for two rare loss-of-function variants in CYP3A4 (*8/*20). This clinical case highlights that a routine pharmacogenetic test panel that includes *22 as the only CYP3A4 variant is not a suitable diagnostic test for individuals with an increased pre-test probability of altered CYP3A4 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)0.001
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 score0.006
Threshold uncertainty score0.010

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.003
Meta-epidemiology (narrow)0.0030.002
Meta-epidemiology (broad)0.0020.002
Bibliometrics0.0030.001
Science and technology studies0.0020.003
Scholarly communication0.0020.002
Open science0.0010.002
Research integrity0.0060.005
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 head0.118
GPT teacher head0.473
Teacher spread0.356 · 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
Published2025
Admission routes2
Has abstractyes

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