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Record W4411441544 · doi:10.1177/01455613251347923

Early-Onset Hearing Loss in Leber’s Hereditary Optic Neuropathy: A Case Report

2025· article· en· W4411441544 on OpenAlexaff
Maria Al Bandari, Enas Nasr, Sharon L. Cushing, Michal Inbar‐Feigenberg

Bibliographic record

VenueEar Nose & Throat Journal · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicMitochondrial Function and Pathology
Canadian institutionsSickKids FoundationUniversity of TorontoLondon Health Sciences CentreHospital for Sick Children
Fundersnot available
KeywordsMedicineSensorineural hearing lossHearing lossOptic neuropathyAuditory neuropathyMagnetic resonance imagingPresentation (obstetrics)AudiologyPediatricsSurgeryOphthalmologyOptic nerveRadiology

Abstract

fetched live from OpenAlex

Leber hereditary optic neuropathy (LHON) is one of the most common mitochondrial disorders that is characterized in young adults and teenagers as bilateral, painless, subacute visual failure. Extraocular manifestations include neurological and cardiac features. Sensorineural hearing loss (SNHL) has not been reported as a clinical feature of this disorder. We report a patient diagnosed with LHON having the common m.11778G>A; p. Arg340 pathogenic variant who was also diagnosed with bilateral mild-to-moderate high-frequency SNHL as a neonate through our provincial newborn screening program. Genetic workup, including a next-generation sequencing "Comprehensive Hereditary Hearing Loss Panel" for common and non-syndromic hearing loss and sequencing of the mitochondrial genome, was negative for a second pathogenic variant. The infectious workup was negative. Non-enhanced magnetic resonance imaging of the brain and internal auditory canal was normal. To our knowledge, SNHL has not been reported before as a clinical feature of patients diagnosed with LHON, and hence this rare and unusual presentation merits reporting.

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)0.002
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 score0.004
Threshold uncertainty score0.012

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0030.001
Science and technology studies0.0030.002
Scholarly communication0.0020.002
Open science0.0010.002
Research integrity0.0040.002
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 head0.015
GPT teacher head0.276
Teacher spread0.261 · 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

Citations1
Published2025
Admission routes1
Has abstractyes

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