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Record W7092359303 · doi:10.17863/cam.122361

A novel hereditary encephalopathy in four related Labrador Retrievers associated with a missense variant in the ALDH5A1 gene.

2025· article· en· W7092359303 on OpenAlexaboutno aff

Bibliographic record

VenueApollo (University of Cambridge) · 2025
Typearticle
Languageen
FieldNeuroscience
TopicNeurological diseases and metabolism
Canadian institutionsnot available
Fundersnot available
KeywordsMissense mutationEncephalopathyDiseaseGenotypingCandidate geneAbnormality

Abstract

fetched live from OpenAlex

BACKGROUND: Hereditary neurodegenerative diseases occur in dogs, and a molecular diagnosis can be of value for treatment and prevention. HYPOTHESIS/OBJECTIVES: To describe the clinical presentation of a novel encephalopathy in 4 related Labrador Retrievers. To identify a candidate causal variant for the disease using whole genome sequencing (WGS). ANIMALS: Four related Labrador Retrievers presenting between 4 and 9 months of age. METHODS: Case information and clinical workup were recorded for the 4 dogs in this case series. Two cases underwent WGS to identify candidate causal variants that were validated by genotyping Labradors related and unrelated to the cases, and by screening WGS of other breeds/canid species. RESULTS: Clinical signs in cases included paroxysmal anxiety episodes, and focal and generalized epileptic seizures. Interictal clinical and neurological examinations were normal in all cases. Magnetic resonance imaging of the brain documented bilaterally symmetrical, T2-weighted image hyperintense, T1-weighted image isointense, and non-contrast-enhancing lesions within the lentiform nuclei, caudal colliculi, substantia nigra, and cerebellar nuclei. Investigations to exclude underlying nutritional, toxic, and metabolic causes were within normal limits. All cases had 2 copies of a missense variant in the aldehyde dehydrogenase 5 family member A1 (ALDH5A1) gene that segregated as expected in the family group and was absent in 70 unrelated Labradors and 2339 WGS of multiple breeds/canids. CONCLUSIONS AND CLINICAL IMPORTANCE: The prognosis of this novel hereditary encephalopathy in a family of Labradors appears fair with reasonable clinical response to administration of anti-seizure drugs. A missense variant in ALDH5A1 has been identified as a candidate causal variant for the disease in these dogs.

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)0.000
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation 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 score0.960
Threshold uncertainty score0.513

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.002
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

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 head0.016
GPT teacher head0.201
Teacher spread0.185 · 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.

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

Citations0
Published2025
Admission routes1
Has abstractyes

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