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Record W4226335934 · doi:10.1212/wnl.0000000000200603

Teaching Video NeuroImage: Peculiar Hobby Horse Gait in Huntington Disease–like 2

2022· article· en· W4226335934 on OpenAlexaff

Bibliographic record

VenueNeurology · 2022
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsCentre for Movement Disorders
Fundersnot available
KeywordsChoreaApathyAthetosisGait DisturbanceHuntington's diseaseHobbyGaitMovement disordersPhenocopy

Abstract

fetched live from OpenAlex

A 59-year-old woman presented at age 48 years with abnormal hand movements, which became generalized 1 year later, along with a clumsy gait. Two years later, severe apathy and minor auditory hallucinations ensued. Her father died undiagnosed with involuntary movements at age 82 years, and 2 of her 6 sisters have also had a similar clinical picture. Neurologic examination revealed generalized chorea and an unusual dystonic gait pattern remarkably similar to the hobby horse gait, described in DYT-TUBB4A (DYT-4), which is characterized by a toe walking stiff-legged and skipping gait1 (Video 1). Walking backward improved the gait. Diagnostic workup revealed acanthocytes and mild caudate atrophy (Figure 1). A pathogenic 46 CTG/CAG repeat expansion on the JPH3 gene allowed the diagnosis of Huntington disease–like 2, an autosomal dominant Huntington disease phenocopy characterized by chorea, dystonia, and parkinsonism.2 This case expands underlying conditions recognized to cause the HHG (please see the Supplement, [links.lww.com/WNL/B931][1]). [1]: http://links.lww.com/WNL/B931

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)0.001
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 score0.022
Threshold uncertainty score0.073

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0020.001
Science and technology studies0.0010.001
Scholarly communication0.0010.001
Open science0.0010.001
Research integrity0.0040.002
Insufficient payload (model declined to judge)0.0220.005

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 head0.018
GPT teacher head0.262
Teacher spread0.244 · 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

Citations8
Published2022
Admission routes1
Has abstractyes

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