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Record W2425867392 · doi:10.1017/cjn.2016.156

P.052 Late-onset adrenoleukodystrophy mimicking primary progressive multiple sclerosis

2016· article· en· W2425867392 on OpenAlexvenueno aff
AJ Schabas, Ana‐Luiza Sayao

Bibliographic record

VenueCanadian Journal of Neurological Sciences / Journal Canadien des Sciences Neurologiques · 2016
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicPeroxisome Proliferator-Activated Receptors
Canadian institutionsnot available
Fundersnot available
KeywordsMedicineMyelopathyAmyotrophic lateral sclerosisAdrenoleukodystrophyHyperintensityMultiple sclerosisSpinal cordAtrophyFamily historyMagnetic resonance imagingPediatricsPhysical medicine and rehabilitationDiseasePathologyRadiologyInternal medicinePeroxisomePsychiatry

Abstract

fetched live from OpenAlex

Background: Adrenoleukodystrophy (ALD) is a peroxisomal disorder that leads to the accumulation of very long chain fatty acids in the body. Younger males typically present with a catastrophic cerebral demyelinating disease, while adult males tend to develop a progressive myelopathy and neuropathy. Methods: Case presentations and literature review. Results:Case1: A 58-year-old male with a three-year history of unsteady gait. His MRI showed multiple T2-hyperintensities most prominently in the posterior corpus collosum (which progressed over time) as well as spinal cord atrophy. Primary progressive multiple sclerosis (PPMS) was suspected. Case 2: The patient’s bother, a 49-year-old, had a ten-year history of difficulty walking. MRI findings included a single large T2 hyper-intensity spanning the anterior aspect of the corpus collosum and an atrophic spinal cord. Given the family history, both brothers were investigated for and diagnosed with ALD. Conclusions: Both cases are of males presenting with a progressive myelopathy in middle age. In the first case, the history, physical exam, and imaging findings were most consistent with PPMS. However, the second case was less typical for MS prompting further investigations. These cases highlight the need to have a broad differential when confronted with atypical cases of MS and reminds the clinician of the phenotypic variability of ADL.

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 score0.011
Threshold uncertainty score0.036

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0110.004

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 head0.028
GPT teacher head0.237
Teacher spread0.209 · 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
Published2016
Admission routes1
Has abstractyes

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