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Record W4402076547 · doi:10.3988/jcn.2024.0086

Myotonic Dystrophy Type 1 With Cerebellar Ataxia and Cerebellar Atrophy

2024· article· en· W4402076547 on OpenAlex

Why this work is in the frame

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

aboutThe title or abstract carries a Canadian signal from the geographic lexicon.
no affNo Canadian affiliation: this work is invisible to an affiliation-only frame.
No Canadian affiliation. An affiliation-only frame, the usual design, would never have seen this work. It is one of the works that make the case for inverting the frame.

Bibliographic record

VenueJournal of Clinical Neurology · 2024
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsnot available
FundersNational Natural Science Foundation of China
KeywordsMyotonic dystrophyCerebellar ataxiaMedicineCerebellumAtrophyAtaxiaNeurosciencePsychologyPathologyInternal medicine

Abstract

fetched live from OpenAlex

Dear Editor, Myotonic dystrophy (DM) is a dominantly inherited muscular dystrophy involving multiple systems. 1 We report a case with cerebellar ataxia and cerebellar atrophy that was finally diagnosed as DM type 1 (DM1).A 50-year-old female presented with a 4-year history of progressive gait imbalance and slurred speech.She had been diagnosed with diabetes about 20 years previously, and had cataracts for 8 years.She developed grip myotonia at the age of 20 years.Her younger brother had diabetes for about 10 years and had been walking unsteadily for almost 2 years.Our patient presented with bilateral temporal muscle atrophy, frontal alopecia, and severe gait ataxia requiring support.A neurological examination revealed dysarthria and weakness of bilateral eyelids and neck flexion.Nystagmus was absent.Bilateral hyperalgesia and reduced vibratory sensation were found distal to the ankle joints.Light-touch and position senses were intact.The strengths of bilateral wrist flexion, bilateral grip and finger extension/flexion, bilateral plantar dorsiflexion, and all proximal limbs were grades 4+, 4, 4, and 5, respectively.Her tendon reflexes and muscle tone were decreased.A negative Romberg sign and clumsy rapidly alternating hand movements were observed.Bilateral finger-to-nose and heel-to-shin tests were abnormal.No pyramidal sign was observed.Magnetic resonance imaging (MRI) demonstrated high-intensity T2-weighted lesions in the white matter of the temporal pole and the periventricular region (Fig. 1A).MRI also showed prominent cerebellar atrophy, which had deteriorated from 2018 to 2022 (Fig. 1B-D).Consistent with the MRI findings, in 2018 the patient starting developing unsteadiness and slurred speech, but she could still walk with assistance and engage in communication.However, by 2022 she had lost the ability to walk and communicate.The Mini-Mental State Examination (19 points) and Montreal Cognitive Assessment (12 points) revealed impaired cognition.A nerve conduction velocity study revealed polyneuropathy mainly involving the sensory and motor axons.Electromyography indicated myogenic damage and myotonia potentials in multiple muscles.Electronystagmography revealed damage to the cerebellum and its related fibers.Genetic sequencing revealed the abnormal expansion of a CTG triplet repeat in the noncoding region of the DMPK gene (135 copies).However, we could not obtain genetic data for her younger brother.Based on genetic findings, the patient was diagnosed with DM1.Manifestations supporting a diagnosis of multiple-system atrophy (MSA) or other neurodegenerative diseases were not found.Whole-exome sequencing did not reveal other pathogenic mutations.Repeat expansions associated with spinocerebellar ataxia were examined in genes including ATXN1, ATXN2, ATXN3, CACNA1A, ATXN7, ATXN8, PPP2R2B, TBP, ATN1, FXN, ATXN10, and NOP56, but no abnormality was found.Neuroimaging abnormalities of DM1 are mainly found in the frontal and anterior temporal lobes, including cortical atrophy and subcortical white-matter hyperintensities. 2,

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.

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: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.513
Threshold uncertainty score0.646

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.000
Science and technology studies0.0000.001
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
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 head0.050
GPT teacher head0.349
Teacher spread0.299 · 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