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Record W4400588491 · doi:10.21275/sr24531200433

A Rare Case of Autosomal Recessive Spastic Ataxia of Charlevoix - Saguenay

2024· article· en· W4400588491 on OpenAlexaboutno aff
Dhruvkumar Vasantbhai Patel, Sagar Nileshbhai Parmar, Dhruvi Kalpesh Joshi, Pratik D Modi

Bibliographic record

VenueInternational Journal of Science and Research (IJSR) · 2024
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsnot available
Fundersnot available
KeywordsAtaxiaMedicineGeneticsBiologyNeuroscience

Abstract

fetched live from OpenAlex

Background: Autosomal recessive spastic ataxia of Charlevoix -Saguenay (ARSACS or SACS) is an early onset neurodegenerative disease with high prevalence (carrier frequency 1/22) in the Charlevoix -Saguenay -Lac -Saint -Jean (CSLSJ) region of Quebec. It is caused by mutations in SACS gene on chromosome 13 which encodes the protein Sacsin. It is characterized by triad of spasticity, cerebellar symptoms and sensory and motor polyneuropathy. Clinical Case: A 29 years old unmarried female born out of non -consanguineous marriage presented to the hospital with a history of difficulty in maintaining balance while walking since 17 years, dysarthria and stiffness in lower limb. She had no significant drug history or other medical illness. There was history of intentional tremors in father since 3 years. Her mother died at the age of 40 due to some neurological issue with unknown cause. On neurological examination, there was all four limbs spasticity, deep tendon reflexes were exaggerated, with bilateral extensor plantar response. Examination also revealed dysmetria, dysdiadochokinesia and abnormal knee -heel -shin test on left side. The patient was swaying from side to side with open eyes and her feet positioned opposite each other. Romberg's test was negative. There were no significant findings on systemic examination. Fundus and retinal examination were normal. Her routine blood works were within normal limits. On MRI brain imaging, there were areas of edema involving the bulky pons and striped Hypointensities with mild cerebral and moderate cerebellar atrophic changes. Nerve conduction study indicated axonal demyelinating polyneuropathy, involving sensory nerves more than motor nerves, and affecting lower limbs more than upper limbs. Conclusion: The diagnosis of Autosomal Recessive Spastic Ataxia of Charlevoix -Saguenay was established based on clinical manifestations, radiological imaging and nerve conduction studies. Index of suspicion should be higher for early diagnosis in young patients with gait ataxia and spasticity with cerebellar atrophy in the brain imaging. treatment in this patient was focused on symptomatic relief and supportive care including physical and occupational therapy.

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)0.002
metaresearch head score (Gemma)0.005
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesScience and technology studies
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.005
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0020.005
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.003
Scholarly communication0.0000.001
Open science0.0010.000
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 head0.077
GPT teacher head0.408
Teacher spread0.331 · 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.

Study designBench or experimental
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
Published2024
Admission routes1
Has abstractyes

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