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Case Report: Gait Deterioration in a Patient with EAST (Epilepsy, Ataxia, Sensorineural Deafness, Tubulopathy) Syndrome

2023· article· en· W4384929256 on OpenAlexaffvenue
Gökce Hatipoglu Majernik, J. Peter Martin

Bibliographic record

VenueJournal of Intellectual Disability - Diagnosis and Treatment · 2023
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsWestern University
Fundersnot available
KeywordsMedicineAtaxiaMyelopathyGait AtaxiaSensorineural hearing lossSpasticityEpilepsyPediatricsHearing lossSurgerySpinal cordAudiologyPhysical medicine and rehabilitationPsychiatry

Abstract

fetched live from OpenAlex

Epilepsy, Ataxia, Sensorineural deafness, Tubulopathy (EAST) syndrome or seizures, sensorineural deafness, ataxia, mental retardation (SeSAME) is a very rare, autosomal recessive disorder resulting from homozygous or compound heterozygous loss-of-function variations in the KCNJ10 gene, affecting <1:1.000.000 individuals. Ambulation problems, such as ambulation delays and ataxia, are common in this syndrome. The disorder can remain undiagnosed in adults due to its rarity. Here, we describe the case of a patient with genetically proven EAST syndrome at the age of 18. The patient experienced a dramatic deterioration in his walking ability 7 years after the diagnosis when he was 25 years old. However, the main reason for gait deterioration was cervical spinal cord compression by cervical disc prolapse which was determined through magnetic resonance imaging during further examinations. Cervical myelopathy caused increased spasticity and gait deterioration in this patient. He underwent single-level anterior cervical decompression and fusion without any complication and improved postoperatively. Although worsening neurological findings in patients with EAST syndrome have been reported, in-depth clinical, radiological, and laboratory examinations can clarify the increasing deterioration in motor functions. Concomitant pathologies can occur, which should be kept in mind for better diagnosis and treatment, especially for patients with EAST syndrome.

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)0.002
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 score0.005
Threshold uncertainty score0.008

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0020.001
Science and technology studies0.0020.002
Scholarly communication0.0010.001
Open science0.0010.001
Research integrity0.0050.003
Insufficient payload (model declined to judge)0.0010.001

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 head0.053
GPT teacher head0.277
Teacher spread0.224 · 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
Published2023
Admission routes2
Has abstractyes

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