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Record W4406627930 · doi:10.1136/jmg-2024-110388

Under-recognised neurocognitive deficits in adults and adolescents with tuberous sclerosis complex

2025· article· en· W4406627930 on OpenAlexaffabout
Mélissa Boisclair, Laury-Anne Blondeau, Florence Bouchard, Rose‐Marie Drouin‐Engler, Jimmy Li, Fayçal Zine-Eddine, Laurent Létourneau‐Guillon, Catherine Larochelle, Philippe Major, Olivier Boucher, Mark R. Keezer

Bibliographic record

VenueJournal of Medical Genetics · 2025
Typearticle
Languageen
FieldMedicine
TopicTuberous Sclerosis Complex Research
Canadian institutionsCentre Hospitalier Universitaire Sainte-JustineCentre Hospitalier de l’Université de MontréalCentre Hospitalier Universitaire de SherbrookeUniversité de SherbrookeUniversité de Montréal
Fundersnot available
KeywordsTuberous sclerosisNeurocognitiveMedicinePsychologyDevelopmental psychologyClinical psychologyPsychiatryCognition

Abstract

fetched live from OpenAlex

BACKGROUND: Tuberous sclerosis complex (TSC) is a genetic disease characterised by the growth of benign tumours. The Tuberous sclerosis Associated Neuropsychiatric Disorders (TAND) Checklist is used to identify patient-reported neurocognitive deficits. Patients may, however, under-recognise mild cognitive impairment. We aimed to determine the frequency of abnormal scores on three objective tests of cognitive function in people with and without diagnosed intellectual disability and examine associations between scores on these tests with self-reported TAND Checklist symptoms. METHODS: We conducted a cross-sectional study where people with TSC (PwTSC; n=46) completed the TAND Checklist and three cognitive tests: Symbol Digit Modalities Test (SDMT), Montreal Cognitive Assessment test and Trail Making Test-Parts A and B. We examined associations between cognitive test scores and the TAND Checklist using Pearson's correlations (95% CI). Receiver operating characteristics (ROC) curves were plotted to determine the screening accuracy of each measure in identifying physician-diagnosed neurocognitive disorders. RESULTS: There were minimal correlations between the cognitive test scores and the TAND Checklist. More than 20% of PwTSC reported no cognitive issues on the TAND Checklist but had abnormal performance on at least one cognitive test. The ROC curves demonstrated similar results, with areas under the curve of 0.93 (95% CI 0.79 to 1.00) for the SDMT but only 0.70 (95% CI 0.45 to 0.95) for the TAND Checklist. CONCLUSION: Objective tests of cognitive function are useful in identifying unrecognised neurocognitive deficits in PwTSC. Deficits likely have multifactorial origins, including undiagnosed intellectual disability and the impact of chronic epilepsy.

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)0.001
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: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.316
Threshold uncertainty score0.600

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.001
Science and technology studies0.0000.000
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 head0.054
GPT teacher head0.322
Teacher spread0.268 · 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.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
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

Citations1
Published2025
Admission routes2
Has abstractyes

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