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Record W4383503929 · doi:10.5327/1516-3180.141s1.502

Cadasil syndrome: a case report

2023· article· en· W4383503929 on OpenAlexaboutno aff
Sérgio Roberto Pereira da Silva Júnior, Isadora Chain Lima, Marcos Venâncio Araújo Ferreira, Rafael Henrique Neves Gomes, Fabiana Carla dos Santos Correia, Marina Chamon Beber, Marcus Vinicius de Sousa, Murilo Justino de Almeida, Leopoldo Antônio Pires

Bibliographic record

Venuenot available
Typearticle
Languageen
FieldMedicine
TopicCerebrovascular and genetic disorders
Canadian institutionsnot available
FundersUniversidade Federal de Uberlândia
KeywordsCADASILLeukoencephalopathyHyperintensityDementiaMedicineStroke (engine)Montreal Cognitive AssessmentAngiopathyVascular dementiaFamily historyMagnetic resonance imagingLeukoaraiosisPediatricsPathologyPsychologyInternal medicineDiseaseEndocrinologyRadiologyDiabetes mellitus

Abstract

fetched live from OpenAlex

Introduction: Cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy (CADASIL) is a genetic disease with an autosomal dominant transmission due to pathogenic variants in the NOTCH3 gene on chromosome 19. This condition causes angiopathy and is associated with high risks of strokes and vascular dementia in young adults. The present case reports a 60-year-old woman with the diagnosis of this condition after moderate cognitive impairment and advanced microangiopathy. Case report: KAO, 60-year-old, presented for evaluation after a 10-year moderate cognitive impairment, with short term memory loss without functional impairment at that point. She also referred multiple episodes of neurological deficits along the years, including vertigo and gait impairment. Neurological examination showed a wide-based gait, dismetria and disdiadococinesia and global hiperreflexia. She scored 15/30 on the Montreal Cognitive Assessment, with noted attentional deficits, memory loss and visuoespacial impairment. Family history was positive for her 62-year-old mother having history of stroke, followed by major cognitive impairment. Brain Magnetic Rrsonance Imaging showed severe white matter impairment with confluent hyperintensities — Fazekas 3, in addition to hipointensity in frontal lobes and in left side of cerebellum, suggesting hemosiderin deposition. Cardiological exams didn’t show any other significant cardiovascular risk factors. The patient was submitted to genetic testing that confirmed an atypical heterozygous pathogenic variant in NOTCH3. Conclusion: CADASIL is caused, in approximately 95% of cases, by point mutations in the NOTCH3 gene (a subtype of transmembrane receptor that acts in signaling between neighboring cells, being located in vascular muscle cells). This gene is located on chromosome 19p13.12 (OMIM #125310). The prevalence is 2:100,000, but varies in different populations. Penetrance is believed to be 100%, but it is age dependent. The severity of symptoms and disease progression are diverse, with great intra and interfamilial phenotypic variability

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)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesInsufficient payload (model declined to judge)
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 score0.040
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
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 head0.016
GPT teacher head0.288
Teacher spread0.272 · 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 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 routes1
Has abstractyes

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