MétaCan
Menu
Back to cohort
Record W4404769316 · doi:10.1016/j.rare.2024.100052

An expansion of the phenotype in individuals with SYNCRIP-Related Neurodevelopmental Disorder

2024· article· en· W4404769316 on OpenAlexaff
Tooba Shafiq, Lindsay A. Phillips, Kara Murias, Marcia Ferguson, Kristin Barañano, Alaina Acchione, Patricia Kipkemoi, Collins Kipkoech, Eunice Chepkemoi, Amina Abubakar, Charles R. Newton, Celia van der Merwe, Emily O’Heir, Alice Galvin, Aixa Gonzalez Garcia, Jennifer Stefanich, Amelle Shillington, Annabelle Tuttle, Erin Torti, Elen Zhu, Margaretha AJ Morsink, Ekaterina Lebayle, Barbara Corneo, Christopher Ricupero, Ping Yee Billie Au, Antonie D. Kline, Meena Balasubramanian, Jennifer Bain, Madelyn A. Gillentine

Bibliographic record

VenueRare · 2024
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetics and Neurodevelopmental Disorders
Canadian institutionsAlberta Children's Hospital
FundersMedical Research CouncilNational Institutes of Health
KeywordsNeurodevelopmental disorderPhenotypePsychologyMedicinePsychiatryGeneticsBiologyAutismGene

Abstract

fetched live from OpenAlex

Disruption of genes within the HNRNP gene family has been observed in neurodevelopmental and neurodegenerative diseases . The HNRNP-Related Neurodevelopmental Disorders (HNRNP-RNDDs), while each unique, have been recently described with similar clinical and molecular features across variation in several genes. However, the phenotypic information on these patients is still lacking. In this case series we aim to describe the phenotypes that are associated with SYNCRIP-Related Neurodevelopmental Disorder (SYNCRIP-RNDD). We describe in depth ten novel individuals and one previously published individual with mostly de novo and predicted damaging variants in SYNCRIP , consistent with a diagnosis of SYNCRIP-RNDD. We also describe previously published patients, many of which are from large cohort studies , as well as individuals from patient databases. Here, we expand the phenotype of SYNCRIP-RNDD beyond a generic neurodevelopmental disorder to a variable syndrome consisting of mild to borderline developmental delay/intellectual disability, speech and language delay, behavioral differences such as autism spectrum disorder , structural brain anomalies , hypotonia , and seizures . Inconsistent dysmorphic features were also observed, with the few recurrent findings including long eyelashes , mildly deep-set eyes, prominent ears, and thin or thick lips. This study increases our understanding of SYNCRIP-RNDD, as well as HNRNP-RNDDs broadly.

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)0.001
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.011

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.001
Scholarly communication0.0000.000
Open science0.0000.001
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0030.001

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 head0.005
GPT teacher head0.214
Teacher spread0.210 · 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 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

Citations4
Published2024
Admission routes1
Has abstractyes

Explore more

Same venueRareSame topicGenetics and Neurodevelopmental DisordersFrench-language works237,207