Evidence map›Paper›PMID 40019509›Full record

ArticleHuman genetics2025

Genetic variants and phenotypic data curated for the CAGI6 intellectual disability panel challenge.

Maria Cristina Aspromonte, Alessio Del Conte, Roberta Polli, Demetrio Baldo, Francesco Benedicenti, Elisa Bettella, Stefania Bigoni, Stefania Boni, Claudia Ciaccio, Stefano D'Arrigo and 13 more

Abstract read
In one paragraph

Article in Human genetics, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

5 citing papers in PubMed.

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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

23 authors.

Maria Cristina AspromonteDepartment of Biomedical Sciences, University of Padova, Padua, Italy.
Alessio Del ConteDepartment of Biomedical Sciences, University of Padova, Padua, Italy.
Roberta PolliMolecular Genetics of Neurodevelopment, Department of Woman and Child Health, University of Padova, Padua, Italy.
Demetrio BaldoMedical Genetics Unit, Treviso Hospital, Treviso, Italy.
Francesco BenedicentiGenetic Counseling Service, Regional Hospital of Bolzano, Bolzano, Italy.
Elisa BettellaMolecular Genetics of Neurodevelopment, Department of Woman and Child Health, University of Padova, Padua, Italy.
Stefania BigoniMedical Genetics Unit, Ferrara University Hospital, Ferrara, Italy.
Stefania BoniMedical Genetics Unit, S. Martino Hospital, Belluno, Italy.
Claudia CiaccioDepartment of Pediatric Neurosciences, Fondazione IRCCS Istituto Neurologico "Carlo Besta", Milan, Italy.
Stefano D'ArrigoDepartment of Pediatric Neurosciences, Fondazione IRCCS Istituto Neurologico "Carlo Besta", Milan, Italy.
Ilaria DonatiUnit of Medical Genetics, AUSL Romagna, Cesena, Italy.
Elisa GranocchioDepartment of Pediatric Neurosciences, Fondazione IRCCS Istituto Neurologico "Carlo Besta", Milan, Italy.
Isabella MammiMedical Genetics Unit, Mirano Hospital, Venice, Italy.
Donatella MilaniFondazione IRCCS, Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.
Susanna NegrinScientific Institute, IRCCS E. Medea, dipartimento/Unità Operativa Conegliano, Treviso, Italy.
Margherita NosadiniPaediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padova, Padua, Italy.
Fiorenza SoliGenetic Unit, UOM Patologia Clinica, S. Chiara Hospital of Trento, Trento, Italy.
Franco StanzialGenetic Counseling Service, Regional Hospital of Bolzano, Bolzano, Italy.
Licia TurollaMedical Genetics Unit, Treviso Hospital, Treviso, Italy.
Damiano PiovesanDepartment of Biomedical Sciences, University of Padova, Padua, Italy.
Silvio C E TosattoDepartment of Biomedical Sciences, University of Padova, Padua, Italy.
Alessandra MurgiaMolecular Genetics of Neurodevelopment, Department of Woman and Child Health, University of Padova, Padua, Italy.
Emanuela LeonardiDepartment of Biomedical Sciences, University of Padova, Padua, Italy. emanuela.leonardi@unipd.it.

Funding

European Reference Network on Rare Congenital Malformations and Rare Intellectual Disability 3HP-HP-FPA ERN-01-2016/739516European Union-NextGenerationEU through 'Italiadomani-PNRR' project CN - G.T.RNA SP. 7 project - National Center for Gene Therapy and Drugs based on RNA Technology (CN3) MUR CN00000041, C.F. 92315700283
6 · The paper itself

Abstract

Neurodevelopmental disorders (NDDs) are common conditions including clinically diverse and genetically heterogeneous diseases, such as intellectual disability, autism spectrum disorders, and epilepsy. The intricate genetic underpinnings of NDDs pose a formidable challenge, given their multifaceted genetic architecture and heterogeneous clinical presentations. This work delves into the intricate interplay between genetic variants and phenotypic manifestations in neurodevelopmental disorders, presenting a dataset curated for the Critical Assessment of Genome Interpretation (CAGI6) ID Panel Challenge. The CAGI6 competition serves as a platform for evaluating the efficacy of computational methods in predicting phenotypic outcomes from genetic data. In this study, a targeted gene panel sequencing has been used to investigate the genetic causes of NDDs in a cohort of 415 paediatric patients. We identified 60 pathogenic and 49 likely pathogenic variants in 102 individuals that accounted for 25% of NDD cases in the cohort. The most mutated genes were ANKRD11, MECP2, ARID1B, ASH1L, CHD8, KDM5C, MED12 and PTCHD1 The majority of pathogenic variants were de novo, with some inherited from mildly affected parents. Loss-of-function variants were the most common type of pathogenic variant. In silico analysis tools were used to assess the potential impact of variants on splicing and structural/functional effects of missense variants. The study highlights the challenges in variant interpretation especially in cases with atypical phenotypic manifestations. Overall, this study provides valuable insights into the genetic causes of NDDs and emphasises the importance of understanding the underlying genetic factors for accurate diagnosis, and intervention development in neurodevelopmental conditions.

Indexed as

Genetic Predisposition to DiseaseGenetic VariationIntellectual DisabilityNeurodevelopmental DisordersChildChild, PreschoolDatabases, GeneticFemaleHumansMaleMutationPhenotype

Identifiers

PMID40019509
PMCPMC11976335

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.