NONO-related X-linked intellectual disability syndrome: Further clinical and molecular delineation

The X-linked NONO gene encodes Non-Pou Domain-Containing Octamer-Binding Protein, a multifunctional member of the DBHS family involved in transcriptional regulation, RNA splicing and DNA repair. Pathogenic variants in NONO cause Intellectual Developmental Disorder, X-linked Syndromic (MIM #300967), characterised by intellectual disability, neurodevelopmental delay, cardiomyopathy, such as left ventricular non-compaction (LVNC), and congenital heart defects such as including atrial septal defect (ASD), ventricular septal defect (VSD), patent ductus arteriosus (PDA), and patent foramen ovale (PFO). This study reports three new patients with pathogenic hemizygous frameshift variants in NONO identified with exome sequencing, broadening the clinical presentation. The patients present with neurodevelopmental delay, macrocephaly, agenesis or hypoplasia of the corpus callosum and LVNC, confirming previous findings. These findings contribute to the understanding of the phenotypic diversity in patients with NONO pathogenic variants and highlight the need for further investigation of genotype-phenotype correlations, particularly with regard to early cardiac development, and prenatal presentations.

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Publication Details

Journal
European Journal of Medical Genetics
Published
2024-12-20
DOI
https://doi.org/10.1016/j.ejmg.2024.104987
Citations
3
Primary Topic
Genetics and Neurodevelopmental Disorders
Type
article
Field-Weighted Citation Impact
1.65
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article

NONO-related X-linked intellectual disability syndrome: Further clinical and molecular delineation

Odile Boute‐Bénéjean, Roseline Caumes, Mélanie Rama, Emilie Ait‐Yahya et al.
3 citations
European Journal of Medical Genetics
Genetics and Neurodevelopmental Disorders
1.65
article

NONO-related X-linked intellectual disability syndrome: Further clinical and molecular delineation

Odile Boute‐Bénéjean, Roseline Caumes, Mélanie Rama, Emilie Ait‐Yahya, C. Thuillier, Jamal Ghoumid, Simon Boussion, Thomas Smol, Catherine Vincent‐Delorme, Pauline Planté‐Bordeneuve, Bruno Delobel
article en
3 citations

Abstract

The X-linked NONO gene encodes Non-Pou Domain-Containing Octamer-Binding Protein, a multifunctional member of the DBHS family involved in transcriptional regulation, RNA splicing and DNA repair. Pathogenic variants in NONO cause Intellectual Developmental Disorder, X-linked Syndromic (MIM #300967), characterised by intellectual disability, neurodevelopmental delay, cardiomyopathy, such as left ventricular non-compaction (LVNC), and congenital heart defects such as including atrial septal defect (ASD), ventricular septal defect (VSD), patent ductus arteriosus (PDA), and patent foramen ovale (PFO). This study reports three new patients with pathogenic hemizygous frameshift variants in NONO identified with exome sequencing, broadening the clinical presentation. The patients present with neurodevelopmental delay, macrocephaly, agenesis or hypoplasia of the corpus callosum and LVNC, confirming previous findings. These findings contribute to the understanding of the phenotypic diversity in patients with NONO pathogenic variants and highlight the need for further investigation of genotype-phenotype correlations, particularly with regard to early cardiac development, and prenatal presentations.

European Journal of Medical GeneticsVol. 73
Université de Lille (FR), Centre Hospitalier Universitaire de Lille (FR), Groupe Hospitalier de l'Institut Catholique de Lille (FR), Institut de Biologie de Lille (FR)
Reduced inequalities
Openalex Percentile: Top 14%
Genetics and Neurodevelopmental Disorders
1.65
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NONO-related X-linked intellectual disability syndrome: Further clinical and molecular delineation — Odile Boute‐Bénéjean, Roseline Caumes, et al. · European Journal of Medical Genetics (2024) | TGRS Research Map | TGRS