TRAPPC9 gene mutations have been linked recently to autosomal recessive mental retardation 13 (MRT13; MIM#613192) with only eight families reported world-wide. We assessed patients from two consanguineous pedigrees of Pakistani descent with non-syndromic intellectual disability and postnatal microcephaly through whole exome sequencing (WES) and cosegregation analysis. Here we report six further patients from two pedigrees with homozygous TRAPPC9 gene mutations, the novel nonsense mutation c.2065G>T (p.E689*) and the previously identified nonsense mutation c.1423C>T (p.R475*). We provide an overview of previously reported clinical features and highlight common symptoms and variability of MRT13. Common findings are intellectual disability and absent speech, and frequently microcephaly, motor delay and pathological findings on MRI including diminished cerebral white matter volume are present. Mutations in TRAPPC9 should be considered in non-syndromic autosomal recessive intellectual disability with severe speech disorder.

Download full-text PDF

Source
http://dx.doi.org/10.1002/ajmg.b.32602DOI Listing

Publication Analysis

Top Keywords

intellectual disability
16
trappc9 gene
12
homozygous trappc9
8
non-syndromic intellectual
8
speech disorder
8
gene mutations
8
autosomal recessive
8
nonsense mutation
8
identification novel
4
novel homozygous
4

Similar Publications

Introduction: , a protein kinase located on human chromosome 21, plays a role in postembryonic neuronal development and degeneration. Alterations to have been consistently associated with cognitive functioning and neurodevelopmental disorders (e.g.

View Article and Find Full Text PDF

Introduction: The fact that inclusive education has existed in Bosnia and Herzegovina for twenty years opens the question of how it affects typically developing children, among other things. This paper aims to examine the differences in general knowledge and mathematics of typically developing students with regard to whether they attend classes that include students with intellectual disabilities or not, as well as to determine the relationship of their knowledge with teachers' characteristics and the inclusiveness of schools they attend.

Methods: The sample included 331 students from 18 regular elementary school classes.

View Article and Find Full Text PDF

Objective: To describe the relationship between executive functions (EF) and symptom's severity, behavioral problems, and adaptive functioning in autistic preschoolers.

Method: Seventy-six autistic preschoolers (age-range: 37-72 months; SD: 8.67 months) without intellectual disability were assessed.

View Article and Find Full Text PDF

Novel p.Arg534del Mutation and MTHFR C667T Polymorphism in Fragile X Syndrome (FXS) With Autism Spectrum Phenotype: A Case Report.

Case Rep Genet

January 2025

Medical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California, 2825 50th Street, Davis, Sacramento 95817, California, USA.

Fragile X syndrome (FXS) presents with autism spectrum disorder (ASD), intellectual disability, developmental delay, seizures, hypotonia during infancy, joint laxity, behavioral issues, and characteristic facial features. The predominant mechanism is due to CGG trinucleotide repeat expansion of more than 200 repeats in the 5'UTR (untranslated region) of (Fragile X Messenger Ribonucleoprotein 1) causing promoter methylation and transcriptional silencing. However, not all patients presenting with the characteristic phenotype and point/frameshift mutations with deletions in have been described in the literature.

View Article and Find Full Text PDF

Background: Child maltreatment is a significant social problem impacting both health and society, with severe and enduring consequences. Certain children, such as those with neurodevelopmental conditions like autism, may be more at risk of experiencing maltreatment. However, little research has examined the characteristics of these children.

View Article and Find Full Text PDF

Want AI Summaries of new PubMed Abstracts delivered to your In-box?

Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!