The results of CNVs analysis in patients with neurodevelopmental delay, dysmorphic features and congenital defects

Array Comparative Genomic Hybridization (aCGH) enables genome-wide detection of copy number variants (CNVs), representing a major advance in diagnosing neurodevelopmental disorders and congenital anomalies. We performed aCGH in 1,468 patients referred for genetic evaluation due to neurodevelopmental delay, dysmorphic features, and congenital malformations. Pathogenic CNVs were detected in 24.5% of patients. The highest frequency of unbalanced aberrations occurred on chromosomes X, 15, 1, and 7. Most CNVs were small (< 10 Mbp), though entire-chromosome duplications (notably on 8, 9, and X) were also observed. The diagnostic yield and CNV distribution differed between prenatal and postnatal analyses, with deletions predominating in prenatal cases and a balanced ratio of deletions to duplications postnatally. The study confirms the high diagnostic utility of aCGH in patients with complex developmental phenotypes and highlights the importance of integrating cytogenetic analysis and periodic genomic data reinterpretation to refine diagnosis and counselling.

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Publication Details

Journal
Journal of Applied Genetics
Published
2026-06-16
DOI
https://doi.org/10.1007/s13353-026-01080-9
Primary Topic
Fetal and Pediatric Neurological Disorders
Type
article
Field-Weighted Citation Impact
0.00

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article

The results of CNVs analysis in patients with neurodevelopmental delay, dysmorphic features and congenital defects

Paweł Skiba, Robert Śmigiel, Elżbieta Szmida, Izabela Łaczmańska et al.
Journal of Applied Genetics
Fetal and Pediatric Neurological Disorders
article

The results of CNVs analysis in patients with neurodevelopmental delay, dysmorphic features and congenital defects

Paweł Skiba, Robert Śmigiel, Elżbieta Szmida, Izabela Łaczmańska, Agnieszka Pawelak, Ryszard Ślężak, Justyna Gil, Agnieszka Stembalska, Gabriela Gardas, Aleksandra Jakubiak, Maria Sąsiadek, Marta Rowińska, Izabella Jabłońska
article en

Abstract

Array Comparative Genomic Hybridization (aCGH) enables genome-wide detection of copy number variants (CNVs), representing a major advance in diagnosing neurodevelopmental disorders and congenital anomalies. We performed aCGH in 1,468 patients referred for genetic evaluation due to neurodevelopmental delay, dysmorphic features, and congenital malformations. Pathogenic CNVs were detected in 24.5% of patients. The highest frequency of unbalanced aberrations occurred on chromosomes X, 15, 1, and 7. Most CNVs were small (< 10 Mbp), though entire-chromosome duplications (notably on 8, 9, and X) were also observed. The diagnostic yield and CNV distribution differed between prenatal and postnatal analyses, with deletions predominating in prenatal cases and a balanced ratio of deletions to duplications postnatally. The study confirms the high diagnostic utility of aCGH in patients with complex developmental phenotypes and highlights the importance of integrating cytogenetic analysis and periodic genomic data reinterpretation to refine diagnosis and counselling.

Journal of Applied Genetics
Wrocław University of Science and Technology (PL), Wroclaw Medical University (PL), AGH University of Krakow (PL)
Uniwersytet Medyczny im. Piastów Slaskich we Wroclawiu
Openalex Percentile: Top 6%
Fetal and Pediatric Neurological Disorders
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