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Arch Iran Med. 2022;25(12): 788-797.
doi: 10.34172/aim.2022.124
PMID: 37543906
PMCID: PMC10685845
Scopus ID: 85158825141
  Abstract View: 1170
  PDF Download: 612

Original Article

Characterizing Genotypes and Phenotypes Associated with Dysfunction of Channel-Encoding Genes in a Cohort of Patients with Intellectual Disability

Naeim Ehtesham 1 ORCID logo, Meysam Mosallaei 1, Maryam Beheshtian 1 ORCID logo, Shahrouz Khoshbakht 1, Mahsa Fadaee 2, Raheleh Vazehan 2, Mehrshid Faraji Zonooz 2, Parvaneh Karimzadeh 3 ORCID logo, Kimia Kahrizi 1, Hossein Najmabadi 1,2* ORCID logo

1 Genetics Research Center, University of Social Welfare and Rehabilitation Sciences, Tehran, Iran
2 Kariminejad – Najmabadi Pathology & Genetics Center, Tehran, Iran
3 Department of Pediatric Neurology, School of Medicine, Pediatric Neurology Research Center, Mofid Children’s Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran
*Corresponding Author: Hossein Najmabadi,, Email: hnajm12@yahoo.com

Abstract

Background: Ion channel dysfunction in the brain can lead to impairment of neuronal membranes and generate several neurological diseases, especially neurodevelopmental disorders.

Methods: In this study, we set out to delineate the genotype and phenotype spectrums of 14 Iranian patients from 7 families with intellectual disability (ID) and/or developmental delay (DD) in whom genetic mutations were identified by next-generation sequencing (NGS) in 7 channel-encoding genes: KCNJ10, KCNQ3, KCNK6, CACNA1C, CACNA1G, SCN8A, and GRIN2B. Moreover, the data of 340 previously fully reported ID and/or DD cases with a mutation in any of these seven genes were combined with our patients to clarify the genotype and phenotype spectrum in this group.

Results: In total, the most common phenotypes in 354 cases with ID/DD in whom mutation in any of these 7 channel-encoding genes was identified were as follows: ID (77.4%), seizure (69.8%), DD (59.8%), behavioral abnormality (29.9%), hypotonia (21.7%), speech disorder (21.5%), gait disturbance (20.9%), and ataxia (20.3%). Electroencephalography abnormality (33.9%) was the major brain imaging abnormality.

Conclusion: The results of this study broaden the molecular spectrum of channel pathogenic variants associated with different clinical presentations in individuals with ID and/or DD.


Cite this article as: Ehtesham N, Mosallaei M, Beheshtian M, Khoshbakht S, Fadaee M, Vazehan R, et al. Characterizing genotypes and phenotypes associated with dysfunction of channel-encoding genes in a cohort of patients with intellectual disability. Arch Iran Med. 2022;25(12):788-797. doi: 10.34172/aim.2022.124
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Submitted: 03 Oct 2021
Revision: 27 Nov 2021
Accepted: 20 Dec 2021
ePublished: 01 Dec 2022
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