Evidence map›Paper›PMID 39722056›Full record

ArticleMolecular genetics and genomics : MGG2024

Analysis of TSC1 and TSC2 genes and evaluation of phenotypic correlations with tuberous sclerosis.

Metin Eser, Gulam Hekimoglu, Busra Kutlubay, Safiye Gunes Sager, Ayberk Turkyilmaz

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Article in Molecular genetics and genomics : MGG, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

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0cells of the map it votes in
3citing papers in PubMed
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1 · What the graph read from it

What it found

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3 · Its place in the literature

Who cites it

3 citing papers in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

5 authors.

Metin EserDepartment of Medical Genetics, Umraniye Education and Research Hospital, University of Health Sciences, Istanbul, Turkey.ORCID http://orcid.org/0000-0001-7118-7958
Gulam HekimogluDepartment of Histology and Embryology, Hamidiye International Faculty of Medicine, University of Health Sciences, Istanbul, Turkey. gulam.hekimoglu@sbu.edu.tr.ORCID http://orcid.org/0000-0002-5027-6756
Busra KutlubayDepartment of Pediatrics Neurology, Umraniye Education and Research Hospital, University of Health Sciences, Istanbul, Turkey.ORCID http://orcid.org/0000-0002-6216-1438
Safiye Gunes SagerDepartment of Pediatrics Neurology, Kartal Dr. Lütfi Kirdar City Hospital, University of Health Sciences, Istanbul, Turkey.ORCID http://orcid.org/0000-0002-9876-2454
Ayberk TurkyilmazDepartment of Medical Genetics, Faculty of Medicine, Karadeniz Technical University, Trabzon, Turkey.ORCID http://orcid.org/0000-0001-9647-8970

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Tuberous sclerosis complex (TSC) is a rare genetic disorder characterized by the formation of benign tumors in various organs, particularly in the central nervous system. We aimed to delineate the molecular profile of Turkish individuals diagnosed with TSC by analyzing the TSC1 and TSC2 genes using next-generation sequencing (NGS). Sophia Genetics' Sophia Inherited Disease Panel was used to perform NGS on 22 individuals diagnosed with TSC and to identify pathogenic variants in the TSC1 and TSC2 genes. Among the 22 cases, mutations were found in 3 (13.6%) for TSC1 and in 16 (73%) for TSC2, while 3 (13.6%) exhibited no detectable mutations. Notably, one individual with a TSC2 mutation presented with angiofibroma, ungual fibroma, and pitted dental enamel, while another had cardiac rhabdomyoma. Autism spectrum disorders were observed in 6 (27%) with TSC2 mutations, including one with autistic behavior. Abnormal motor development was noted in 3 (13.6%), of which 2 had TSC2 mutations. Severe intellectual disability was found in 3 (13.6%) with TSC2 mutations, and developmental delay was seen in 2 (9%) with TSC2 mutations. Epileptic encephalopathy occurred in 3 (13.6%), with 2 having TSC2 mutations. Additionally, 6 (27%) exhibited drug resistance for focal seizures, with 5 of them having TSC2 mutations. These findings are consistent with other research indicating that TSC2 mutations are associated with a more severe phenotypic range compared to TSC1 mutations. Moreover, our analysis showed that some people with TSC1/TSC2 mutations did not match diagnostic criteria. This highlights the importance of genetic testing and molecular profiling in understanding the clinical variability and aiding in the management of TSC patients.

Indexed as

MutationPhenotypeTuberous SclerosisTuberous Sclerosis Complex 1 ProteinTuberous Sclerosis Complex 2 ProteinAdolescentAdultAutism Spectrum DisorderChildChild, PreschoolFemaleGenetic Association StudiesHigh-Throughput Nucleotide SequencingHumansInfantIntellectual DisabilityTSC1 protein, humanTSC2 protein, humanTuberous Sclerosis Complex 1 ProteinTuberous Sclerosis Complex 2 ProteinAutosomal dominantNGSTSC1TSC2Tuberous Sclerosis

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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.