Assesment of pathogenic variants in the PAH gene and genotype-phenotype correlation in Phenylketonuria patients from Turkey.

Author:

Balasar Özgür1,YILMAZ Banu KADIOĞLU2,BAŞDEMİRCİ Müşerref1,EKER Hatice KOÇAK1,ÇAVDARTEPE Büşra ESER1,ŞİMŞEK Levent1,TUNÇEZ Ebru1,DUYMUŞ Fahrettin1

Affiliation:

1. konya city hospital

2. Selçuk University

Abstract

Abstract

This study aims to determine the allele and genotype frequency, evaluate genotype-phenotype correlation, and contribute to the spectrum of pathogenic variants in the PAH gene. Ninety-three individuals diagnosed with PKU were included in the study. Next-generation sequencing was utilized for detecting variants in the PAH gene. Copy Number Variations in patients without biallelic pathogenic variant were investigated by Multiplex Ligation-dependent Probe Amplification method. Genotype-phenotype correlations and genotype-based phenotype predictions were examined by comparing molecular test results with BIOPKUdb database. The clinical distributions of the patients were as follows: classic PKU 21% (n=19), mild PKU 3% (n=3) and mild hyperphenylalaninemia 76% (n=71), respectively. Thirty-nine distinct variants and 70 distinct genotypes were found in patients. The most frequently observed variant was p.Ala300Ser (15%) and the most frequently observed genotype was p.Ala300Ser)/p.Ala300Ser (5,4%). Compound heterozygous genotypes (%69) were more prevalent than homozygous genotypes. Two novel variants, c.1089G>C and c.441+4A>C, were observed. Predicted metabolic phenotypes in the database showed consistency with patient phenotypes (n=33/41). BH4 responsiveness showed partial consistency with database predictions (n=13/25). Establishing genotype-phenotype correlations can facilitate personalized management approaches. Overall, this study contributes to understanding the genetic basis and clinical course of PKU.

Publisher

Springer Science and Business Media LLC

Reference16 articles.

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3. PAH Pathogenic Variants and Clinical Correlations in a Group of Hyperphenylalaninemia Patients from North-Western Romania;Iuhas A;Diagnostics,2023

4. Genetic etiology and clinical challenges of phenylketonuria;Elhawary NA;Hum Genomics,2022

5. Blau N, van Spronsen FJ, Levy HL, Phenylketonuria (2010) Lancet 376(9750):1417–1427

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