Literature DB >> 26542770

Mutational and phenotypical spectrum of phenylalanine hydroxylase deficiency in Denmark.

A Bayat1,2, S Yasmeen1, A Lund3, J B Nielsen1, L B Møller4.   

Abstract

We describe the genotypes of the complete cohort, from 1967 to 2014, of phenylketonuria (PKU) patients in Denmark, in total 376 patients. A total of 752 independent alleles were investigated. Mutations were identified on 744 PKU alleles (98.9%). In total, 82 different mutations were present in the cohort. The most frequent mutation c.1315+1G>A (IVS12+1G>A) was found on 25.80% of the 744 alleles. Other very frequent mutations were c.1222C>T (p.R408W) (16.93%) and c.1241A>G (p.Y414C) (11.15%). Among the identified mutations, five mutations; c.532G>A (p.E178K), c.730C>T (p.P244S), c.925G>A (p.A309T), c.1228T>A (p.F410I), and c.1199+4A>G (IVS11+4A>G) have not been reported previously. The metabolic phenotypes of PKU are classified into four categories; 'classical PKU', 'moderate PKU', 'mild PKU' and 'mild hyperphenylalaninemia'. In this study, we assigned the phenotypic outcome of three of the five novel mutations and furthermore six not previously classified mutations to one of the four PKU categories.
© 2015 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  PAH; PKU; classification; genotype-phenotype

Mesh:

Substances:

Year:  2015        PMID: 26542770     DOI: 10.1111/cge.12692

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  6 in total

1.  Hyperphenylalaninaemias in Estonia: Genotype-Phenotype Correlation and Comparative Overview of the Patient Cohort Before and After Nation-Wide Neonatal Screening.

Authors:  Hardo Lilleväli; Karit Reinson; Kai Muru; Kristi Simenson; Ülle Murumets; Tõnu Möls; Katrin Õunap
Journal:  JIMD Rep       Date:  2017-09-28

2.  Genetic study of the PAH locus in the Iranian population: familial gene mutations and minihaplotypes.

Authors:  Masoumeh Razipour; Elaheh Alavinejad; Seyede Zahra Sajedi; Saeed Talebi; Mona Entezam; Neda Mohajer; Golnaz-Ensieh Kazemi-Sefat; Jalal Gharesouran; Aria Setoodeh; Seyyed Mojtaba Mohaddes Ardebili; Mohammad Keramatipour
Journal:  Metab Brain Dis       Date:  2017-07-04       Impact factor: 3.584

3.  Genotypes of 2579 patients with phenylketonuria reveal a high rate of BH4 non-responders in Russia.

Authors:  Polina Gundorova; Anna A Stepanova; Irina A Kuznetsova; Sergey I Kutsev; Aleksander V Polyakov
Journal:  PLoS One       Date:  2019-01-22       Impact factor: 3.240

4.  Development of a porcine model of phenylketonuria with a humanized R408W mutation for gene editing.

Authors:  Robert A Kaiser; Daniel F Carlson; Kari L Allen; Dennis A Webster; Caitlin J VanLith; Clara T Nicolas; Lori G Hillin; Yue Yu; Catherine W Kaiser; William R Wahoff; Raymond D Hickey; Adrienne L Watson; Shelley R Winn; Beat Thöny; Douglas R Kern; Cary O Harding; Joseph B Lillegard
Journal:  PLoS One       Date:  2021-01-25       Impact factor: 3.240

5.  Mutational and phenotypic spectrum of phenylalanine hydroxylase deficiency in Zhejiang Province, China.

Authors:  Ting Chen; Weize Xu; Dingwen Wu; Jiamin Han; Ling Zhu; Fan Tong; Rulai Yang; Zhengyan Zhao; Pingping Jiang; Qiang Shu
Journal:  Sci Rep       Date:  2018-11-20       Impact factor: 4.379

6.  Development of a mutation hotspot detection kit for the phenylalanine hydroxylase gene by ARMS-PCR combined with fluorescent probe technology.

Authors:  Lin Wang; JinHua He; Rong Qiang; Wei Jie Xu; Wei Li; Na Cai; Xiao Bin Wang; RuiXue Zhang; Li Ping Zhang; Xiao Ping Ma; Chen Wei; ChengRong Song; WenWen Yu; Xiang Wang; Xu Li
Journal:  Biosci Rep       Date:  2021-02-26       Impact factor: 3.840

  6 in total

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