Literature DB >> 33564846

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

Lin Wang1, JinHua He2, Rong Qiang3,4,1, Wei Jie Xu5, Wei Li1, Na Cai1, Xiao Bin Wang1, RuiXue Zhang1, Li Ping Zhang1, Xiao Ping Ma1, Chen Wei1, ChengRong Song1, WenWen Yu1, Xiang Wang3,4, Xu Li3,4.   

Abstract

To develop a screening kit for detecting mutation hotspots of the phenylalanine hydroxylase (PAH) gene. Thirteen exons of the PAH gene were sequenced in 84 cases with phenylketonuria (PKU) diagnosed during neonatal genetic and metabolic disease screening in Shaanxi province, and their mutations were analyzed. We designed and developed a screening kit to detect nine mutation sites covering more than 50% of the PAH mutations found in Shaanxi province (c.728G>A, c.1197A>T, c.331C>T, c.1068C>A, c.611A>G, c.1238G>C, c.721C>T, c.442-1G>A, and c.158G>A) by using amplification refractory mutation system-polymerase chain reaction (ARMS-PCR) combined with fluorescent probe technology. Peripheral blood and dried blood samples from PKU families were used for clinical verification of the newly developed kit. PAH gene mutations were detected in 84 children diagnosed with PKU. A total of 159 mutant alleles were identified, consisting of 100 missense mutations, 28 shear mutations, 24 nonsense mutations, and 7 deletion mutations. Exon 7 had the highest mutation frequency (32.08%). Among them, the mutation frequency of p.R243Q was the highest, accounting for 20.13% of all mutations, followed by p.R111X, IVS4-1G>A, EX6-96A>G, and p.R413P; these five loci accounted for 47.17% (75/159) of all mutations. In addition, we identified three previously unreported PAH gene mutations (p.C334X, p.G46D, and p.G256D). Fifteen mutation sites were identified in the 47 PAH carriers identified by next-generation sequencing (NGS), which were verified by the newly developed kit, with an agreement rate of 100%. This newly developed kit based on ARMS-PCR combined with fluorescent probe technology can be used to detect common PAH gene mutations.
© 2021 The Author(s).

Entities:  

Keywords:  amplification refractory mutation system PCR; gene mutation; next-generation sequencing; phenylalanine hydroxylase; phenylketonuria

Mesh:

Substances:

Year:  2021        PMID: 33564846      PMCID: PMC7897920          DOI: 10.1042/BSR20201660

Source DB:  PubMed          Journal:  Biosci Rep        ISSN: 0144-8463            Impact factor:   3.840


  11 in total

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Authors:  Nenad Blau
Journal:  Hum Mutat       Date:  2016-03-18       Impact factor: 4.878

2.  Microhaplotype identified and performed in genetic investigation using PCR-SSCP.

Authors:  Peng Chen; Jing Zhu; Yan Pu; Youjing Jiang; Dan Chen; Hui Wang; Jiong Mao; Bin Zhou; Linbo Gao; Peng Bai; Weibo Liang; Lin Zhang
Journal:  Forensic Sci Int Genet       Date:  2017-01-20       Impact factor: 4.882

3.  [Consensus about the diagnosis and treatment of hyperphenylalaninemia].

Authors:  Yanling Yang; Yun Ye
Journal:  Zhonghua Er Ke Za Zhi       Date:  2014-06

4.  Mutation spectrum of hyperphenylalaninemia candidate genes and the genotype-phenotype correlation in the Chinese population.

Authors:  Ruifang Wang; Nan Shen; Jun Ye; Lianshu Han; Wenjuan Qiu; Huiwen Zhang; Lili Liang; Yu Sun; Yanjie Fan; Lili Wang; Yu Wang; Zhuwen Gong; Huili Liu; Jianguo Wang; Hui Yan; Nenad Blau; Xuefan Gu; Yongguo Yu
Journal:  Clin Chim Acta       Date:  2018-02-28       Impact factor: 3.786

5.  Mutational and phenotypical spectrum of phenylalanine hydroxylase deficiency in Denmark.

Authors:  A Bayat; S Yasmeen; A Lund; J B Nielsen; L B Møller
Journal:  Clin Genet       Date:  2015-12-12       Impact factor: 4.438

Review 6.  Early births and congenital birth defects: a complex interaction.

Authors:  Jonathan R Swanson; Robert A Sinkin
Journal:  Clin Perinatol       Date:  2013-09-20       Impact factor: 3.430

7.  Phenylalanine hydroxylase deficiency: diagnosis and management guideline.

Authors:  Jerry Vockley; Hans C Andersson; Kevin M Antshel; Nancy E Braverman; Barbara K Burton; Dianne M Frazier; John Mitchell; Wendy E Smith; Barry H Thompson; Susan A Berry
Journal:  Genet Med       Date:  2013-10-10       Impact factor: 8.822

8.  Expanded Newborn Screening for Inborn Errors of Metabolism and Genetic Characteristics in a Chinese Population.

Authors:  Kejian Guo; Xuan Zhou; Xigui Chen; Yili Wu; Chuanxin Liu; Qingsheng Kong
Journal:  Front Genet       Date:  2018-04-20       Impact factor: 4.599

9.  ACMG position statement on prenatal/preconception expanded carrier screening.

Authors:  Wayne W Grody; Barry H Thompson; Anthony R Gregg; Lora H Bean; Kristin G Monaghan; Adele Schneider; Roger V Lebo
Journal:  Genet Med       Date:  2013-04-25       Impact factor: 8.822

10.  Spectrum of PAH gene variants among a population of Han Chinese patients with phenylketonuria from northern China.

Authors:  Ning Liu; Qiuying Huang; Qingge Li; Dehua Zhao; Xiaole Li; Lixia Cui; Ying Bai; Yin Feng; Xiangdong Kong
Journal:  BMC Med Genet       Date:  2017-10-05       Impact factor: 2.103

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