Literature DB >> 8097262

Genetic background of clinical homogeneity of phenylketonuria in Poland.

J Jaruzelska1, R Matuszak, S Lyonnet, F Rey, J Rey, J Filipowicz, K Borski, A Munnich.   

Abstract

In order to elucidate the clinical homogeneity and severity of the hyperphenylalaninaemias in Poland, a total of 71 children with typical phenylketonuria (PKU) originating from western and northern Poland were screened for 13 mutations in the phenylalanine hydroxylase (PAH) gene. Eighty percent of all PKU alleles tested were found to carry an identified mutation. One mutation, namely the R408W mutation, accounted for more than 63% of mutant PAH alleles in Poland, the other 27% being accounted for by six mutations: IVS12nt1 (5%), IVSnt546 (5%), Y414C (4%), R252W (1.5%), R261Q (< 1%), and G272ter (< 1%). The predominance of the R408W mutation resulted in a high rate of homozygotes (35.2%) and compound heterozygotes for this mutation in children from western and northern Poland. The frequency and deleterious nature of this mutation probably accounts for the clinical homogeneity and severity of the hyperphenylalaninaemias in Poland. In addition, the high rate of the R408W mutation and its association with mutant haplotype 2 at the PAH locus in Poland give additional support to the Balto-Slavic origin of this mutant gene.

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Year:  1993        PMID: 8097262      PMCID: PMC1016306          DOI: 10.1136/jmg.30.3.232

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  18 in total

1.  CpG dinucleotides are mutation hot spots in phenylketonuria.

Authors:  V Abadie; S Lyonnet; N Maurin; M Berthelon; C Caillaud; F Giraud; J F Mattei; J Rey; F Rey; A Munnich
Journal:  Genomics       Date:  1989-11       Impact factor: 5.736

2.  Geographical distribution gradients of the major PKU mutations and the linked haplotypes.

Authors:  L Kalaydjieva; B Dworniczak; V Kucinskas; V Yurgeliavicius; E Kunert; J Horst
Journal:  Hum Genet       Date:  1991-02       Impact factor: 4.132

3.  Spectrum of phenylketonuria mutations in western Europe and north Africa, and their relation to polymorphic DNA haplotypes at the phenylalanine hydroxylase locus.

Authors:  M Berthelon; C Caillaud; F Rey; P Labrune; D Melle; J Feingold; J Frézal; M L Briard; J P Farriaux; P Guibaud
Journal:  Hum Genet       Date:  1991-02       Impact factor: 4.132

4.  Tight linkage between a splicing mutation and a specific DNA haplotype in phenylketonuria.

Authors:  A G DiLella; J Marvit; A S Lidsky; F Güttler; S L Woo
Journal:  Nature       Date:  1986 Aug 28-Sep 3       Impact factor: 49.962

5.  The codon 408 mutation associated with haplotype 2 is predominant in Polish families with phenylketonuria.

Authors:  J Jaruzelska; K F Henriksen; F Güttler; O Riess; K Borski; N Blin; R Slomski
Journal:  Hum Genet       Date:  1991-01       Impact factor: 4.132

6.  Phenylketonuria: detection of a frequent haplotype 4 allele mutation.

Authors:  B Dworniczak; C Aulehla-Scholz; J Horst
Journal:  Hum Genet       Date:  1989-12       Impact factor: 4.132

7.  An amino-acid substitution involved in phenylketonuria is in linkage disequilibrium with DNA haplotype 2.

Authors:  A G DiLella; J Marvit; K Brayton; S L Woo
Journal:  Nature       Date:  1987 May 28-Jun 3       Impact factor: 49.962

8.  Molecular genetics of PKU in eastern Europe: a nonsense mutation associated with haplotype 4 of the phenylalanine hydroxylase gene.

Authors:  T Wang; Y Okano; R C Eisensmith; G Fekete; D Schuler; G Berencsi; I Nasz; S L Woo
Journal:  Somat Cell Mol Genet       Date:  1990-01

9.  Two mutations within the coding sequence of the phenylalanine hydroxylase gene.

Authors:  E Svensson; B Andersson; L Hagenfeldt
Journal:  Hum Genet       Date:  1990-08       Impact factor: 4.132

10.  Missense mutations associated with RFLP haplotypes 1 and 4 of the human phenylalanine hydroxylase gene.

Authors:  Y Okano; T Wang; R C Eisensmith; B Steinmann; R Gitzelmann; S L Woo
Journal:  Am J Hum Genet       Date:  1990-01       Impact factor: 11.025

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  3 in total

1.  Prevalence of tetrahydrobiopterine (BH4)-responsive alleles among Austrian patients with PAH deficiency: comprehensive results from molecular analysis in 147 patients.

Authors:  Elisabeth Sterl; Karl Paul; Eduard Paschke; Johannes Zschocke; Michaela Brunner-Krainz; Eva Windisch; Vassiliki Konstantopoulou; Dorothea Möslinger; Daniela Karall; Sabine Scholl-Bürgi; Wolfgang Sperl; Florian Lagler; Barbara Plecko
Journal:  J Inherit Metab Dis       Date:  2012-04-25       Impact factor: 4.982

2.  Recurrence of the R408W mutation in the phenylalanine hydroxylase locus in Europeans.

Authors:  R C Eisensmith; A A Goltsov; C O'Neill; L A Tyfield; E I Schwartz; A I Kuzmin; S S Baranovskaya; G L Tsukerman; E Treacy; C R Scriver
Journal:  Am J Hum Genet       Date:  1995-01       Impact factor: 11.025

3.  Phenylketonuria in Portugal: Genotype-phenotype correlations using molecular, biochemical, and haplotypic analyses.

Authors:  Filipa Ferreira; Luísa Azevedo; Raquel Neiva; Carmen Sousa; Helena Fonseca; Ana Marcão; Hugo Rocha; Célia Carmona; Sónia Ramos; Anabela Bandeira; Esmeralda Martins; Teresa Campos; Esmeralda Rodrigues; Paula Garcia; Luísa Diogo; Ana Cristina Ferreira; Silvia Sequeira; Francisco Silva; Luísa Rodrigues; Ana Gaspar; Patrícia Janeiro; António Amorim; Laura Vilarinho
Journal:  Mol Genet Genomic Med       Date:  2021-01-19       Impact factor: 2.183

  3 in total

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