Literature DB >> 15159646

Discordant PKU phenotype in one family due to disparate genotypes and a novel mutation.

J J Johnston1, U Lichter-Konecki, E Wilson, B R Cobb, B M Evans, R E Schnur, L-J C Wong.   

Abstract

Classical phenylketonuria (PKU) and mild hyperphenylalaninaemia (MHP) are two ends of the broad diagnostic spectrum in phenylalanine hydroxylase (PAH) deficiency. We have analysed a family in which classical PKU, MHP and a normal phenotype occurred in family members with different mutations. Sequence analysis revealed three mutations segregating in the family. The individual with classical PKU had two previously reported deleterious mutations. A third novel mutation was identified in the other two individuals. This report demonstrates that when discordant phenotypes occur in a family, without protein loading or phenylalanine tolerance test, complete analysis of the PAH gene may be performed in order to support the diagnosis and assist in accurate genetic counselling and patient management.

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Year:  2004        PMID: 15159646     DOI: 10.1023/B:BOLI.0000028782.39513.20

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  13 in total

1.  Detection of the XmnI RFLP at the human PAH locus by PCR.

Authors:  A A Goltsov; R C Eisensmith; S L Woo
Journal:  Nucleic Acids Res       Date:  1992-02-25       Impact factor: 16.971

2.  A rapid non-enzymatic method for the preparation of HMW DNA from blood for RFLP studies.

Authors:  D K Lahiri; J I Nurnberger
Journal:  Nucleic Acids Res       Date:  1991-10-11       Impact factor: 16.971

3.  In vivo disposal of phenylalanine in phenylketonuria: a study of two siblings.

Authors:  E Treacy; J J Pitt; K Seller; G N Thompson; S Ramus; R G Cotton
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

4.  Human phenylalanine hydroxylase mutations and hyperphenylalaninemia phenotypes: a metanalysis of genotype-phenotype correlations.

Authors:  E Kayaalp; E Treacy; P J Waters; S Byck; P Nowacki; C R Scriver
Journal:  Am J Hum Genet       Date:  1997-12       Impact factor: 11.025

5.  Mutation analysis in families with discordant phenotypes of phenylalanine hydroxylase deficiency. Inheritance and expression of the hyperphenylalaninaemias.

Authors:  P Guldberg; H L Levy; R Koch; C M Berlin; B Francois; K F Henriksen; F Güttler
Journal:  J Inherit Metab Dis       Date:  1994       Impact factor: 4.982

6.  A single polymorphic STR system in the human phenylalanine hydroxylase gene permits rapid prenatal diagnosis and carrier screening for phenylketonuria.

Authors:  A A Goltsov; R C Eisensmith; E R Naughton; L Jin; R Chakraborty; S L Woo
Journal:  Hum Mol Genet       Date:  1993-05       Impact factor: 6.150

7.  Associations between mutations and a VNTR in the human phenylalanine hydroxylase gene.

Authors:  A A Goltsov; R C Eisensmith; D S Konecki; U Lichter-Konecki; S L Woo
Journal:  Am J Hum Genet       Date:  1992-09       Impact factor: 11.025

8.  Application of natural and amplification created restriction sites for the diagnosis of PKU mutations.

Authors:  H G Eiken; E Odland; H Boman; L Skjelkvåle; L F Engebretsen; J Apold
Journal:  Nucleic Acids Res       Date:  1991-04-11       Impact factor: 16.971

9.  Hepatocyte nuclear factor 1 inactivation results in hepatic dysfunction, phenylketonuria, and renal Fanconi syndrome.

Authors:  M Pontoglio; J Barra; M Hadchouel; A Doyen; C Kress; J P Bach; C Babinet; M Yaniv
Journal:  Cell       Date:  1996-02-23       Impact factor: 41.582

10.  Molecular analysis of the inheritance of phenylketonuria and mild hyperphenylalaninemia in families with both disorders.

Authors:  F D Ledley; H L Levy; S L Woo
Journal:  N Engl J Med       Date:  1986-05-15       Impact factor: 91.245

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