Literature DB >> 3939586

Outcome of pregnancy in the rat with mild hyperphenylalaninaemia and hypertyrosinaemia: implications for the management of "human maternal PKU".

S A Lewis, I C Lyon, R B Elliott.   

Abstract

In attempting to determine the effects of mildly elevated maternal phenylalanine (Phe) blood levels on the developing fetal rat brain, a dietary supplement of Phe was given, under taste cover of Aspartame. Phe and tyrosine (Tyr) levels were mildly elevated throughout pregnancy without evidence of malnutrition. Mild hyperphenylalaninaemia with concurrent hypertyrosinaemia induced in rats prior to conception resulted in microcephaly and lasting behavioural problems in the offspring, specifically hyperactivity and learning difficulties. Dams fed Tyr to produce Tyr levels equivalent to the Phe-fed animals showed only the learning difficulties among the offspring. alpha-Methyl Phe, a Phe hydroxylase inhibitor, fed in conjunction with Phe, at the level relevant to these experiments, resulted in raised Tyr levels and does not provide a better method of determining whether mildly elevated maternal Phe levels alone, or Phe and Tyr in combination, cause the abnormality found in the offspring of Phe-supplemented dams. Therapeutic addition of Tyr to diets of mothers with even mild hyperphenylalaninaemia should be approached with caution as mild co-elevation of Phe and Tyr in the fetus may be harmful. In the face of such a possible therapeutic dilemma alternatives, such as dietary additions of other essential amino acids to limit fetal brain damage, need to be explored.

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Year:  1985        PMID: 3939586     DOI: 10.1007/bf01819292

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


  12 in total

1.  Experimental phenylketonuria in rats.

Authors:  H L WANG; H A WAISMAN
Journal:  Proc Soc Exp Biol Med       Date:  1961-11

2.  The effects of bilateral hippocampal lesions in rats.

Authors:  D P KIMBLE
Journal:  J Comp Physiol Psychol       Date:  1963-04

3.  Enduring behavioral changes in rats with experimental phenylketonuria.

Authors:  A E Andersen; G Guroff
Journal:  Proc Natl Acad Sci U S A       Date:  1972-04       Impact factor: 11.205

4.  The influence of high phenylalanine and tyrosine on the concentrations of essential amino acids in brain.

Authors:  C M McKean; D E Boggs; N A Peterson
Journal:  J Neurochem       Date:  1968-03       Impact factor: 5.372

5.  Relative inability of mother and child to convert phenylalanine of tyrosine--a possible cause of nonspecific mental retardation.

Authors:  A Fujimoto; R Crawford; S P Bessman
Journal:  Biochem Med       Date:  1979-06

6.  Phenylalanine hydroxylase and tyrosine aminotransferase in human fetal and adult liver.

Authors:  J A Delvalle; O Greengard
Journal:  Pediatr Res       Date:  1977-01       Impact factor: 3.756

7.  Effects of untreated maternal phenylketonuria and hyperphenylalaninemia on the fetus.

Authors:  H L Levy; S E Waisbren
Journal:  N Engl J Med       Date:  1983-11-24       Impact factor: 91.245

8.  Effects of p-chlorophenylalanine and alpha-methylphenylalanine on amino acid uptake and protein synthesis in mouse neuroblastoma cells.

Authors:  C J Kelly; T C Johnson
Journal:  Biochem J       Date:  1978-09-15       Impact factor: 3.857

9.  Aspartylphenylalanine methyl ester: a low-calorie sweetener.

Authors:  M R Cloninger; R E Baldwin
Journal:  Science       Date:  1970-10-02       Impact factor: 47.728

10.  Maternal phenylketonuria and hyperphenylalaninemia. An international survey of the outcome of untreated and treated pregnancies.

Authors:  R R Lenke; H L Levy
Journal:  N Engl J Med       Date:  1980-11-20       Impact factor: 91.245

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  4 in total

1.  Maternal tyrosinaemia II: management and successful outcome.

Authors:  D E Francis; D M Kirby; G N Thompson
Journal:  Eur J Pediatr       Date:  1992-03       Impact factor: 3.183

2.  Pregnancy in phenylketonuria: dietary treatment aimed at normalising maternal plasma phenylalanine concentration.

Authors:  G N Thompson; D E Francis; D M Kirby; R Compton
Journal:  Arch Dis Child       Date:  1991-11       Impact factor: 3.791

Review 3.  The complete European guidelines on phenylketonuria: diagnosis and treatment.

Authors:  A M J van Wegberg; A MacDonald; K Ahring; A Bélanger-Quintana; N Blau; A M Bosch; A Burlina; J Campistol; F Feillet; M Giżewska; S C Huijbregts; S Kearney; V Leuzzi; F Maillot; A C Muntau; M van Rijn; F Trefz; J H Walter; F J van Spronsen
Journal:  Orphanet J Rare Dis       Date:  2017-10-12       Impact factor: 4.123

4.  L-tyrosine supplementation does not ameliorate skeletal muscle dysfunction in zebrafish and mouse models of dominant skeletal muscle α-actin nemaline myopathy.

Authors:  Adriana M Messineo; Charlotte Gineste; Tamar E Sztal; Elyshia L McNamara; Christophe Vilmen; Augustin C Ogier; Dorothee Hahne; David Bendahan; Nigel G Laing; Robert J Bryson-Richardson; Julien Gondin; Kristen J Nowak
Journal:  Sci Rep       Date:  2018-07-31       Impact factor: 4.379

  4 in total

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