Literature DB >> 8828604

Untreated non-phenylketonuric-hyperphenylalaninaemia: intellectual and neurological outcome.

J Weglage1, K Ullrich, M Pietsch, B Fünders, R Zass, H G Koch.   

Abstract

The intellectual, neurological, and neuropsychological outcome of patients with non-phenylketonuric-hyperphenylalaninaemia (PKU-HPA) (serum phenylalanine levels under free diet < 600 mumol/l) has not been systematically studied so far. We therefore tested 28 patients (mean age = 21.8, SD = 4.2 years) for IQ (WAIS-R/WISC-R), school performance, job career, clinical neurological examination, fine motor performance (motor performance task), and selective and sustained attention (stroop task, Dot Pattern Exercise from the Sonneville visual attention task). In addition, cranial MRI (1.5 T unit) was obtained in 10 of these patients. Clinical-neurological examination revealed no significant abnormalities in the non-PKU-HPA patients. They also had a normal IQ (mean = 101.9, SD = 13.6). Compared to their healthy siblings, they attended a normal school and had a normal job career. The motor performance task revealed no deficits in fine motor abilities. The patients performed normally in the stroop task and the dot pattern exercise. Their MRIs were normal. Our results indicate that patients with non-PKU-HPA are not at risk for developing intellectual, neurological, and neuropsychological impairment, as described for patients with treated mild or classical phenylketonuria. From this point of view a dietary treatment is not necessary in patients with hyperphenylalaninaemia.

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Year:  1996        PMID: 8828604     DOI: 10.1007/pl00014244

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  9 in total

1.  Deficits in selective and sustained attention processes in early treated children with phenylketonuria--result of impaired frontal lobe functions?

Authors:  J Weglage; M Pietsch; B Funders; H G Koch; K Ullrich
Journal:  Eur J Pediatr       Date:  1996-03       Impact factor: 3.183

2.  Phenylalaninaemia. Differential diagnosis.

Authors:  M E Blaskovics; G E Schaeffler; S Hack
Journal:  Arch Dis Child       Date:  1974-11       Impact factor: 3.791

3.  Intelligence and quality of dietary treatment in phenylketonuria.

Authors:  I Smith; M G Beasley; A E Ades
Journal:  Arch Dis Child       Date:  1990-05       Impact factor: 3.791

4.  Intelligence in mild atypical phenylketonuria.

Authors:  P M Costello; M G Beasley; S L Tillotson; I Smith
Journal:  Eur J Pediatr       Date:  1994-04       Impact factor: 3.183

5.  Recommendations on the dietary management of phenylketonuria. Report of Medical Research Council Working Party on Phenylketonuria.

Authors: 
Journal:  Arch Dis Child       Date:  1993-03       Impact factor: 3.791

6.  Psychological and social findings in adolescents with phenylketonuria.

Authors:  J Weglage; B Fünders; B Wilken; D Schubert; E Schmidt; P Burgard; K Ullrich
Journal:  Eur J Pediatr       Date:  1992-07       Impact factor: 3.183

7.  Sustained attention in adult phenylketonuria: the influence of the concurrent phenylalanine-blood-level.

Authors:  E Schmidt; A Rupp; P Burgard; J Pietz; J Weglage; L de Sonneville
Journal:  J Clin Exp Neuropsychol       Date:  1994-10       Impact factor: 2.475

8.  Neurological findings in early treated phenylketonuria.

Authors:  J Weglage; M Pietsch; B Fünders; H G Koch; K Ullrich
Journal:  Acta Paediatr       Date:  1995-04       Impact factor: 2.299

9.  Cranial MRI in PKU: evaluation of a critical threshold for blood phenylalanine.

Authors:  K Ullrich; J Weglage; G Schuierer; B Fünders; M Pietsch; H G Koch; H Hahn-Ullrich
Journal:  Neuropediatrics       Date:  1994-10       Impact factor: 1.947

  9 in total
  6 in total

1.  Predictability and inconsistencies in the cognitive outcome of early treated PKU patients.

Authors:  Filippo Manti; Francesca Nardecchia; Sabrina Paci; Flavia Chiarotti; Claudia Carducci; Carla Carducci; Silvia Dalmazzone; Graziella Cefalo; Elisabetta Salvatici; Giuseppe Banderali; Vincenzo Leuzzi
Journal:  J Inherit Metab Dis       Date:  2017-08-23       Impact factor: 4.982

2.  Cognitive function in untreated subjects with mild hyperphenylalaninemia: a systematic review.

Authors:  Noushin Rostampour; Rojin Chegini; Silva Hovsepian; Farzaneh Zamaneh; Mahin Hashemipour
Journal:  Neurol Sci       Date:  2022-06-21       Impact factor: 3.830

3.  Influence of PAH Genotype on Sapropterin Response in PKU: Results of a Single-Center Cohort Study.

Authors:  Sarah Leuders; Eva Wolfgart; Torsten Ott; Marcel du Moulin; Agnes van Teeffelen-Heithoff; Lydia Vogelpohl; Ulrike Och; Thorsten Marquardt; Josef Weglage; Reinhold Feldmann; Frank Rutsch
Journal:  JIMD Rep       Date:  2013-11-05

4.  Meta-analysis of neuropsychological symptoms of adolescents and adults with PKU.

Authors:  J J Moyle; A M Fox; M Arthur; M Bynevelt; J R Burnett
Journal:  Neuropsychol Rev       Date:  2007-04-05       Impact factor: 7.444

Review 5.  Mild hyperphenylalaninemia: to treat or not to treat.

Authors:  Francjan J van Spronsen
Journal:  J Inherit Metab Dis       Date:  2011-02-24       Impact factor: 4.982

6.  Cognitive functioning in mild hyperphenylalaninemia.

Authors:  Alicia de la Parra; María Ignacia García; Susan E Waisbren; Verónica Cornejo; Erna Raimann
Journal:  Mol Genet Metab Rep       Date:  2015-10-29
  6 in total

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