Literature DB >> 19747868

Reassessment of phenylalanine tolerance in adults with phenylketonuria is needed as body mass changes.

Erin L MacLeod1, Sally T Gleason, Sandra C van Calcar, Denise M Ney.   

Abstract

Lifelong treatment of phenylketonuria (PKU) includes a phenylalanine (phe) restricted diet that provides sufficient phe for growth and maintenance plus phe-free amino acid formula to meet requirements for protein, energy and micronutrients. Phe tolerance (mg phe/kg body weight/day) is the amount of phe those with PKU can consume and maintain acceptable blood phe levels; it requires individual assessment because of varying phenylalanine hydroxylase activity. The objective was to reassess phe tolerance in eight adults with PKU considering phe requirements, blood phe levels, genotype and phe tolerance at 5 years of age. Subjects had not received a personalized assessment of phe tolerance in several years, and five subjects were overweight, body mass index (BMI) 25-28. With the guidance of a metabolic dietitian, seven subjects increased phe tolerance (by 15-173%) without significantly increasing blood phe concentration. Increased phe tolerance was associated with both improved dietary compliance and inadequate phe intake at the onset of the protocol compared with current requirements. Improved dietary compliance reflected increased consumption of protein equivalents from amino acid formula and increased frequency of formula intake, from 2.2 to 3 times per day. Predictors of higher final phe tolerance following reassessment included being male and having a lower BMI (R(2)=0.588). This suggests that the rising trend of overweight and obesity may affect assessment of phe tolerance in adults. Therefore, interaction with the metabolic dietitian to reassess phe tolerance in relation to body mass is essential throughout adulthood to insure adequate intake of phe to support protein synthesis and prevent catabolism.

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Year:  2009        PMID: 19747868      PMCID: PMC2783926          DOI: 10.1016/j.ymgme.2009.07.016

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  38 in total

Review 1.  Nutritional management of PKU with glycomacropeptide from cheese whey.

Authors:  D M Ney; S T Gleason; S C van Calcar; E L MacLeod; K L Nelson; M R Etzel; G M Rice; J A Wolff
Journal:  J Inherit Metab Dis       Date:  2008-10-29       Impact factor: 4.982

Review 2.  What we know that could influence future treatment of phenylketonuria.

Authors:  C N Sarkissian; A Gámez; C R Scriver
Journal:  J Inherit Metab Dis       Date:  2008-08-03       Impact factor: 4.982

3.  Genotype-phenotype correlations analysis of mutations in the phenylalanine hydroxylase (PAH) gene.

Authors:  Dani Bercovich; Arava Elimelech; Joel Zlotogora; Sigal Korem; Tal Yardeni; Nurit Gal; Nurit Goldstein; Bela Vilensky; Roni Segev; Smadar Avraham; Ron Loewenthal; Gerard Schwartz; Yair Anikster
Journal:  J Hum Genet       Date:  2008-02-26       Impact factor: 3.172

Review 4.  The truth of treating patients with phenylketonuria after childhood: the need for a new guideline.

Authors:  F J van Spronsen; P Burgard
Journal:  J Inherit Metab Dis       Date:  2008-08-12       Impact factor: 4.982

5.  Phenylalanine tolerance can already reliably be assessed at the age of 2 years in patients with PKU.

Authors:  F J van Spronsen; M van Rijn; B Dorgelo; M Hoeksma; A M Bosch; M F Mulder; J B C de Klerk; T de Koning; M Estela Rubio-Gozalbo; M de Vries; P H Verkerk
Journal:  J Inherit Metab Dis       Date:  2009-01-10       Impact factor: 4.982

Review 6.  Optimizing the use of sapropterin (BH(4)) in the management of phenylketonuria.

Authors:  Nenad Blau; Amaya Bélanger-Quintana; Mübeccel Demirkol; François Feillet; Marcello Giovannini; Anita MacDonald; Friedrich K Trefz; Francjan J van Spronsen
Journal:  Mol Genet Metab       Date:  2009-02-08       Impact factor: 4.797

Review 7.  Growth and body composition in children with classical phenylketonuria: results in 34 patients and review of the literature.

Authors:  M Huemer; C Huemer; D Möslinger; D Huter; S Stöckler-Ipsiroglu
Journal:  J Inherit Metab Dis       Date:  2007-07-11       Impact factor: 4.982

8.  Effect of BH(4) supplementation on phenylalanine tolerance.

Authors:  A Burlina; N Blau
Journal:  J Inherit Metab Dis       Date:  2008-12-09       Impact factor: 4.982

9.  Improved nutritional management of phenylketonuria by using a diet containing glycomacropeptide compared with amino acids.

Authors:  Sandra C van Calcar; Erin L MacLeod; Sally T Gleason; Mark R Etzel; Murray K Clayton; Jon A Wolff; Denise M Ney
Journal:  Am J Clin Nutr       Date:  2009-02-25       Impact factor: 7.045

10.  PKU-what is daily practice in various centres in Europe? Data from a questionnaire by the scientific advisory committee of the European Society of Phenylketonuria and Allied Disorders.

Authors:  F J van Spronsen; K Kiaer Ahring; M Gizewska
Journal:  J Inherit Metab Dis       Date:  2009-01-13       Impact factor: 4.982

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  15 in total

1.  Development of Metabolic Phenotype in Phenylketonuria: Evaluation of the Blaskovics Protein Loading Test at 5 Years of Age.

Authors:  P Burgard; E Mönch; J Zschocke; U Wendel; U Langenbeck
Journal:  JIMD Rep       Date:  2015-12-19

2.  Nutritional Management of Phenylketonuria.

Authors:  Erin L Macleod; Denise M Ney
Journal:  Ann Nestle Eng       Date:  2010-06

Review 3.  Food products made with glycomacropeptide, a low-phenylalanine whey protein, provide a new alternative to amino Acid-based medical foods for nutrition management of phenylketonuria.

Authors:  Sandra C van Calcar; Denise M Ney
Journal:  J Acad Nutr Diet       Date:  2012-08       Impact factor: 4.910

4.  Transition of young adults with phenylketonuria from pediatric to adult care.

Authors:  Ulrike Mütze; Annika Roth; Johannes F W Weigel; Skadi Beblo; Christoph G Baerwald; Peter Bührdel; Wieland Kiess
Journal:  J Inherit Metab Dis       Date:  2011-02-09       Impact factor: 4.982

Review 5.  Phenylketonuria: Current Treatments and Future Developments.

Authors:  Uta Lichter-Konecki; Jerry Vockley
Journal:  Drugs       Date:  2019-04       Impact factor: 9.546

Review 6.  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

Review 7.  Recommendations for the nutrition management of phenylalanine hydroxylase deficiency.

Authors:  Rani H Singh; Fran Rohr; Dianne Frazier; Amy Cunningham; Shideh Mofidi; Beth Ogata; Patricia L Splett; Kathryn Moseley; Kathleen Huntington; Phyllis B Acosta; Jerry Vockley; Sandra C Van Calcar
Journal:  Genet Med       Date:  2014-01-02       Impact factor: 8.822

8.  Long-term follow-up of patients with phenylketonuria treated with tetrahydrobiopterin: a seven years experience.

Authors:  Iris Scala; Daniela Concolino; Roberto Della Casa; Anna Nastasi; Carla Ungaro; Serena Paladino; Brunella Capaldo; Margherita Ruoppolo; Aurora Daniele; Giuseppe Bonapace; Pietro Strisciuglio; Giancarlo Parenti; Generoso Andria
Journal:  Orphanet J Rare Dis       Date:  2015-02-08       Impact factor: 4.123

9.  Efficacy, safety and population pharmacokinetics of sapropterin in PKU patients <4 years: results from the SPARK open-label, multicentre, randomized phase IIIb trial.

Authors:  Ania C Muntau; Alberto Burlina; François Eyskens; Peter Freisinger; Corinne De Laet; Vincenzo Leuzzi; Frank Rutsch; H Serap Sivri; Suresh Vijay; Milva Orquidea Bal; Gwendolyn Gramer; Renata Pazdírková; Maureen Cleary; Amelie S Lotz-Havla; Alain Munafo; Diane R Mould; Flavie Moreau-Stucker; Daniela Rogoff
Journal:  Orphanet J Rare Dis       Date:  2017-03-09       Impact factor: 4.123

10.  Nutrient intake, body composition, and blood phenylalanine control in children with phenylketonuria compared to healthy controls.

Authors:  Melissa Sailer; Gabriela Elizondo; Julie Martin; Cary O Harding; Melanie B Gillingham
Journal:  Mol Genet Metab Rep       Date:  2020-05-11
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