Literature DB >> 25480112

Management of adult patients with phenylketonuria: survey results from 24 countries.

Friedrich K Trefz1, Francjan J van Spronsen, Anita MacDonald, François Feillet, Ania C Muntau, Amaya Belanger-Quintana, Alberto Burlina, Mübeccel Demirkol, Marcello Giovannini, Christoph Gasteyger.   

Abstract

UNLABELLED: Phenylketonuria (PKU) is no longer considered merely a pediatric concern; current guidelines recommend life-long treatment. However, information on the adult PKU patient population is scarce. A survey was initiated on behalf of the European PKU Group (EPG) that focused specifically on early-treated adult patients diagnosed by neonatal screening. The online survey was sent via email to 204 healthcare professionals (HCPs) in 33 countries. Eighty-one HCPs from 24 countries responded. The main findings were that the majority of adult patients with PKU in active follow-up are under 30 years of age and are managed in centers that also treat children. Seventy-eight percent of adult PKU patients in follow-up receive treatment, mainly by diet (71 %), with BH4 treatment rarely used in adulthood. Only 26 % of responding HCPs perform routine neurocognitive testing in all their adult patients. There was little consensus regarding target blood phenylalanine (Phe) concentrations, although the majority of respondents reported that their patients achieved blood Phe concentrations below 1200 μmol/l.
CONCLUSION: This survey highlights the need for blood Phe concentration target recommendations and consensus guidelines, more research into adult PKU patient management, and the need to identify those patients lost to follow-up to ensure PKU is managed for life.

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Year:  2014        PMID: 25480112     DOI: 10.1007/s00431-014-2458-4

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


  26 in total

1.  Living situation, occupation and health-related quality of life in adult patients with classic galactosemia.

Authors:  Björn Hoffmann; Nico Dragano; Susanne Schweitzer-Krantz
Journal:  J Inherit Metab Dis       Date:  2012-03-24       Impact factor: 4.982

2.  Phenylketonuria in adulthood: a collaborative study.

Authors:  R Koch; B Burton; G Hoganson; R Peterson; W Rhead; B Rouse; R Scott; J Wolff; A M Stern; F Guttler; M Nelson; F de la Cruz; J Coldwell; R Erbe; M T Geraghty; C Shear; J Thomas; C Azen
Journal:  J Inherit Metab Dis       Date:  2002-09       Impact factor: 4.982

Review 3.  Adult phenylketonuria outcome and management.

Authors:  F Trefz; F Maillot; K Motzfeldt; M Schwarz
Journal:  Mol Genet Metab       Date:  2011-08-26       Impact factor: 4.797

4.  Blood phenylalanine control in phenylketonuria: a survey of 10 European centres.

Authors:  K Ahring; A Bélanger-Quintana; K Dokoupil; H Gokmen-Ozel; A M Lammardo; A MacDonald; K Motzfeldt; M Nowacka; M Robert; M van Rijn
Journal:  Eur J Clin Nutr       Date:  2010-12-01       Impact factor: 4.016

Review 5.  National Institutes of Health Consensus Development Conference Statement: phenylketonuria: screening and management, October 16-18, 2000.

Authors: 
Journal:  Pediatrics       Date:  2001-10       Impact factor: 7.124

6.  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

7.  Screening for cognitive and social-emotional problems in individuals with PKU: tools for use in the metabolic clinic.

Authors:  S Waisbren; D A White
Journal:  Mol Genet Metab       Date:  2010       Impact factor: 4.797

8.  Reaching out to the lost generation of adults with early-treated phenylketonuria (PKU).

Authors:  Barbara K Burton; Lauren Leviton
Journal:  Mol Genet Metab       Date:  2010-06-22       Impact factor: 4.797

9.  Phenylalanine-restricted diet should be life long. A case report on long-term follow-up of an adolescent with untreated phenylketonuria.

Authors:  Joav Merrick; Shoshana Aspler; Gerard Schwarz
Journal:  Int J Adolesc Med Health       Date:  2003 Apr-Jun

10.  High phenylalanine levels directly affect mood and sustained attention in adults with phenylketonuria: a randomised, double-blind, placebo-controlled, crossover trial.

Authors:  Amber E ten Hoedt; Leo M J de Sonneville; Baudouin Francois; Nienke M ter Horst; Mirian C H Janssen; M Estela Rubio-Gozalbo; Frits A Wijburg; Carla E M Hollak; Annet M Bosch
Journal:  J Inherit Metab Dis       Date:  2010-12-10       Impact factor: 4.982

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  15 in total

1.  Dietary patterns, cost and compliance with low-protein diet of phenylketonuria and other inherited metabolic diseases.

Authors:  T Mlčoch; R Puda; P Ješina; M Lhotáková; Š Štěrbová; T Doležal
Journal:  Eur J Clin Nutr       Date:  2017-06-28       Impact factor: 4.016

2.  Multicompartment analysis of protein-restricted phenylketonuric mice reveals amino acid imbalances in brain.

Authors:  Kara R Vogel; Erland Arning; Teodoro Bottiglieri; K Michael Gibson
Journal:  J Inherit Metab Dis       Date:  2016-10-19       Impact factor: 4.982

3.  "I Feel Lucky" - Gratitude Among Young Adults with Phenylketonuria (PKU).

Authors:  Plata Sofie Diesen
Journal:  J Genet Couns       Date:  2016-02-18       Impact factor: 2.537

Review 4.  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

5.  Diagnostic and management practices for phenylketonuria in 19 countries of the South and Eastern European Region: survey results.

Authors:  Maria Giżewska; Anita MacDonald; Amaya Bélanger-Quintana; Alberto Burlina; Maureen Cleary; Turgay Coşkun; François Feillet; Ania C Muntau; Friedrich K Trefz; Francjan J van Spronsen; Nenad Blau
Journal:  Eur J Pediatr       Date:  2015-09-08       Impact factor: 3.183

6.  The Predictive Value of Genetic Analyses in the Diagnosis of Tetrahydrobiopterin (BH4)-Responsiveness in Chinese Phenylalanine Hydroxylase Deficiency Patients.

Authors:  Tianwen Zhu; Jun Ye; Lianshu Han; Wenjuan Qiu; Huiwen Zhang; Lili Liang; Xuefan Gu
Journal:  Sci Rep       Date:  2017-07-28       Impact factor: 4.379

7.  Development of the US English version of the phenylketonuria - quality of life (PKU-QOL) questionnaire.

Authors:  Elaina Jurecki; Amy Cunningham; Vanessa Birardi; Grégory Gagol; Catherine Acquadro
Journal:  Health Qual Life Outcomes       Date:  2017-03-09       Impact factor: 3.186

8.  Development of a practical dietitian road map for the nutritional management of phenylketonuria (PKU) patients on pegvaliase.

Authors:  Júlio César Rocha; Heather Bausell; Amaya Bélanger-Quintana; Laurie Bernstein; Hülya Gökmen-Özel; Alexandra Jung; Anita MacDonald; Fran Rohr; Esther van Dam; Margret Heddrich-Ellerbrok
Journal:  Mol Genet Metab Rep       Date:  2021-05-25

9.  Ten years of specialized adult care for phenylketonuria - a single-centre experience.

Authors:  Ulrike Mütze; Alena Gerlinde Thiele; Christoph Baerwald; Uta Ceglarek; Wieland Kiess; Skadi Beblo
Journal:  Orphanet J Rare Dis       Date:  2016-03-24       Impact factor: 4.123

10.  The cardiovascular phenotype of adult patients with phenylketonuria.

Authors:  Aline Azabdaftari; Markus van der Giet; Mirjam Schuchardt; Julia B Hennermann; Ursula Plöckinger; Uwe Querfeld
Journal:  Orphanet J Rare Dis       Date:  2019-09-06       Impact factor: 4.123

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