Literature DB >> 20938748

The national Austrian newborn screening program - eight years experience with mass spectrometry. past, present, and future goals.

David C Kasper1, Rene Ratschmann, Thomas F Metz, Thomas P Mechtler, Dorothea Möslinger, Vassiliki Konstantopoulou, Chike B Item, Arnold Pollak, Kurt R Herkner.   

Abstract

BACKGROUND: the National Austrian Newborn Screening Program for inherited metabolic and endocrinologic disorders was introduced in 1966. The program continuously evolved by expanding the screening panel from phenylketonuria and galactosemia to congenital hypothyroidism, biotinidase deficiency, cystic fibrosis, and congenital adrenal hyperplasia. In 2002, the introduction of tandem mass spectrometry (MS/MS) substantially increased the number of detectable inborn errors of metabolism and now includes disorders of fatty acid oxidation, organic acidurias and various disorders of amino acid metabolism.
OBJECTIVE: in this study we report our eight years experience with MS/MS in Austria and give an overview of the incidence of diseases, organization, updates on methods and current development and future aspects.
METHODS: a total of 622,489 newborns were screened by MS/MS for more than 20 diseases in Austria between April 2002 and December 2009. Dried blood spot samples were collected and sent to the National Laboratory for Newborn Screening located at the Medical University of Vienna, Vienna, Austria.
RESULTS: The resulting overall prevalence of inherited metabolic disorder identified by MS/MS was 1:2855, including 125 newborns with amino acidemias (1:4,980), 46 with organic acidurias (1:13,532), and 47 with fatty acid oxidation disorders (1:13,244).
CONCLUSION: the introduction of MS/MS technology in Austria significantly increased the detection of inherited metabolic disorders that were previously not covered. A primary goal is the continuous effort by developing second-tier strategies with the inclusion of more specific markers in order to minimize the risk of false-negatives and to improve the positive predictive value of screening results. Early recognition of these disorders enables diagnosis and treatment before the onset of symptoms.

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Year:  2010        PMID: 20938748     DOI: 10.1007/s00508-010-1457-3

Source DB:  PubMed          Journal:  Wien Klin Wochenschr        ISSN: 0043-5325            Impact factor:   1.704


  33 in total

Review 1.  Population screening in the age of genomic medicine.

Authors:  Muin J Khoury; Linda L McCabe; Edward R B McCabe
Journal:  N Engl J Med       Date:  2003-01-02       Impact factor: 91.245

2.  Combined liquid chromatography-tandem mass spectrometry as an analytical method for high throughput screening for X-linked adrenoleukodystrophy and other peroxisomal disorders: preliminary findings.

Authors:  Walter C Hubbard; Ann B Moser; Silvia Tortorelli; Anita Liu; David Jones; Hugo Moser
Journal:  Mol Genet Metab       Date:  2006-07-07       Impact factor: 4.797

3.  Guideline for the diagnosis and management of glutaryl-CoA dehydrogenase deficiency (glutaric aciduria type I).

Authors:  S Kölker; E Christensen; J V Leonard; C R Greenberg; A B Burlina; A P Burlina; M Dixon; M Duran; S I Goodman; D M Koeller; E Müller; E R Naughten; E Neumaier-Probst; J G Okun; M Kyllerman; R A Surtees; B Wilcken; G F Hoffmann; P Burgard
Journal:  J Inherit Metab Dis       Date:  2007-01-03       Impact factor: 4.982

4.  Rapid diagnosis of phenylketonuria by quantitative analysis for phenylalanine and tyrosine in neonatal blood spots by tandem mass spectrometry.

Authors:  D H Chace; D S Millington; N Terada; S G Kahler; C R Roe; L F Hofman
Journal:  Clin Chem       Date:  1993-01       Impact factor: 8.327

5.  The tandem mass spectrometry newborn screening experience in North Carolina: 1997-2005.

Authors:  D M Frazier; D S Millington; S E McCandless; D D Koeberl; S D Weavil; S H Chaing; J Muenzer
Journal:  J Inherit Metab Dis       Date:  2006-02       Impact factor: 4.982

6.  Mild citrullinemia in Caucasians is an allelic variant of argininosuccinate synthetase deficiency (citrullinemia type 1).

Authors:  Johannes Häberle; Silke Pauli; Eva Schmidt; Barbara Schulze-Eilfing; Christoph Berning; Hans Georg Koch
Journal:  Mol Genet Metab       Date:  2003-11       Impact factor: 4.797

7.  Tandem mass spectrometry for the direct assay of lysosomal enzymes in dried blood spots: application to screening newborns for mucopolysaccharidosis I.

Authors:  Sophie Blanchard; Martin Sadilek; C Ronald Scott; Frantisek Turecek; Michael H Gelb
Journal:  Clin Chem       Date:  2008-12       Impact factor: 8.327

8.  Screening newborns for inborn errors of metabolism by tandem mass spectrometry.

Authors:  Bridget Wilcken; Veronica Wiley; Judith Hammond; Kevin Carpenter
Journal:  N Engl J Med       Date:  2003-06-05       Impact factor: 91.245

9.  Early detection of Pompe disease by newborn screening is feasible: results from the Taiwan screening program.

Authors:  Yin-Hsiu Chien; Shu-Chuan Chiang; Xiaokui Kate Zhang; Joan Keutzer; Ni-Chung Lee; Ai-Chu Huang; Chun-An Chen; Mei-Hwan Wu; Pei-Hsin Huang; Fu-Jen Tsai; Yuan-Tsong Chen; Wuh-Liang Hwu
Journal:  Pediatrics       Date:  2008-06-02       Impact factor: 7.124

10.  Newborn population screening for classic homocystinuria by determination of total homocysteine from Guthrie cards.

Authors:  Hongying Gan-Schreier; Moustafa Kebbewar; Junmin Fang-Hoffmann; Julia Wilrich; Ghassan Abdoh; Tawfeg Ben-Omran; Noora Shahbek; Abdulbari Bener; Hilal Al Rifai; Abdul Latif Al Khal; Martin Lindner; Johannes Zschocke; Georg F Hoffmann
Journal:  J Pediatr       Date:  2009-11-14       Impact factor: 4.406

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  21 in total

1.  [Newborn screening as a predictive genetic test: principles and challenges].

Authors:  Johannes Zschocke
Journal:  Wien Med Wochenschr       Date:  2012-03-28

Review 2.  Acute management of propionic acidemia.

Authors:  Kimberly A Chapman; Andrea Gropman; Erin MacLeod; Kathy Stagni; Marshall L Summar; Keiko Ueda; Nicholas Ah Mew; Jill Franks; Eddie Island; Dietrich Matern; Loren Pena; Brittany Smith; V Reid Sutton; Tiina Urv; Charles Venditti; Anupam Chakrapani
Journal:  Mol Genet Metab       Date:  2011-09-24       Impact factor: 4.797

3.  Spectrum analysis of common inherited metabolic diseases in Chinese patients screened and diagnosed by tandem mass spectrometry.

Authors:  Lianshu Han; Feng Han; Jun Ye; Wenjuan Qiu; Huiwen Zhang; Xiaolan Gao; Yu Wang; Wenjun Ji; Xuefan Gu
Journal:  J Clin Lab Anal       Date:  2014-05-05       Impact factor: 2.352

4.  Networking Across Borders for Individuals with Organic Acidurias and Urea Cycle Disorders: The E-IMD Consortium.

Authors:  Stefan Kölker; Dries Dobbelaere; Johannes Häberle; Peter Burgard; Florian Gleich; Marshall L Summar; Steven Hannigan; Samantha Parker; Anupam Chakrapani; Matthias R Baumgartner
Journal:  JIMD Rep       Date:  2015-02-22

5.  Prevalence of tetrahydrobiopterine (BH4)-responsive alleles among Austrian patients with PAH deficiency: comprehensive results from molecular analysis in 147 patients.

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Journal:  J Inherit Metab Dis       Date:  2012-04-25       Impact factor: 4.982

6.  [Screening and clinical analysis of isovaleric acidemia newborn in Zhejiang province].

Authors:  Zhenzhen Hu; Jianbin Yang; Lingwei Hu; Yunfei Zhao; Chao Zhang; Rulai Yang; Xinwen Huang
Journal:  Zhejiang Da Xue Xue Bao Yi Xue Ban       Date:  2020-10-25

7.  Birth Prevalence of Fatty Acid β-Oxidation Disorders in Iberia.

Authors:  Hugo Rocha; Daisy Castiñeiras; Carmen Delgado; José Egea; Raquel Yahyaoui; Yolanda González; Manuel Conde; Inmaculada González; Inmaculada Rueda; Luis Rello; Laura Vilarinho; José Cocho
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8.  Expanded newborn screening in New South Wales: missed cases.

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Journal:  J Inherit Metab Dis       Date:  2014-06-27       Impact factor: 4.982

Review 9.  Impact of newborn screening on the reported incidence and clinical outcomes associated with medium- and long-chain fatty acid oxidation disorders.

Authors:  Deborah Marsden; Camille L Bedrosian; Jerry Vockley
Journal:  Genet Med       Date:  2021-01-25       Impact factor: 8.822

10.  Cost-effectiveness analysis of universal newborn screening for medium chain acyl-CoA dehydrogenase deficiency in France.

Authors:  Françoise F Hamers; Catherine Rumeau-Pichon
Journal:  BMC Pediatr       Date:  2012-06-08       Impact factor: 2.125

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