Literature DB >> 9797590

Prospective surveillance study of medium chain acyl-CoA dehydrogenase deficiency in the UK.

R J Pollitt1, J V Leonard.   

Abstract

BACKGROUND: Medium chain acyl-CoA dehydrogenase (MCAD) deficiency is a common disorder of fatty acid oxidation in north west Europe. It is very variable in its clinical consequences and is believed to be considerably underdiagnosed.
OBJECTIVE: To investigate the diagnosis and outcome of MCAD deficiency in the UK.
METHOD: A prospective surveillance study through the British Paediatric Surveillance Unit.
RESULTS: Of 62 affected individuals identified, 57 were from England, giving an incidence of 4.5 cases/100,000 births. Forty six cases presented with an acute illness (10 of whom died), 13 cases were identified because of family history, and three for other reasons. Six of the survivors were neurologically impaired.
CONCLUSIONS: Despite increased clinical awareness, the mortality and morbidity from MCAD deficiency remain high. The frequency and severity of the disease support the case for the introduction of universal neonatal screening in England and Scotland.

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Year:  1998        PMID: 9797590      PMCID: PMC1717653          DOI: 10.1136/adc.79.2.116

Source DB:  PubMed          Journal:  Arch Dis Child        ISSN: 0003-9888            Impact factor:   3.791


  16 in total

Review 1.  Neonatal screening for inborn errors of metabolism: cost, yield and outcome.

Authors:  R J Pollitt; A Green; C J McCabe; A Booth; N J Cooper; J V Leonard; J Nicholl; P Nicholson; J R Tunaley; N K Virdi
Journal:  Health Technol Assess       Date:  1997       Impact factor: 4.014

2.  Neonatal presentation of medium-chain acyl-CoA dehydrogenase deficiency in two families.

Authors:  J M Kirk; I A Laing; N Smith; W S Uttley
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

3.  Neonatal symptoms in medium chain acyl coenzyme A dehydrogenase deficiency.

Authors:  B Wilcken; K H Carpenter; J Hammond
Journal:  Arch Dis Child       Date:  1993-09       Impact factor: 3.791

4.  Most cases of medium-chain acyl-CoA dehydrogenase deficiency escape detection in France.

Authors:  B Fromenty; A Mansouri; J P Bonnefont; F Courtois; A Munnich; D Rabier
Journal:  Hum Genet       Date:  1996-03       Impact factor: 4.132

5.  A survey of the newborn populations in Belgium, Germany, Poland, Czech Republic, Hungary, Bulgaria, Spain, Turkey, and Japan for the G985 variant allele with haplotype analysis at the medium chain Acyl-CoA dehydrogenase gene locus: clinical and evolutionary consideration.

Authors:  K Tanaka; N Gregersen; A Ribes; J Kim; S Kølvraa; V Winter; H Eiberg; G Martinez; T Deufel; B Leifert; R Santer; B François; E Pronicka; A László; S Kmoch; I Kremensky; L Kalaydjicva; I Ozalp; M Ito
Journal:  Pediatr Res       Date:  1997-02       Impact factor: 3.756

6.  Heterogeneity for mutations in medium chain acyl-CoA dehydrogenase deficiency in the UK population.

Authors:  D Curtis; A I Blakemore; P C Engel; D Macgregor; G Besley; S Kolvraa; N Gregersen
Journal:  Clin Genet       Date:  1991-10       Impact factor: 4.438

7.  Scottish frequency of the common G985 mutation in the medium-chain acyl-CoA dehydrogenase (MCAD) gene and the role of MCAD deficiency in sudden infant death syndrome (SIDS).

Authors:  M Dundar; W G Lanyon; J M Connor
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

8.  Medium-chain acyl-coenzyme A dehydrogenase deficiency: clinical course in 120 affected children.

Authors:  A K Iafolla; R J Thompson; C R Roe
Journal:  J Pediatr       Date:  1994-03       Impact factor: 4.406

Review 9.  Medium chain acyl-CoA dehydrogenase deficiency.

Authors:  E H Touma; C Charpentier
Journal:  Arch Dis Child       Date:  1992-01       Impact factor: 3.791

10.  Morbidity and mortality in medium chain acyl coenzyme A dehydrogenase deficiency.

Authors:  B Wilcken; J Hammond; M Silink
Journal:  Arch Dis Child       Date:  1994-05       Impact factor: 3.791

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  31 in total

1.  Screening for inherited metabolic disease in newborn infants using tandem mass spectrometry.

Authors:  James V Leonard; Carol Dezateux
Journal:  BMJ       Date:  2002-01-05

Review 2.  The clinical manifestation of MCAD deficiency: challenges towards adulthood in the screened population.

Authors:  Ulrich A Schatz; Regina Ensenauer
Journal:  J Inherit Metab Dis       Date:  2010-06-08       Impact factor: 4.982

Review 3.  Newborn screening for medium chain acyl-CoA dehydrogenase deficiency: evaluating the effects on outcome.

Authors:  Carol Dezateux
Journal:  Eur J Pediatr       Date:  2003-11-20       Impact factor: 3.183

4.  Newborn and childhood screening programmes: criteria, evidence, and current policy.

Authors:  D A C Elliman; C Dezateux; H E Bedford
Journal:  Arch Dis Child       Date:  2002-07       Impact factor: 3.791

Review 5.  Data required for the evaluation of newborn screening programmes.

Authors:  Bernhard Liebl; Uta Nennstiel-Ratzel; Adelbert Roscher; Rüdiger von Kries
Journal:  Eur J Pediatr       Date:  2003-11-13       Impact factor: 3.183

6.  Beyond counting cases: public health impacts of national Paediatric Surveillance Units.

Authors:  D Grenier; E J Elliott; Y Zurynski; R Rodrigues Pereira; M Preece; R Lynn; R von Kries; H Zimmermann; N P Dickson; D Virella
Journal:  Arch Dis Child       Date:  2006-12-11       Impact factor: 3.791

7.  Frequency of metabolic disorders: more than one needle in the haystack.

Authors:  P J Lee; P Cook
Journal:  Arch Dis Child       Date:  2006-11       Impact factor: 3.791

Review 8.  Surveillance for rare disorders by the BPSU. The British Paediatric Surveillance Unit.

Authors:  C Verity; M Preece
Journal:  Arch Dis Child       Date:  2002-10       Impact factor: 3.791

9.  Expanded newborn screening: Lessons learned from MCAD deficiency.

Authors:  Sarah Dyack
Journal:  Paediatr Child Health       Date:  2004-04       Impact factor: 2.253

10.  Abnormal Newborn Screening in a Healthy Infant of a Mother with Undiagnosed Medium-Chain Acyl-CoA Dehydrogenase Deficiency.

Authors:  Lise Aksglaede; Mette Christensen; Jess H Olesen; Morten Duno; Rikke K J Olsen; Brage S Andresen; David M Hougaard; Allan M Lund
Journal:  JIMD Rep       Date:  2015-03-13
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