Literature DB >> 15626540

Clinical onset and prognosis of Asian children with organic acidemias, as detected by analysis of urinary organic acids using GC/MS, instead of mass screening.

Daisuke Hori1, Yuki Hasegawa, Masahiko Kimura, Yanling Yang, Iswar C Verma, Seiji Yamaguchi.   

Abstract

Organic acidemias (OAs) have been detected worldwide in symptomatic patients using gas chromatography mass spectrometry. We diagnosed 188 Asian cases of OAs by analysis of urinary organic acids and investigated their clinical onset and outcome. Methylmalonic acidemia (MMA) was most common (74 cases), followed by propionic acidemia (23 cases), ornitine transcarbamylase deficiency (22 cases), and multiple carboxylase deficiency (15 cases). For these 188 patients, onset was most frequent in the neonatal period or early infancy. Approximately 30% of the patients had a family history of similar symptoms or diseases. Although the outcome of OA patients varied, patients with early onset generally had poor outcomes despite early detection. Of the 45 MMA patients whose clinical data were available, 25 were clinically vitamin B12-responsive, while the remaining 20 were non-responsive. A favorable outcome was obtained in 7 of the 25 B12-responsive patients, and in only 3 of the 20 B12-nonresponsive patients. It was suggested that even in B12-responsive MMA cases, earlier detection and B12 therapy were needed to improve the prognosis. We concluded that detection of such patients at the presymptomatic stages using newborn mass screening is essential for prognosis improvement with OAs.

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Year:  2005        PMID: 15626540     DOI: 10.1016/j.braindev.2004.04.004

Source DB:  PubMed          Journal:  Brain Dev        ISSN: 0387-7604            Impact factor:   1.961


  16 in total

1.  Mutation Profile of the MUT Gene in Chinese Methylmalonic Aciduria Patients.

Authors:  Mei-Ying Liu; Tze-Tze Liu; Yang-Ling Yang; Ying-Chen Chang; Ya-Ling Fan; Shu-Fen Lee; Yu-Ting Teng; Szu-Hui Chiang; Dau-Ming Niu; Shio-Jean Lin; Mei-Chun Chao; Shuan-Pei Lin; Lian-Shu Han; Yu Qi; Kwang-Jen Hsiao
Journal:  JIMD Rep       Date:  2012-01-31

2.  Methylmalonic Acidemia Diagnosis by Laboratory Methods.

Authors:  Fatemeh Keyfi; Saeed Talebi; Abdol-Reza Varasteh
Journal:  Rep Biochem Mol Biol       Date:  2016-10

3.  Organic acidurias: an updated review.

Authors:  Kannan Vaidyanathan; M P Narayanan; D M Vasudevan
Journal:  Indian J Clin Biochem       Date:  2011-04-29

4.  Diagnosis of major organic acidurias in children: two years experience at a tertiary care centre.

Authors:  M P Narayanan; Vaidyanathan Kannan; K P Vinayan; D M Vasudevan
Journal:  Indian J Clin Biochem       Date:  2011-02-01

Review 5.  'Classical' organic acidurias, propionic aciduria, methylmalonic aciduria and isovaleric aciduria: long-term outcome and effects of expanded newborn screening using tandem mass spectrometry.

Authors:  Carlo Dionisi-Vici; Federica Deodato; Wulf Röschinger; William Rhead; Bridget Wilcken
Journal:  J Inherit Metab Dis       Date:  2006 Apr-Jun       Impact factor: 4.982

6.  Analysis of methylcitrate in dried blood spots by liquid chromatography-tandem mass spectrometry.

Authors:  Osama Y Al-Dirbashi; Nathan McIntosh; Christine McRoberts; Larry Fisher; Mohamed S Rashed; Nawal Makhseed; Michael T Geraghty; Tomofumi Santa; Pranesh Chakraborty
Journal:  JIMD Rep       Date:  2014-07-06

7.  Severe Neonatal Metabolic Decompensation in Methylmalonic Acidemia Caused by CblD Defect.

Authors:  R Parini; F Furlan; A Brambilla; D Codazzi; S Vedovati; C Corbetta; T Fedeli; B Merinero; B Pérez; M Ugarte
Journal:  JIMD Rep       Date:  2013-05-19

Review 8.  Combined liver-kidney transplant for the management of methylmalonic aciduria: a case report and review of the literature.

Authors:  Peter J Mc Guire; Elizabeth Lim-Melia; George A Diaz; Kimiyo Raymond; Alexandra Larkin; Melissa P Wasserstein; Claude Sansaricq
Journal:  Mol Genet Metab       Date:  2007-10-26       Impact factor: 4.797

9.  Propionic acidemia and optic neuropathy: a report of two cases.

Authors:  Carolina Arias; Erna Raimann; Pilar Peredo; Juan Francisco Cabello; Gabriela Castro; Alf Valiente; Alicia de la Parra; Paulina Bravo; Cecilia Okuma; Verónica Cornejo
Journal:  JIMD Rep       Date:  2013-07-02

10.  Insulin-resistant hyperglycaemia complicating neonatal onset of methylmalonic and propionic acidaemias.

Authors:  L Filippi; E Gozzini; C Cavicchi; A Morrone; P Fiorini; G Donzelli; S Malvagia; G la Marca
Journal:  J Inherit Metab Dis       Date:  2009-07-09       Impact factor: 4.982

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