Literature DB >> 7957396

Neurological manifestations of organic acid disorders.

G F Hoffmann1, K M Gibson, F K Trefz, W L Nyhan, H J Bremer, D Rating.   

Abstract

Neurological manifestations are very common and can be the leading and/or presenting feature in organic acid disorders, sometimes in the absence of metabolic derangement. Review of the time course and presentation of neurological disease in organic acid disorders reveals characteristic clinical findings of ataxia, myoclonus, extrapyramidal symptoms, metabolic stroke and megalencephaly. A group of organic acid disorders presents exclusively with neurological symptoms. These include glutaryl-CoA dehydrogenase deficiency (glutaric aciduria type I), succinic semialdehyde dehydrogenase deficiency (4-hydroxybutyric aciduria), mevalonic aciduria, N-acetylaspartic aciduria (Canavan disease) and L-2-hydroxyglutaric aciduria. As a group these "cerebral" organic acid disorders appear to remain often undiagnosed and their true incidence is much less well-known than that of the "classical" organic acid disorders. Unfortunately, stringent guidelines for a clinical preselection of neuropaediatric patients to be investigated for organic acid disorders cannot be provided. Today, screening for neurometabolic disorders should be as comprehensive as possible and include determinations of amino acids, purines and pyrimidines and markers of peroxisomal function in addition to organic acid analysis.

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Mesh:

Year:  1994        PMID: 7957396     DOI: 10.1007/BF02138786

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


  29 in total

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Authors:  G F Hoffmann; C Jakobs; D Rating; L Sweetman; F K Trefz
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Journal:  AJNR Am J Neuroradiol       Date:  1991 May-Jun       Impact factor: 3.825

3.  The first adult case with 4-hydroxybutyric aciduria.

Authors:  C Jakobs; L M Smit; J Kneer; T Michael; K M Gibson
Journal:  J Inherit Metab Dis       Date:  1990       Impact factor: 4.982

4.  L-2-hydroxyglutaric acidemia: a novel inherited neurometabolic disease.

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Journal:  Ann Neurol       Date:  1992-07       Impact factor: 10.422

Review 5.  Prenatal diagnosis of inherited metabolic disorders by quantitation of characteristic metabolites in amniotic fluid: facts and future.

Authors:  C Jakobs; H J Ten Brink; F Stellaard
Journal:  Prenat Diagn       Date:  1990-04       Impact factor: 3.050

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7.  Variable clinical presentation in patients with defective E1 component of pyruvate dehydrogenase complex.

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8.  Aspartoacylase deficiency and N-acetylaspartic aciduria in patients with Canavan disease.

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Journal:  Am J Med Genet       Date:  1988-02

9.  Mevalonic aciduria--an inborn error of cholesterol and nonsterol isoprene biosynthesis.

Authors:  G Hoffmann; K M Gibson; I K Brandt; P I Bader; R S Wappner; L Sweetman
Journal:  N Engl J Med       Date:  1986-06-19       Impact factor: 91.245

Review 10.  Physiology and pathophysiology of organic acids in cerebrospinal fluid.

Authors:  G F Hoffmann; W Meier-Augenstein; S Stöckler; R Surtees; D Rating; W L Nyhan
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

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  19 in total

1.  Lactate dehydrogenase activity is inhibited by methylmalonate in vitro.

Authors:  Laura O Saad; Sandra R Mirandola; Evelise N Maciel; Roger F Castilho
Journal:  Neurochem Res       Date:  2006-05-09       Impact factor: 3.996

2.  The aetiology of neurological complications of organic acidaemias--a role for the blood-brain barrier.

Authors:  S Kölker; S W Sauer; R A H Surtees; J V Leonard
Journal:  J Inherit Metab Dis       Date:  2006-10-14       Impact factor: 4.982

3.  Molecular cloning, expression and catalytic activity of a human AKR7 member of the aldo-keto reductase superfamily: evidence that the major 2-carboxybenzaldehyde reductase from human liver is a homologue of rat aflatoxin B1-aldehyde reductase.

Authors:  L S Ireland; D J Harrison; G E Neal; J D Hayes
Journal:  Biochem J       Date:  1998-05-15       Impact factor: 3.857

4.  Organic acidurias: an updated review.

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

5.  Evidence that 2-methylacetoacetate induces oxidative stress in rat brain.

Authors:  Guilhian Leipnitz; Bianca Seminotti; Alexandre U Amaral; Carolina G Fernandes; Carlos S Dutra-Filho; Moacir Wajner
Journal:  Metab Brain Dis       Date:  2010-09-14       Impact factor: 3.584

6.  D-2-Hydroxyglutaric aciduria in a patient with a severe clinical phenotype and unusual MRI findings.

Authors:  M Wajne; C R Vargas; C Funayama; A Fernandez; M L C Elias; S I Goodman; C Jakobs; M S van der Knaap
Journal:  J Inherit Metab Dis       Date:  2002-02       Impact factor: 4.982

7.  Induction of a Proinflammatory Response in Cortical Astrocytes by the Major Metabolites Accumulating in HMG-CoA Lyase Deficiency: the Role of ERK Signaling Pathway in Cytokine Release.

Authors:  Carolina Gonçalves Fernandes; Marília Danyelle Nunes Rodrigues; Bianca Seminotti; Ana Laura Colín-González; Abel Santamaria; André Quincozes-Santos; Moacir Wajner
Journal:  Mol Neurobiol       Date:  2015-06-23       Impact factor: 5.590

8.  Insights into the molecular mechanisms of methylmalonic acidemia using microarray technology.

Authors:  Lianshu Han; Shengnan Wu; Feng Han; Xuefan Gu
Journal:  Int J Clin Exp Med       Date:  2015-06-15

9.  Methylmalonic acidaemia with bilateral globus pallidus involvement: a neuropathological study.

Authors:  A Larnaout; M A Mongalgi; N Kaabachi; D Khiari; A Debbabi; A Mebazza; M Ben Hamida; F Hentati
Journal:  J Inherit Metab Dis       Date:  1998-08       Impact factor: 4.982

Review 10.  The role of oxidative damage in the neuropathology of organic acidurias: insights from animal studies.

Authors:  M Wajner; A Latini; A T S Wyse; C S Dutra-Filho
Journal:  J Inherit Metab Dis       Date:  2004       Impact factor: 4.982

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