Literature DB >> 21113738

Amino acid metabolism in patients with propionic acidaemia.

Sabine Scholl-Bürgi1, Jörn Oliver Sass, Johannes Zschocke, Daniela Karall.   

Abstract

Propionic acidaemia (PA) is an inborn error of intermediary metabolism caused by deficiency of propionyl-CoA carboxylase. The metabolic block leads to a profound failure of central metabolic pathways, including the urea and the citric acid cycles. This review will focus on changes in amino acid metabolism in this inborn disorder of metabolism. The first noted disturbance of amino acid metabolism was hyperglycinaemia, which is detectable in nearly all PA patients. Additionally, hyperlysinaemia is a common observation. In contrast, concentrations of branched chain amino acids, especially of isoleucine, are frequently reported as decreased. These non-proportional changes of branched-chain amino acids (BCAAs) compared with aromatic amino acids are also reflected by the Fischer's ratio (concentration ratio of BCAAs to aromatic amino acids), which is decreased in PA patients. As restricted dietary intake of valine and isoleucine as precursors of propionyl-CoA is part of the standard treatment in PA, decreased plasma concentrations of BCAAs may be a side effect of treatment. The concentration changes of the nitrogen scavenger glutamine have to be interpreted in the light of ammonia levels. In contrast to other hyperammonaemic syndromes, in PA plasma glutamine concentrations do not increase in hyperammonaemia, whereas CSF glutamine concentrations are elevated. Despite lactic acidaemia in PA patients, hyperalaninaemia is only rarely reported. The mechanisms underlying the observed changes in amino acid metabolism have not yet been elucidated, but most of the changes can be at least partly interpreted as consequence of disturbance of anaplerosis.

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Year:  2010        PMID: 21113738     DOI: 10.1007/s10545-010-9245-9

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  39 in total

1.  Idiopathic hyperglycinemia and hyperglycinuria: a new disorder of amino acid metabolism. I.

Authors:  B CHILDS; W L NYHAN; M BORDEN; L BARD; R E COOKE
Journal:  Pediatrics       Date:  1961-04       Impact factor: 7.124

2.  Propionic acidemia revisited: a workshop report.

Authors:  J O Sass; M Hofmann; D Skladal; E Mayatepek; B Schwahn; W Sperl
Journal:  Clin Pediatr (Phila)       Date:  2004 Nov-Dec       Impact factor: 1.168

Review 3.  Stable isotope studies in propionic and methylmalonic acidaemia.

Authors:  J V Leonard
Journal:  Eur J Pediatr       Date:  1997-08       Impact factor: 3.183

4.  Identification of the alpha-aminoadipic semialdehyde synthase gene, which is defective in familial hyperlysinemia.

Authors:  K A Sacksteder; B J Biery; J C Morrell; B K Goodman; B V Geisbrecht; R P Cox; S J Gould; M T Geraghty
Journal:  Am J Hum Genet       Date:  2000-04-20       Impact factor: 11.025

5.  Pseudo-cystinuria-lysinuria in neonatal propionic acidemia.

Authors:  P Parvy; J Bardet; D Rabier; P Kamoun
Journal:  Clin Chem       Date:  1988-10       Impact factor: 8.327

6.  Propionic acidaemia: clinical, biochemical and therapeutic aspects. Experience in 30 patients.

Authors:  W Lehnert; W Sperl; T Suormala; E R Baumgartner
Journal:  Eur J Pediatr       Date:  1994       Impact factor: 3.183

7.  Effect of 2-methylcitrate on citrate metabolism: implications for the management of patients with propionic acidemia and methylmalonic aciduria.

Authors:  S Cheema-Dhadli; C C Leznoff; M L Halperin
Journal:  Pediatr Res       Date:  1975-12       Impact factor: 3.756

Review 8.  Branched chain amino acids in heptatic encephalopathy.

Authors:  J Howard James
Journal:  Am J Surg       Date:  2002-04       Impact factor: 2.565

Review 9.  Ammonia toxicity to the brain and creatine.

Authors:  Claude Bachmann; Olivier Braissant; Anne-Marie Villard; Olivier Boulat; Hugues Henry
Journal:  Mol Genet Metab       Date:  2004-04       Impact factor: 4.797

10.  Amino acid cerebrospinal fluid/plasma ratios in children: influence of age, gender, and antiepileptic medication.

Authors:  Sabine Scholl-Bürgi; Edda Haberlandt; Peter Heinz-Erian; Florian Deisenhammer; Ursula Albrecht; Sara Baumgartner Sigl; Markus Rauchenzauner; Hanno Ulmer; Daniela Karall
Journal:  Pediatrics       Date:  2008-03-10       Impact factor: 7.124

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  14 in total

1.  Anaplerotic therapy in propionic acidemia.

Authors:  Nicola Longo; Leisa B Price; Eduard Gappmaier; Nancy L Cantor; Sharon L Ernst; Carrie Bailey; Marzia Pasquali
Journal:  Mol Genet Metab       Date:  2017-07-12       Impact factor: 4.797

2.  Tricarboxylic acid cycle enzyme activities in a mouse model of methylmalonic aciduria.

Authors:  Parith Wongkittichote; Gary Cunningham; Marshall L Summar; Elena Pumbo; Patrick Forny; Matthias R Baumgartner; Kimberly A Chapman
Journal:  Mol Genet Metab       Date:  2019-10-17       Impact factor: 4.797

3.  Revealing the protein propionylation activity of the histone acetyltransferase MOF (males absent on the first).

Authors:  Zhen Han; Hong Wu; Sunjoo Kim; Xiangkun Yang; Qianjin Li; He Huang; Houjian Cai; Michael G Bartlett; Aiping Dong; Hong Zeng; Peter J Brown; Xiang-Jiao Yang; Cheryl H Arrowsmith; Yingming Zhao; Y George Zheng
Journal:  J Biol Chem       Date:  2018-01-10       Impact factor: 5.157

Review 4.  α-Ketoglutaramate: an overlooked metabolite of glutamine and a biomarker for hepatic encephalopathy and inborn errors of the urea cycle.

Authors:  Arthur J L Cooper; Tomiko Kuhara
Journal:  Metab Brain Dis       Date:  2013-11-14       Impact factor: 3.584

Review 5.  Methylmalonic and propionic acidemias: clinical management update.

Authors:  Jamie L Fraser; Charles P Venditti
Journal:  Curr Opin Pediatr       Date:  2016-12       Impact factor: 2.856

Review 6.  "Classical organic acidurias": diagnosis and pathogenesis.

Authors:  Guglielmo Rd Villani; Giovanna Gallo; Emanuela Scolamiero; Francesco Salvatore; Margherita Ruoppolo
Journal:  Clin Exp Med       Date:  2016-09-09       Impact factor: 3.984

Review 7.  Structure and function of biotin-dependent carboxylases.

Authors:  Liang Tong
Journal:  Cell Mol Life Sci       Date:  2012-08-07       Impact factor: 9.261

8.  Pantothenate kinase activation relieves coenzyme A sequestration and improves mitochondrial function in mice with propionic acidemia.

Authors:  Chitra Subramanian; Matthew W Frank; Rajendra Tangallapally; Mi-Kyung Yun; Anne Edwards; Stephen W White; Richard E Lee; Charles O Rock; Suzanne Jackowski
Journal:  Sci Transl Med       Date:  2021-09-15       Impact factor: 17.956

Review 9.  Single amino acid supplementation in aminoacidopathies: a systematic review.

Authors:  Danique van Vliet; Terry G J Derks; Margreet van Rijn; Martijn J de Groot; Anita MacDonald; M Rebecca Heiner-Fokkema; Francjan J van Spronsen
Journal:  Orphanet J Rare Dis       Date:  2014-01-13       Impact factor: 4.123

10.  Dietary practices in propionic acidemia: A European survey.

Authors:  A Daly; A Pinto; S Evans; M F Almeida; M Assoun; A Belanger-Quintana; S M Bernabei; S Bollhalder; D Cassiman; H Champion; H Chan; J Dalmau; F de Boer; C de Laet; A de Meyer; A Desloovere; A Dianin; M Dixon; K Dokoupil; S Dubois; F Eyskens; A Faria; I Fasan; E Favre; F Feillet; A Fekete; G Gallo; C Gingell; J Gribben; K Kaalund Hansen; N M Ter Horst; C Jankowski; R Janssen-Regelink; I Jones; C Jouault; G E Kahrs; I L Kok; A Kowalik; C Laguerre; S Le Verge; R Lilje; C Maddalon; D Mayr; U Meyer; A Micciche; U Och; M Robert; J C Rocha; H Rogozinski; C Rohde; K Ross; I Saruggia; A Schlune; K Singleton; E Sjoqvist; R Skeath; L H Stolen; A Terry; C Timmer; L Tomlinson; A Tooke; K Vande Kerckhove; E van Dam; T van den Hurk; L van der Ploeg; M van Driessche; M van Rijn; A van Wegberg; C Vasconcelos; H Vestergaard; I Vitoria; D Webster; F J White; L White; H Zweers; A MacDonald
Journal:  Mol Genet Metab Rep       Date:  2017-10-03
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