Literature DB >> 26205433

Coenzyme Q10 and Pyridoxal Phosphate Deficiency Is a Common Feature in Mucopolysaccharidosis Type III.

Dèlia Yubero1, Raquel Montero1,2, Mar O'Callaghan1, Mercè Pineda1, Silvia Meavilla1, Veronica Delgadillo1, Cristina Sierra1, Laura Altimira1, Plácido Navas2,3, Simon Pope4, Marcus Oppenheim4, Viruna Neergheen4, Arunabha Ghosh5, Phillipa Mills6, Peter Clayton6, Emma Footitt6, Maureen Cleary6, Iain Hargreaves4, Simon A Jones5, Simon Heales7,4,6, Rafael Artuch8,9.   

Abstract

Mucopolysaccharidoses (MPS) are a group of lysosomal storage disorders caused by deficiencies of lysosomal enzymes catalyzing degradation of glycosaminoglycans (GAGs). Previously, we reported a secondary plasma coenzyme Q10 (CoQ) deficiency in MPS patients. For this study, nine MPS patients were recruited in the Hospital Sant Joan de Déu (HSJD, Barcelona) and two patients in the Neurometabolic Unit, National Hospital (NMU, London), to explore the nutritional status of MPS type III patients by analyzing several vitamins and micronutrients in blood and in cerebrospinal fluid. Plasma CoQ and plasma and cerebrospinal fluid pyridoxal phosphate (PLP) content were analyzed by high-pressure liquid chromatography (HPLC) with electrochemical and fluorescence detection, respectively. We found that most MPS-III patients disclosed low plasma pyridoxal phosphate (PLP) values (seven out of nine) and also low plasma CoQ concentrations (eight out of nine). We observed significantly lower median values of PLP, tocopherol, and CoQ (Mann-Whitney U test, p = 0.006, p = 0.004, and p = 0.001, respectively) in MPS patients when compared with age-matched controls. Chi-square test showed a significant association between the fact of having low plasma PLP and CoQ values in the whole cohort of patients. Cerebrospinal fluid PLP values were clearly deficient in the two patients studied. In conclusion, we report a combined CoQ and PLP deficiency in MPS-III patients. These observations could be related to the complexity of the physiopathology of the disease. If our results are confirmed in larger series of patients, CoQ and PLP therapy could be trialed as coadjuvant therapy with the current MPS treatments.

Entities:  

Year:  2015        PMID: 26205433      PMCID: PMC5059182          DOI: 10.1007/8904_2015_421

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  19 in total

Review 1.  Molecular genetics of mucopolysaccharidosis type IIIA and IIIB: Diagnostic, clinical, and biological implications.

Authors:  G Yogalingam; J J Hopwood
Journal:  Hum Mutat       Date:  2001-10       Impact factor: 4.878

2.  Muscle coenzyme Q10 concentrations in patients with probable and definite diagnosis of respiratory chain disorders.

Authors:  R Montero; R Artuch; P Briones; A Nascimento; A García-Cazorla; M A Vilaseca; J A Sánchez-Alcázar; P Navas; J Montoya; M Pineda
Journal:  Biofactors       Date:  2005       Impact factor: 6.113

3.  Genistein supplementation in patients affected by Sanfilippo disease.

Authors:  Verónica Delgadillo; Maria Del Mar O'Callaghan; Rafael Artuch; Raquel Montero; Mercedes Pineda
Journal:  J Inherit Metab Dis       Date:  2011-05-10       Impact factor: 4.982

4.  Tocopherol in inborn errors of intermediary metabolism.

Authors:  D Moyano; M A Vilaseca; M Pineda; J Campistol; A Vernet; P Póo; R Artuch; C Sierra
Journal:  Clin Chim Acta       Date:  1997-07-25       Impact factor: 3.786

5.  Gastrointestinal pathology in a mouse model of mucopolysaccharidosis type IIIA.

Authors:  Ainslie L K Derrick Roberts; Gordon S Howarth; Wan Chin Liaw; Simon Moretta; Stamatiki Kritas; Kerry A Lymn; Roger Yazbeck; Cuong Tran; Janice M Fletcher; Ross N Butler; Sharon Byers
Journal:  J Cell Physiol       Date:  2009-05       Impact factor: 6.384

6.  Rapid HPLC measurement of thiamine and its phosphate esters in whole blood.

Authors:  Jun Lu; Elizabeth L Frank
Journal:  Clin Chem       Date:  2008-03-20       Impact factor: 8.327

7.  Mucopolysaccharidosis IIIB: oxidative damage and cytotoxic cell involvement in the neuronal pathogenesis.

Authors:  Guglielmo R D Villani; Carmela Di Domenico; Annapaola Musella; Francesca Cecere; Daniele Di Napoli; Paola Di Natale
Journal:  Brain Res       Date:  2009-05-03       Impact factor: 3.252

8.  Treatment effect of coenzyme Q(10) and an antioxidant cocktail in fibroblasts of patients with Sanfilippo disease.

Authors:  Leslie Matalonga; Angela Arias; María Josep Coll; Judit Garcia-Villoria; Laura Gort; Antonia Ribes
Journal:  J Inherit Metab Dis       Date:  2013-12-18       Impact factor: 4.982

9.  Enzyme replacement therapy for mucopolysaccharidosis I: a randomized, double-blinded, placebo-controlled, multinational study of recombinant human alpha-L-iduronidase (laronidase).

Authors:  James E Wraith; Lorne A Clarke; Michael Beck; Edwin H Kolodny; Gregory M Pastores; Joseph Muenzer; David M Rapoport; Kenneth I Berger; Stuart J Swiedler; Emil D Kakkis; Tanja Braakman; Elenie Chadbourne; Karen Walton-Bowen; Gerald F Cox
Journal:  J Pediatr       Date:  2004-05       Impact factor: 4.406

Review 10.  Mucopolysaccharidosis type III (Sanfilippo syndrome) and misdiagnosis of idiopathic developmental delay, attention deficit/hyperactivity disorder or autism spectrum disorder.

Authors:  Frits A Wijburg; Grzegorz Węgrzyn; Barbara K Burton; Anna Tylki-Szymańska
Journal:  Acta Paediatr       Date:  2013-02-06       Impact factor: 2.299

View more
  2 in total

Review 1.  The Inflammation in the Cytopathology of Patients With Mucopolysaccharidoses- Immunomodulatory Drugs as an Approach to Therapy.

Authors:  Anna-Maria Wiesinger; Brian Bigger; Roberto Giugliani; Maurizio Scarpa; Tobias Moser; Christina Lampe; Christoph Kampmann; Florian B Lagler
Journal:  Front Pharmacol       Date:  2022-05-13       Impact factor: 5.988

2.  Plasma coenzyme Q10 status is impaired in selected genetic conditions.

Authors:  Raquel Montero; Delia Yubero; Maria C Salgado; María Julieta González; Jaume Campistol; Maria Del Mar O'Callaghan; Mercè Pineda; Verónica Delgadillo; Joan Maynou; Guerau Fernandez; Julio Montoya; Eduardo Ruiz-Pesini; Silvia Meavilla; Viruna Neergheen; Angels García-Cazorla; Placido Navas; Iain Hargreaves; Rafael Artuch
Journal:  Sci Rep       Date:  2019-01-28       Impact factor: 4.379

  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.