| Literature DB >> 26205433 |
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