Mitochondrial Fatty Acid β-Oxidation and Resveratrol Effect in Fibroblasts from Patients with Autism Spectrum Disorder
| Autor: | Indrapal Singh, Jean Bastin, Patrick John McCarty, Rita Barone, Fatima Djouadi, Richard E. Frye, Mohammad A. Karim, Antonino Casabona, Amrit Ammanamanchi, Rose Shannon, Renata Rizzo, Leanna Delhey |
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| Přispěvatelé: | Gestionnaire, Hal Sorbonne Université, University of Catania [Italy], Centre de Recherche des Cordeliers (CRC (UMR_S_1138 / U1138)), École pratique des hautes études (EPHE), Université Paris sciences et lettres (PSL)-Université Paris sciences et lettres (PSL)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Sorbonne Université (SU)-Université Paris Cité (UPCité), Phoenix Children's Hospital, Arkansas Children's Research Institute, Université Paris sciences et lettres (PSL)-Université Paris sciences et lettres (PSL)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Sorbonne Université (SU)-Université de Paris (UP) |
| Jazyk: | angličtina |
| Rok vydání: | 2021 |
| Předmět: |
0301 basic medicine
medicine.medical_specialty genetic structures [SDV]Life Sciences [q-bio] Medicine (miscellaneous) autism spectrum disorder Resveratrol resveratrol behavioral disciplines and activities Article Mitochondrial fatty acid 03 medical and health sciences Social Responsiveness Scale chemistry.chemical_compound 0302 clinical medicine Internal medicine mental disorders energy metabolism medicine In patient Fibroblast Beta oxidation fatty acid oxidation business.industry Mean age medicine.disease [SDV] Life Sciences [q-bio] 030104 developmental biology medicine.anatomical_structure Endocrinology chemistry acyl-carnitines Autism spectrum disorder Medicine business 030217 neurology & neurosurgery |
| Zdroj: | Journal of Personalized Medicine Journal of Personalized Medicine, MDPI, 2021, 11 (6), pp.510. ⟨10.3390/jpm11060510⟩ Journal of Personalized Medicine, Vol 11, Iss 510, p 510 (2021) Volume 11 Issue 6 |
| ISSN: | 2075-4426 |
| Popis: | Patients with autism spectrum disorder (ASD) may have an increase in blood acyl-carnitine (AC) concentrations indicating a mitochondrial fatty acid β-oxidation (mtFAO) impairment. However, there are no data on systematic mtFAO analyses in ASD. We analyzed tritiated palmitate oxidation rates in fibroblasts from patients with ASD before and after resveratrol (RSV) treatment, according to methods used for the diagnosis of congenital defects in mtFAO. ASD participants (N = 10, 60% male mean age (SD) 7.4 (3.2) years) were divided in two age-equivalent groups based on the presence (N = 5) or absence (N = 5) of elevated blood AC levels. In addition, electron transport chain (ETC) activity in fibroblasts and muscle biopsies and clinical characteristics were compared between the ASD groups. Baseline fibroblast mtFAO was not significantly different in patients in comparison with control values. However, ASD patients with elevated AC exhibited significantly decreased mtFAO rates, muscle ETC complex II activity, and fibroblast ETC Complex II/III activity (p < 0.05), compared with patients without an AC signature. RSV significantly increased the mtFAO activity in all study groups (p = 0.001). The highest mtFAO changes in response to RSV were observed in fibroblasts from patients with more severe symptoms on the Social Responsiveness Scale total (p = 0.001) and Awareness, Cognition, Communication and Motivation subscales (all p < 0.01). These findings suggested recognition of an ASD patient subset characterized by an impaired mtFAO flux associated with abnormal blood AC. The study elucidated that RSV significantly increased fibroblast mtFAO irrespective of plasma AC status, and the highest changes to RSV effects on mtFAO were observed in the more severely affected patients. |
| Databáze: | OpenAIRE |
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