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Peak oxygen uptake (VO<subscript>2peak</subscript>) across childhood, adolescence and young adulthood in Barth syndrome: Data from cross-sectional and longitudinal studies.
In: PLoS ONE, Jg. 13 (2018-05-24), Heft 5, S. 1-12
Online
academicJournal
Zugriff:
Barth syndrome (BTHS) is an ultra-rare, X-linked recessive disorder characterized by cardio-skeletal myopathy, exercise intolerance, and growth delay. Oxygen uptake during peak exercise (VO2peak) has been shown to be severely limited in individuals with BTHS however; the trajectory of VO2peak from childhood to young adulthood is unknown. The objective of this study was to describe VO2peak from childhood through young adulthood in BTHS. Methods and Materials: VO2peak over time was presented through cross-sectional (n = 33 participants) and a longitudinal analyses (n = 12 participants). Retrospective data were obtained through maximal exercise testing on a cycle ergometer from individuals with BTHS who were or are currently enrolled in a research study during July 2006-September 2017. Participants included in the cross-sectional analysis were divided into 3 groups for analysis: 1) children (n = 13), 2) adolescents (n = 8), and 3) young adults (n = 12). Participants in the longitudinal analysis had at least two exercise tests over a span of 2–9 years. Results: VO2peak relative to body weight (ml/kgBW/min), fat-free mass (FFM) and by percent of predicted VO2peak obtained were not significantly different between children, adolescents and young adults. VO2peak did not longitudinally change over a mean time of ~5 years in late adolescent and young adult participants with repeated tests. A model including both cardiac and skeletal muscle variables best predicted VO2peak. Conclusions: In conclusion, VO2peak relative to body weight and fat-free mass demonstrates short- and long-term stability from childhood to young adulthood in BTHS with some variability among individuals. [ABSTRACT FROM AUTHOR]
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Peak oxygen uptake (VO<subscript>2peak</subscript>) across childhood, adolescence and young adulthood in Barth syndrome: Data from cross-sectional and longitudinal studies.
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Autor/in / Beteiligte Person: | Cade, William Todd ; Bohnert, Kathryn L. ; Reeds, Dominic N. ; Peterson, Linda R. ; Bittel, Adam J. ; Bashir, Adil ; Byrne, Barry J. ; Taylor, Carolyn L. |
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Zeitschrift: | PLoS ONE, Jg. 13 (2018-05-24), Heft 5, S. 1-12 |
Veröffentlichung: | 2018 |
Medientyp: | academicJournal |
ISSN: | 1932-6203 (print) |
DOI: | 10.1371/journal.pone.0197776 |
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