Blom, K.J. (2017) Long-term trajectories of cardiorespiratory fitness in patients with juvenile dermatomyositis. thesis, Human Movement Sciences.
Full text available on request.Abstract
Background. Juvenile dermatomyositis (JDM) is a rare systemic autoimmune vasculopathy characterized by capillary inflammation that predominantly affects the musculoskeletal and cutaneous systems. Previous research demonstrated decreased cardiorespiratory fitness (CRF) in patients with JDM during active disease and remission. However, it is not known whether patients with JDM fully regain their physical fitness in the years after recovery, because longitudinal follow-up of CRF is currently lacking. Longitudinal data is vital in order to provide relevant clinical information concerning the need for additional physical activity or exercise training in the follow-up of this population. Objective. The aim of this study was to determine trajectories of CRF in patients with both monocyclic and chronic JDM compared with healthy children. The second aim was to indicate possible predictors of these trajectories. Methods. Thirty-six patients with JDM (median age (range) at diagnosis: 8.2 (3.0-15.4) years) treated in the paediatric rheumatology outpatient clinic of the Wilhelmina Children Hospital, were included. All patients performed multiple cardiopulmonary exercise tests between 2003 and 2016. Relevant CRF parameters were compared to healthy children and analysed, including peak oxygen uptake, maximal workload, mechanical efficacy and oxygen uptake at ventilatory anaerobic threshold. Trajectories up to 10 years after diagnosis were analysed and predictors (disease duration, age of onset, diagnostic delay and prednisone) of CRF outcome parameters determined by multilevel analyses. Results. Trajectories display significantly decreased CRF compared to healthy children (VO2peak -2.77 Z-score) during the active phase of the disease, and subsequent improvement of CRF (VO2peak -1.42 Z-score) during the first years after diagnosis. However, after the first years a decrease was observed over time in CRF (VO2peak -1.57 Z-score). Multilevel analysis regarding possible predictors indicates that a longer disease duration, younger age of onset and higher prednisone dose negatively influenced the outcome of CRF. Conclusions. Patients with both monocyclic and chronic JDM show decreased long-term CRF trajectories. Longer disease duration, younger age of onset and higher prednisone dose negatively influence CRF. This study stresses the need for regular evaluation of CRF and implementation of exercise interventions to improve CRF in patients with JDM. Key words: children, dermatomyositis, aerobic fitness, long-term
| Item Type: | Thesis (UNSPECIFIED) |
|---|---|
| Supervisor name: | Brussel, M. van and Huijgen, B.C.H. and Brink, M.S. |
| Faculty: | Medical Sciences |
| Date Deposited: | 04 May 2022 09:37 |
| Last Modified: | 04 May 2022 09:37 |
| URI: | https://umcg.studenttheses.ub.rug.nl/id/eprint/3222 |
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