Exercise and sports participation in patients with thoracic aortic disease: a review

Thijssen, Carlijn G E; Bons, Lidia R.; Gökalp, Arjen L.; Kimmenade, Roland R.J. van; Mokhles, Mostafa M; Pelliccia, Antonio; Takkenberg, Johanna J.M.; Roos‐Hesselink, Jolien W.

doi:10.1080/14779072.2019.1585807

Cited by 43 publications

(19 citation statements)

References 64 publications

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“…Importantly, several parents reported confusion over the safety of physical activity for their daughter as it related to her heart, which is supported by Sienkiewicz-Dianzenza et al, who reported physicians excluded 1 in 4 adolescents with TS from participating in school physical education classes (Sienkiewicz-Dianzenza et al, 2006). Although the risk of aortic dissection is higher in TS than the general population, the consensus is that moderate intensity physical activity is safe for the vast majority of girls with TS (Gravholt et al, 2017) and may be beneficial even among individuals with thoracic aortic disease according to a recent systematic review (Thijssen et al, 2019). Clinical implications and recommendations based on key meta-inferences from mixed methods joint analysis are listed in Table 3.…”

Section: Discussionmentioning

confidence: 99%

A mixed methods study of physical activity and quality of life in adolescents with Turner syndrome

Thompson

Zieba

Howell

et al. 2019

American J of Med Genetics Pt A

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Women with Turner syndrome (TS) have a high morbidity from both medical and psychological conditions with a negative impact on quality of life (QoL). Physical activity is a modifiable behavior shown to reduce risk for these chronic medical and mental health conditions and enhance QoL in other populations. Limited research suggests that adolescents and women with TS are less likely to engage in or enjoy physical activity than peers. This mixed methods study aimed to document physical activity levels in a sample of youth with TS and explore how factors unique to TS contribute to and are affected by physical activity. A cross-sectional sample of 21 girls (12-21 years) with TS and their parents (n=21) completed standardized questionnaires to quantify habitual physical activity (3-Day Physical Activity Recall) and QoL (PROMIS) and participated in individual interviews focused on their experience with physical activity. Quantitative and qualitative results were synthesized using a phenomenological mixed methods approach. Results indicate that our sample engaged in less physical activity than peers and only 19% met recommendations for one hour per day of moderate-to-vigorous physical activity (MVPA). Parents reported significant problems with peer relationships and psychological stress, and peer relationships scores correlated with physical activity. Reported barriers to physical activity included physical and psychosocial complications related to TS as well as unique developmental considerations specific to adolescence. Quantitative and qualitative results supported that structured fitness options embedded into routines enhanced activity levels. Results were compiled into specific recommendations for clinical care and areas of future research.

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Section: Discussionmentioning

confidence: 99%

A mixed methods study of physical activity and quality of life in adolescents with Turner syndrome

Thompson

Zieba

Howell

et al. 2019

American J of Med Genetics Pt A

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“…Hence, there still is no evidence of contraindications for an exercise prescription in patients with MFS. Moreover, there is a need to investigate the precise effects of training in such patients has been recently mentioned (10,26). In this context, the preliminary results of our study that is underway are very encouraging.…”

Section: Resultsmentioning

confidence: 58%

“…Moreover, regular, moderate to intense exercises have been reported to significantly lower blood pressure and therefore may have a significant impact on the appearance of cardiovascular diseases and aorthopathy (23)(24)(25). Patients with MFS might also benefit from regular physical activity but, to date, there are no randomized studies evaluating the longitudinal effect of exercise on survival or risk of aortic dissection in patients with aorthopathy including MFS patients (26).…”

Section: Resultsmentioning

confidence: 99%

Effects of a personalized home-based training program among patients suffering from Marfan syndrome: a pilot randomized and controlled study

Jouini

Eliahou

Jondeau

et al. 2021

IRDR

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Marfan, telerehabilitation, personalized trainingMarfan syndrome (MFS) is an autosomal hereditary pathology affecting 1:5000 peoples. Alteration of the fibrillin 1 gene (FBN1) results in haplo-insufficiency of the FBN1 protein mainly altering the vascular system. International recommendations have gradually allowed MFS patients to perform training programs because of its potential benefits. However, to date, there are no data on the effect of a long training period in these patients. The aim of the present study is to investigate the effect of a 3-month personalized home-based training on quality of life (QoL) of patients suffering from MFS. At least 50 MFS patients were included in the study. They were randomly placed into 4 groups: control group; endurance; resistance and endurance + resistance training groups. The training program lasted 3 months and is performed at patients' home. There were 2 training sessions per week telemonitored by a specialist of physical activity and cardiology. Pre and post-training evaluations were performed at the Bichat-Paris Hospital, France. They consisted of assessing psychometrics based on self-administered questionnaires (FiRST, GPAQ, ISP-25, MOS SF-36) and physiological parameters such as the peak oxygen consumption, aorta diameter, cardiac ventricle function and skeletal muscle power at rest and during exercise. Our preliminary results showed an improvement of 50% in QoL, cardiorespiratory fitness and skeletal muscle power in a patient who completed the combined training program. This experimental approach might be a new alternative way for MFS patients' care that may improve their QoL, cardiorespiratory fitness and skeletal muscle power.

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“…It is equally unclear what the impact of voluntarily running might be on cardiovascular disease in MFS. Contrary to the believe that increased blood pressure during voluntarily running would exacerbate TAA progression, it has recently been reported that moderate exercise actually decreased the rate of aneurysm growth in mice with a mild non‐lethal form of MFS (Gibson et al, ; Thijssen et al, ). Unfortunately there are no data in the literature of controlled trials that have longitudinally evaluated how exercise influences TAA progression in MFS patients (Thijssen et al, ).…”

Section: Introductionmentioning

confidence: 90%

The influence of fibrillin‐1 and physical activity upon tendon tissue morphology and mechanical properties in mice

et al. 2019

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Fibrillin‐1 mutations cause pathological changes in connective tissue that constitute the complex phenotype of Marfan syndrome. In this study, we used fibrillin‐1 hypomorphic and haploinsufficient mice (Fbn1mgr/mgR and Fbn1+/− mice, respectively) to investigate the impact of fibrillin‐1 deficiency alone or in combination with regular physical activity on tendon tissue morphology and mechanical properties. Morphological and biomechanical analyses revealed that Fbn1mgr/mgR but not Fbn1+/− mice displayed smaller tendons with physical properties that were unremarkable when normalized to tendon size. Fbn1mgR/mgR mice (n = 43) Fbn1+/−mice (n = 27) and wild‐type mice (WT, n = 25) were randomly assigned to either control cage conditions (n = 54) or to a running on a running wheel for 4 weeks (n = 41). Both fibrillin‐1‐deficient mice ran voluntarily on the running wheel in a manner similar to WT mice (3–4 km/24 h). Regular exercise did not mitigate aneurysm progression in Fbn1mgR/mgR mice (P < 0.05) as evidenced by unmodified median survival. In spite of the smaller size, tendons of fibrillin‐1‐deficient mice subjected to regular exercise showed no evidence of overt histopathological changes or tissue overload. We therefore concluded that lack of optimal fibrillin‐1 synthesis leads to a down regulation of integrated tendon formation, rather than to a loss of tendon quality, which also implies that fibrillin‐1 deficiency in combination with exercise is not a suitable animal model for studying the development of tendon overuse (tendinopathy).

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Exercise and sports participation in patients with thoracic aortic disease: a review

Cited by 43 publications

References 64 publications

A mixed methods study of physical activity and quality of life in adolescents with Turner syndrome

A mixed methods study of physical activity and quality of life in adolescents with Turner syndrome

Effects of a personalized home-based training program among patients suffering from Marfan syndrome: a pilot randomized and controlled study

The influence of fibrillin‐1 and physical activity upon tendon tissue morphology and mechanical properties in mice

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