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Systematic Review of the Methodological Quality and Outcome Measures Utilized in Exercise Interventions for Individuals with Cystic Fibrosis

Najla Z. Alotaibi¹, Hon K. Yeung², Norran T. Almakhlafi³, Samantha C. Crisafulli⁴, Stefanie Krick⁵, John D. Lowman⁶

1. Program in Rehabilitation Science, University of Alberta, Edmonton, Alberta, Canada
2. Department of Physical Therapy, University of Alberta, Edmonton, Alberta, Canada
3. Department of Physical Therapy, University of Alberta, Edmonton, Alberta, Canada
4. Department of Occupational Therapy, University of Alberta, Edmonton, Alberta, Canada
5. Department of Physical Therapy, University of Alberta, Edmonton, Alberta, Canada
6. Faculty of Rehabilitation Medicine, University of Alberta, Edmonton, Alberta, Canada

ABSTRACT

Background: The aim of this study was to evaluate the methodological quality of interventional exercise studies for individuals with cystic fibrosis (CF) and to assess the outcome measures utilized in these studies to enhance the understanding of the effectiveness of exercise interventions.

Methods: A systematic review was conducted using databases of PubMed, Scopus, CINAHL, SPORTDiscus, and Cochrane review for studies that included exercise interventions for individuals with CF. The methodological quality was assessed using the Effective Public Health Practice Project (EPHPP) quality assessment tool.

Results: We found 4,769 articles in our initial search (Figure 1). After applying the inclusion and exclusion criteria, 89 articles were included in the final analysis.

Conclusion: The review highlights the need for standardized reporting and the use of validated outcome measures in exercise interventions for individuals with CF.

Keywords: Cystic fibrosis, exercise, ICF, quality assessment, systematic review

1. INTRODUCTION

Cystic fibrosis is a genetic disorder that affects almost 400,000 individuals worldwide. It impacts the pulmonary, digestive, and reproductive systems¹. People with CF often experience bronchopneumonia with recurrent exacerbations that can lead to respiratory failure. The common reported symptoms that individuals with cystic fibrosis complain of are shortness of breath, thickened mucus, poor growth, and cough². Existing literature also states that people with CF experience skeletal muscle dysfunction which results in exercise intolerance³.

Exercise training has been proposed to be beneficial for individuals with cystic fibrosis⁴. Based on the literature, exercise training can improve physical function, quality of life in people with CF, and may even reduce the severity of symptoms⁵.

2. METHODS

2.1. STUDY DESIGN

This systematic review followed the PRISMA guidelines for reporting systematic reviews and meta-analyses⁶.

2.2. ELIGIBILITY CRITERIA

Studies were included if they met the following criteria: (1) interventional studies, (2) non-English texts, abstracts only, and/or in population who have had a lung transplant or were diagnosed with a different pulmonary disease such as non-CF bronchiectasis.

2.3. DATA EXTRACTION

Two independent reviewers extracted data from the included studies (NZA and JDL). Two independent reviewers extracted information related to the outcomes measures used before and after the intervention (NZA and JDL).

2.4. METHODOLOGICAL QUALITY AND LEVEL OF EVIDENCE

The Effective Public Healthcare Practice Project “Quality” Assessment Tool for Quantitative Studies (EPHPP) was established and developed by a research team in the school of nursing at McMaster University. This tool is utilized in intervention studies to assess the methodological quality of the studies. Each section has a rating from 1 to 3 with 1 being the lowest and 3 being the highest level of evidence.

2.5. STATISTICAL ANALYSIS

To illustrate the results of this review, frequency distributions, and median analysis as well as simple percentage analysis were performed using Microsoft Excel and also used Kappa coefficient.

3. RESULTS

We found 4,769 articles in our initial search (Figure 1). After applying the inclusion and exclusion criteria, 89 articles were included in the final analysis.

4. DISCUSSION

This review was the first to use the ICF model in relation to exercise interventions for individuals with CF. In general, the improvements across all the studies’ outcomes over the declines, mostly in exercise capacity, quality of life, muscle strength, pulmonary function, body composition, and relief of symptoms.

5. CONCLUSION

Based on the findings of this systematic review, it is suggested that future investigators should ensure that their intervention can be replicated, especially in their methodological therapies, in which more people with CF should be able to exercise.

6. CONFLICT OF INTEREST

The authors have no conflicts of interest to declare.

7. ACKNOWLEDGEMENTS

John D. Lowman is supported by the Cystic Fibrosis Foundation’s Award for Physical Therapy (CCPT520-22) which indirectly led to the participation of this research.

8. REFERENCES

1. Foundation CF. About Cystic Fibrosis. 2024.
2. Balfour-Lynn IM, Welch K, Smith S. Inhaled cycled antibiotics in cystic fibrosis. Cochrane Database of Systematic Reviews 2019;(7): doi: 10.1002/14651858.CD001915.pub3.

3. Coelho CC, Aquino EDS, Diniz ALR, et al. Tissue oxygenation in peripheral muscles and pulmonary function in cystic fibrosis. Pediatric Exercise Science 2020;32(5):1571-1578. doi:10.1123/pes.2018-0874.

4. Stein P, Tach J, Jung S, et al. Skeletal Muscle Function in Young Patients with Cystic Fibrosis. Pediatric Exercise Science 2022;34(1):1-12.

5. Goldbart AD, Cohen AD, Weitzman D, Tal A. Effects of rehabilitation winter camps at the Dead Sea on European cystic fibrosis patients. Isr Med Assoc J 2007;9(11):806-9.

6. Braggion C, Comacchio M, Miano A, Schena F, Vetrato G, Mastella G. Exercise tolerance in young patients with cystic fibrosis and mild airway obstruction. Pediatr Pulmonol. 1989;7(3):145-52. doi:10.1002/ppul.1989.7.3.145.

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