Table 3 Quality assessment om included studies; quantitative studies, qualitative studies and review studies
Quantitative studies | |
|---|---|
Quality assessment criteria: 1. Is the study design identified and appropriate? 2. How representative are the study group for the population? 3. Is there adequate control group? 4. Is the validity for measurement acceptable? 5. Is the study complete with regard to dropout/missing data and reporting respond rate? 6. Do the authors describe and discuss limitations with the study? 7. To what extent are study results influenced by factors that negatively impact their credibility? 8. Does the study contribute to (new) knowledge about fatigue in HTAAD? | Ratings: Very good, Good, Acceptable, Fair and Poor |
Authors Years | HTAAD diagnosis | 1. Study design | 2. Representative sample | 3. Control groups | 4. Fatigue measure validity | 5. Dropout/ missing data | 6. Discuss limitations | 7. Credibility | 8. Novel knowledge about fatigue |
|---|---|---|---|---|---|---|---|---|---|
Adults | |||||||||
Marfan syndrome (MFS) | |||||||||
[17] Bathen et al. 2014 | MFS: verified Ghent 1 | Good | Good | Good | Good | Good | Good | Good | Very good/good |
[71] Benninghoven et al. 2017 | MFS and one LDS: Verified diagnoses | Good | Acceptable | Good | Good | Good | Very good | Good | Very good/good |
[62] Fusar Poli et al. 2008 | MFS: All verified diagnosis | Good | Good | Acceptable | Good | Acceptable/good | Acceptable | Good | Acceptable |
[52] Ghanta et al. 2015 | MFS: 22 of 49 had verified diagnosis | Good | Acceptable | Good | Acceptable | Good | Good | Acceptable | Acceptable/good |
[63] Moon et al. 2016 | MFS: All verified diagnosis | Very good | Good | Good | Good/very good | Good | Very good/good | Very good | Good/very good |
[72] Percheron et al. 2007 | MFS All verified diagnosis, | Good/acceptable | Acceptable | Good | Acceptable | Good | Good | Good | Good |
[53] Peters et al. 2001 | MFS: only self-reported diagnosis | Good | Acceptable | Good | Acceptable | Good | Good/very good | Good, | Good |
[31] Rand-Hendriksen et al. 2007 | MFS, All verified diagnosis | Good | Acceptable | Good | Good | Good | Acceptable | Good | Very good |
[13] Rand Hendriksen 2010 | MFS: All had verified MFS, | Good | Good | Very good | Good | Good | Acceptable | Good | Acceptable |
[64] Rao et al. 2016 | MFS: All had verified diagnosis | Very good | Very good | Good | Acceptable | Acceptable | Fair | Acceptable | Good |
[54] Ratiue et al. 2018 | MFS: Self-reported diagnosis | Good | Acceptable | Acceptable | Acceptable | Acceptable | Very good- | Acceptable/good | Good/acceptable |
[65] Schoorman et al. 2012 | MFS All had verified diagnosis | Very good | Good | Good | Good | Good | Very good | Very good | Good |
[32] Van Andel et al. 2022 | MFS: All had verified diagnosis | Very good | Good | Good | Very good | Good | Good/very good | Good | Very good |
[55] Van Dijk et al. 2008 | MFS: Verified for some, but not all | Good | Acceptable | Good | Good | Acceptable | Very good | Good | Good |
[73] Vanem et al. 2021 | MFS: All had verified diagnosis | Very Good | Good | Very good | Good | Very good | Very good/good | Very good | Acceptable |
[66] Velvin et al. 2015 | MFS All had verified diagnosis | Good | Good | Good | Good | Acceptable | Fair | Acceptable- | Acceptable |
[67] Velvin et al. 2016 | MFS All had verified diagnosis | Good | Good | Good | Good | Good | Good | Good | Good |
Ehlers Danlos syndrome | |||||||||
[68] Schubart et al. 2019 | HCTP Verified diagnosis, criteria 2015 | Acceptable | Good | Good | Good | Good | Good | Good | Acceptable |
[57] Voermans et al. 2010 | EDS: Several subgroups of EDS, without medically verified diagnoses | Good | Acceptable | Good | Good/acceptable | Good | Good | Good | Good |
Marfan syndrome and Ehlers Danlos syndrome | |||||||||
[56] Verbraecken et al. 2001 | MFS/EDS: Self-reported diagnosis | Good | Acceptable | Good | Good | Acceptable, | Good | Good | Acceptable |
Vascular Ehlers Danlos (vEDS) Syndrome and Loeys Dietz syndrome (LDS) | |||||||||
[11] Johansen et al. 2019 | LDS/vEDS: All verified diagnosis, | Good | Good | Acceptable | Good | Good | Good | Good | Good |
[69] Johansen et al. 2021 | LDS/vEDS: All verified diagnosis, | Good | Good | Good | Good | Good | Good | Good | Good |
[16] Johansen et al. 2022 | LDS/vEDS: All verified diagnosis | Good | Good | Good | Good | Good | Good | Good | Very Good |
Different HTAAD diseases (including LDS, MFS and other HTAADs) | |||||||||
[58] Thijssen et al. 2020 | HTAADs: 86,5% verified diagnoses | Good | Good- | Good | Good | Good | Good | Good | Good |
Children/Adolescents | |||||||||
[70] Warnink-Kavelaars, et al. 2020 | MFS: All children had verified diagnosis, | Good | Good | Good | Good | Acceptable/good | Good | Good | Good |
[61] Warnink-Kavelaars, et al. 2021 | MFS, LDS, EDS and hEDS: Unclear if the children had verified diagnosis | Good | Acceptable | Good | Good | Good | Good | Good- | Very good |
Qualitative studies | |
|---|---|
Quality assessment criteria: 1. Is the research questions(s) clearly and explicitly stated? 2. How was the participants selected (described selection process)? 3. The researchers role and has it been taken in account? 4. Is the method appropriate for collecting data? 5. Is the method appropriate for analyzing the data and for ensuring scientific rigor? 6. The credibility of the study ( as a whole) 7. Do the study contribute to novel knowledge on the particular issue (fatigue)? | Ratings: Very good, Good, Acceptable, Fair and Poor |
Author, year | Diagnosis (es) | 1.Research question (s) | 2.Recruitment | 3.The role of researcher (s) | 4.Appropriate method | 5.Appropriate analysis (es) | 6.Limitations | 7.Credibility | 8.Contribute to new knowledge 8 |
|---|---|---|---|---|---|---|---|---|---|
Adults | |||||||||
[12] Velvin et al. 2021 | MFS, LDS;, vEDS All verified diagnoses | Good | Good | Good | Good | Good | Good | Good | Acceptable |
Children/adolescents | |||||||||
[59] Kelleher et al. 2015 | MFS: Not verified diagnoses, only self-reported | Good | Acceptable | Acceptable | Very Good | Good | Good | Good | Good |
[74] Warnink-Kavelaars, et al. 2019 | MFS: All had confirmed diagnosis | Very good | Good | Acceptable | Very good | Very good | Very good | Very good | Very good |
[60] Warnink-Kavelaars, et al. 2019 | MFS: No description of verified diagnosis | Good | Good | Acceptable | Very good | Good | Very good | Good | Good |
Review articles | |
|---|---|
Quality assessment criteria: 1. Is the review question clearly and explicitly stated? 2. Were the inclusion criteria appropriate for the review question? 3. Was the search strategy appropriate? 4. Were the criteria for appraising studies appropriate? 5. Was critical appraisal conducted by two or more reviewers independently? 6. Were there methods to minimize errors in data extraction? 7. Were the methods used to combine studies appropriate? 8. How is the credibility of the study? (limitations described, transparency, method, analyses and total impression) 9. Contribution to new knowledge on fatigue in HTAADs? (benefits worth the harms and costs), implication for practice and recommendation for further research | Ratings: Very good, Good, Acceptable, Fair and Poor |
Authors | Diagnosis(es) | Research questions1 | Inclusion criteria2 | Search strategy3 | Criteria for appraisal4 | Process of critical appraisal5 | Methods for minimizing error I data extraction6 | Methods for combining studies7 | Credibility8 | Contribution to new knowledge of fatigue9 |
|---|---|---|---|---|---|---|---|---|---|---|
[20] Nielsen et al. 2019 | MFS All types of articles on MFS | Good - | Good | Good | Not relevant | Good | Acceptable - | Good | Good | Good |
[19] Velvin et al. 2014 | MFS All types of articles on MFS | Good | Good | Good | Good | Good | Acceptable | Good | Good | Acceptable |
[51] Velvin et al. 2019 | HTAAD Only articles on MFS were identified | Good | Good | Good | Good | Good | Acceptable | Good | Good | Acceptable |