The Impact of Dysphagia in Myositis: A Systematic Review and Meta-Analysis
Abstract
:1. Introduction
2. Methods
2.1. Review
2.1.1. Inclusion and Exclusion Criteria in the Review
- Cohort: the article had to report on dysphagia in at least one subject with IIM. If the cohort included less than five subjects, it had to be stated that diagnostic criteria of definitive or probable IIM according to either Bohan and Peter [8,9], Griggs [10], Needham and Mastaglia [11], the European Neuromuscular Center [12] or the ACR/EULAR criteria [3] were met. If this was not the case, articles were only included if based on the information provided the current ACR/EULAR criteria [3] for definitive or probable IIM were met or if the diagnosis was confirmed by muscle biopsy.
- Topic: the articles had to report on at least one of the following topics:
- Epidemiology or prevalence of dysphagia in a population with a minimum of five subjects;
- Pathophysiology of dysphagia;
- Outcome of a patient cohort with dysphagia;
- Therapeutic effects on dysphagia or swallowing.
- Patients had other diseases associated with dysphagia, e.g., myasthenia gravis. However, this exclusion criterion was not applied to diseases associated with IIM such as rheumatological diseases in case of overlap syndromes;
- They exclusively reported on gastroesophageal reflux as manifestation of dysphagia;
- Dysphagia was reported exclusively as manifestation of structures distal to the esophagus;
- Conflicting results were reported within the article (e.g., differing prevalence rates).
2.1.2. Search Strategy
2.2. Meta-Analysis
2.2.1. Inclusion and Exclusion Criteria in the Meta-Analysis
2.2.2. Bias Risk in Individual Studies
2.2.3. Statistical Analysis
3. Results
3.1. Review
3.1.1. Epidemiology
3.1.1.1. Dysphagia and Disease Course
3.1.1.2. Factors Associated with Dysphagia
3.1.2. Pathophysiology
Inflammation of Swallowing Muscles
Dysphagia Pathology
3.1.3. Outcome
3.1.4. Therapy
Immunomodulatory Therapy
Therapy of Malignancy
Non-Pharmacological Interventional Therapy
Behavioral Therapy
3.2. Meta-Analysis
4. Discussion
5. Conclusions
Supplementary Materials
Author Contributions
Funding
Acknowledgments
Conflicts of Interest
References
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Patient Group | n, Studies | n, Subjects | Prevalence | CI Lower | CI Upper | I-Squared | p-Value Egger’s Test | Low Bias Risk |
---|---|---|---|---|---|---|---|---|
total cohort | 109 | 10382 | 36% | 33% | 40% | 87% | >0.01 * | 6% |
PM | 21 | 882 | 23% | 18% | 27% | 52% | 0.03 * | 5% |
DM | 49 | 3274 | 31% | 26% | 35% | 80% | >0.01 * | 2% |
IBM | 23 | 1352 | 56% | 47% | 65% | 76% | >0.01 * | 22% |
low bias risk | 6 | 115 | 82% | 65% | 98% | 0% | 0.70 | 100% |
malignancy+ | 13 | 271 | 51% | 43% | 60% | 0% | 0.39 | 0% |
malignancy− | 13 | 1120 | 23% | 17% | 30% | 85% | 0.02 * | 0% |
NXP2+ | 5 | 196 | 56% | 45% | 66% | 0% | 0.42 | 0% |
NXP2− | 5 | 1188 | 33% | 28% | 37% | 42% | 0.22 | 0% |
MDA5+ | 3 | 89 | 12% | 0% | 23% | 61% | 0.13 | 0% |
MDA5− | 3 | 538 | 21% | 10% | 32% | 86% | 0.22 | 0% |
SEA+ | 2 | 17 | 76% | 35% | 100% | 0% | n.a. | 0% |
SEA− | 2 | 589 | 35% | 20% | 49% | 81% | n.a. | 0% |
SRP+ | 3 | 51 | 62% | 40% | 84% | 0% | 0.69 | 0% |
SRP− | 3 | 943 | 36% | 26% | 45% | 81% | 0.15 | 0% |
TIF1y+ | 3 | 103 | 45% | 32% | 58% | 0% | 0.67 | 0% |
TIF1y− | 3 | 519 | 23% | 0% | 48% | 98% | 0.12 | 0% |
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Labeit, B.; Pawlitzki, M.; Ruck, T.; Muhle, P.; Claus, I.; Suntrup-Krueger, S.; Warnecke, T.; Meuth, S.G.; Wiendl, H.; Dziewas, R. The Impact of Dysphagia in Myositis: A Systematic Review and Meta-Analysis. J. Clin. Med. 2020, 9, 2150. https://doi.org/10.3390/jcm9072150
Labeit B, Pawlitzki M, Ruck T, Muhle P, Claus I, Suntrup-Krueger S, Warnecke T, Meuth SG, Wiendl H, Dziewas R. The Impact of Dysphagia in Myositis: A Systematic Review and Meta-Analysis. Journal of Clinical Medicine. 2020; 9(7):2150. https://doi.org/10.3390/jcm9072150
Chicago/Turabian StyleLabeit, Bendix, Marc Pawlitzki, Tobias Ruck, Paul Muhle, Inga Claus, Sonja Suntrup-Krueger, Tobias Warnecke, Sven G. Meuth, Heinz Wiendl, and Rainer Dziewas. 2020. "The Impact of Dysphagia in Myositis: A Systematic Review and Meta-Analysis" Journal of Clinical Medicine 9, no. 7: 2150. https://doi.org/10.3390/jcm9072150