Oropharyngeal dysphagia as dominant and life-threatening symptom in dermatomyositis

Djakovic, Zorana; Vesić, Sonja; Tomovic, Maja; Vuković, Jelena

doi:10.2298/vsp0908671d

Cited by 2 publications

(4 citation statements)

References 11 publications

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“…In contrast, a less strict criterion of refractoriness was observed in previous studies and data on severe symptoms such as dysphagia were not reported hampering comparison with the present [4][5][6][7][8][9][10][11]. In fact, among patients evaluated in the present study, more than two-thirds had dysphagia, a known serious problem in patients with IIM that can be associated with nutritional deficiency, aspiration pneumonia and poor prognosis [18]. The rituximab protocol was a more aggressive approach than others previously reported [4][5][6][7][8][9][10][11], and included pre-infusion of methylprednisolone and intravenous human immunoglobulin for the majority of the patients.…”

Section: Discussioncontrasting

confidence: 73%

Favorable rituximab response in patients with refractory idiopathic inflammatory myopathies

et al. 2018

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Background: Interpretation of rituximab efficacy for refractory idiopathic inflammatory myopathies (IIM) is hampered by the absence of a uniform definition of refractory myositis and clinical response. Therefore, rigorous criteria of refractoriness, together with a homogenous definition of clinical improvement, were used to evaluate rituximab one-year response.Methods: A retrospective cohort study including 43 IIM (15 antisynthetase syndrome, 16 dermatomyositis, 12 polymyositis) was conducted. All patients had refractory disease (inadequate response to at least two immunosuppressives/immunomodulatories and no less than three months sequentially or concomitantly glucocorticoid tapering) criteria. Clinical/laboratory improvement at one-year was based on modified International Myositis Assessment & Clinical Studies Group (IMACS) core set measures. The patients received two infusions of rituximab (1 g each) at baseline, followed by repeated dose after 6 months. Baseline immunosuppressive therapy was maintained and glucocorticoid dose was tapered according to clinical/laboratory parameters. Results: Five patients had side effects at the first rituximab application and were excluded. Therefore, 38 out of 43 patients completed the one-year follow up. Almost 75% of the patients attained clinical and laboratory response after one-year. A significant reduction in median glucocorticoid dose (18.8 vs. 6.3 mg/day) was achieved and 42% patients were able to discontinue prednisone. In contrast, young individuals and patients with dysphagia had a tendency to be non-responders to rituximab. No severe infections were observed. Conclusion: This study provides convincing evidence that rituximab is an effective and safe therapy for refractory IIM.

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Section: Discussioncontrasting

confidence: 73%

Favorable rituximab response in patients with refractory idiopathic inflammatory myopathies

et al. 2018

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“…IIM can result in impairment of the oral [ 29 , 58 , 59 , 60 , 61 , 62 , 63 , 64 ], pharyngeal [ 7 , 23 , 24 , 27 , 29 , 31 , 32 , 33 , 44 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 65 , 66 , 67 , 68 , 69 , 70 , 71 , 72 , 73 , 74 , 75 , 76 , 77 , 78 , 79 , 80 , 81 , 82 , 83 , 84 , 85 , 86 , 87 , 88 , 89 , 90 , 91 , 92 , 93 , 94 , 95 ,…”

Section: Resultsunclassified

“…Reduced pharyngeal contractility can result in insufficient pharyngeal bolus clearance [ 139 ]. Consequently, myositis patients often show pharyngeal residue after swallowing [ 7 , 27 , 29 , 31 , 33 , 44 , 58 , 59 , 60 , 61 , 65 , 66 , 67 , 69 , 71 , 73 , 74 , 75 , 76 , 77 , 78 , 79 , 80 , 83 , 84 , 86 , 87 , 91 , 93 , 96 , 100 , 101 , 103 , 104 , 106 , 111 , 112 , 115 ]. These can impact on swallowing safety and ultimately cause aspiration, which is a frequently reported finding [ 33 , 78 , 81 , 86 ].…”

Section: Resultsmentioning

confidence: 99%

“…These can impact on swallowing safety and ultimately cause aspiration, which is a frequently reported finding [ 33 , 78 , 81 , 86 ]. Further findings indicating reduced pharyngeal contractility are absent or inadequate peristalsis or bolus propulsion [ 27 , 31 , 69 , 95 , 106 , 111 ], inadequate pharyngeal contraction [ 29 , 61 , 75 , 87 , 100 , 116 , 126 ], nasal regurgitation due to velopharyngeal insufficiency [ 31 , 44 , 62 , 74 , 79 , 80 , 97 , 98 , 104 , 109 , 110 , 111 , 116 ], piecemeal deglutition when swallowing larger boluses [ 82 , 140 ] or reduced pharyngeal pressure in manometry [ 7 , 29 , 32 ].…”

Section: Resultsmentioning

confidence: 99%

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The Impact of Dysphagia in Myositis: A Systematic Review and Meta-Analysis

Labeit

Pawlitzki

Ruck

et al. 2020

JCM

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(1) Background: Dysphagia is a clinical hallmark and part of the current American College of Rheumatology/European League Against Rheumatism (ACR/EULAR) diagnostic criteria for idiopathic inflammatory myopathy (IIM). However, the data on dysphagia in IIM are heterogenous and partly conflicting. The aim of this study was to conduct a systematic review on epidemiology, pathophysiology, outcome and therapy and a meta-analysis on the prevalence of dysphagia in IIM. (2) Methods: Medline was systematically searched for all relevant articles. A random effect model was chosen to estimate the pooled prevalence of dysphagia in the overall cohort of patients with IIM and in different subgroups. (3) Results: 234 studies were included in the review and 116 (10,382 subjects) in the meta-analysis. Dysphagia can occur as initial or sole symptom. The overall pooled prevalence estimate in IIM was 36% and with 56% particularly high in inclusion body myositis. The prevalence estimate was significantly higher in patients with cancer-associated myositis and with NXP2 autoantibodies. Dysphagia is caused by inflammatory involvement of the swallowing muscles, which can lead to reduced pharyngeal contractility, cricopharyngeal dysfunction, reduced laryngeal elevation and hypomotility of the esophagus. Swallowing disorders not only impair the quality of life but can lead to serious complications such as aspiration pneumonia, thus increasing mortality. Beneficial treatment approaches reported include immunomodulatory therapy, the treatment of associated malignant diseases or interventional procedures targeting the cricopharyngeal muscle such as myotomy, dilatation or botulinum toxin injections. (4) Conclusion: Dysphagia should be included as a therapeutic target, especially in the outlined high-risk groups.

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Oropharyngeal dysphagia as dominant and life-threatening symptom in dermatomyositis

Cited by 2 publications

References 11 publications

Favorable rituximab response in patients with refractory idiopathic inflammatory myopathies

Favorable rituximab response in patients with refractory idiopathic inflammatory myopathies

The Impact of Dysphagia in Myositis: A Systematic Review and Meta-Analysis

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