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Dysphagia as Isolated Manifestation of Jo-1 Associated Myositis?

Published on Jul 9, 2019in Frontiers in Neurology2.635
· DOI :10.3389/fneur.2019.00739
Bendix Labeit1
Estimated H-index: 1
,
Paul Muhle7
Estimated H-index: 7
+ 4 AuthorsTobias Warnecke24
Estimated H-index: 24
Abstract
Dysphagia can be predominant or sole symptom of myositis. However, diagnostic evaluation is difficult in such cases. Here, we present evidence for dysphagia as sole manifestation of Jo-1 associated myositis. A 77-year-old patient suffering from isolated dysphagia was assessed by flexible endoscopic evaluation of swallowing, videofluoroscopy, high resolution oesophageal manometry, whole body muscle MRI, electroneurographic and electromyographic examination, cerebrospinal fluid analysis, screening for autoantibodies, and body plethysmography. We detected isolated oropharyngeal dysphagia including a decreased pressure of the upper oesophageal sphincter leading to cachexia in an anti-Jo-1 positive patient without any abnormalities in the other diagnostics. Immunosuppressive therapy with cortisone and azathioprine led to long-term improvement of dysphagia. This is the first report of isolated dysphagia as manifestation of Jo-1 associated myositis. Therefore, Jo-1 associated myositis should be considered as a possible differential diagnosis for isolated dysphagia. Typical signs for myositis in instrumental dysphagia assessment are presented.
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References22
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#2Benjamin Granger (UPMC: Pierre-and-Marie-Curie University)H-Index: 7
Last. Olivier Benveniste (French Institute of Health and Medical Research)H-Index: 36
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Importance Idiopathic inflammatory myopathies are heterogeneous in their pathophysiologic features and prognosis. The emergence of myositis-specific autoantibodies suggests that subgroups of patients exist. Objective To develop a new classification scheme for idiopathic inflammatory myopathies based on phenotypic, biological, and immunologic criteria. Design, Setting, and Participants An observational, retrospective cohort study was performed using a database of the French myositis network. Pati...
30 CitationsSource
#1Sun Im (Catholic University of Korea)H-Index: 10
#2Sonja Suntrup-Krueger (WWU: University of Münster)H-Index: 7
Last. Tobias Warnecke (WWU: University of Münster)H-Index: 24
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2 CitationsSource
#1Ingrid E. Lundberg (Karolinska University Hospital)H-Index: 55
#2Anna Tjärnlund (Karolinska University Hospital)H-Index: 10
Last. Lisa G. Rider (HHS: United States Department of Health and Human Services)H-Index: 53
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Objective To develop and validate new classification criteria for adult and juvenile idiopathic inflammatory myopathies (IIM) and their major subgroups. Methods Candidate variables were assembled from published criteria and expert opinion using consensus methodology. Data were collected from 47 rheumatology, dermatology, neurology and paediatric clinics worldwide. Several statistical methods were used to derive the classification criteria. Results Based on data from 976 IIM patients (74% adults;...
99 CitationsSource
#1Maria Casal-Dominguez (Johns Hopkins University)H-Index: 4
#2Iago Pinal-Fernandez (Hebron University)H-Index: 14
Last. Albert Selva-O’Callaghan (Hebron University)H-Index: 26
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#1Paul D. Neubauer (Yale University)H-Index: 4
#2Alfred W Rademaker (NU: Northwestern University)H-Index: 91
Last. Steven B. Leder (Yale University)H-Index: 32
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The Yale Pharyngeal Residue Severity Rating Scale was developed, standardized, and validated to provide reliable, anatomically defined, and image-based assessment of post-swallow pharyngeal residue severity as observed during fiberoptic endoscopic evaluation of swallowing (FEES). It is a five-point ordinal rating scale based on residue location (vallecula and pyriform sinus) and amount (none, trace, mild, moderate, and severe). Two expert judges reviewed a total of 261 FEES evaluations and selec...
32 CitationsSource
#1C. Hey (Goethe University Frankfurt)H-Index: 9
#2P Pluschinski (Goethe University Frankfurt)H-Index: 6
Last. Timo Stoever (Goethe University Frankfurt)H-Index: 18
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Hintergrund Die Penetrations-Aspirations-Skala wurde von Rosenbek et al. zur standardisierten Dokumentation des Kardinalmerkmals einer Schluckstorung entwickelt.
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#1P. Claire Langdon (SCGH: Sir Charles Gairdner Hospital)H-Index: 1
#2Kylie Patricia Mulcahy (SCGH: Sir Charles Gairdner Hospital)H-Index: 2
Last. Frank L. Mastaglia (UWA: University of Western Australia)H-Index: 62
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Dysphagia has previously been reported in the inflammatory myopathies (IMs): inclusion body myositis (IBM), dermatomyositis (DM), and polymyositis (PM). Patients report coughing, choking, and bolus sticking in the pharynx. Myotomy has been the treatment of choice, with variable success reported. We sought to determine underlying causes of dysphagia in IM patients using instrumental evaluation. Eighteen subjects participated in the study: four with DM, six with PM, and eight with IBM. They underw...
23 CitationsSource
#1Ken-ya Murata (Wakayama Medical University)H-Index: 6
#2Ken Kouda (Wakayama Medical University)H-Index: 8
Last. Tomoyoshi Kondo (Wakayama Medical University)H-Index: 19
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The nature of the swallowing impairment in patients with sporadic inclusion body myositis (s-IBM) has not been well characterized. In this study, we examined ten consecutive s-IBM patients using videofluoroscopy (VF) and computed pharyngoesophageal manometry (CPM). The patients were divided into two groups: patients with complaint and without complaint of dysphagia. VF results indicated pharyngeal muscle propulsion (PP) at the hypopharyngeal and upper esophagus sphincter (UES) in all s-IBM patie...
18 CitationsSource
#1Kylie Patricia Mulcahy (SCGH: Sir Charles Gairdner Hospital)H-Index: 2
#2Patricia Claire Langdon (SCGH: Sir Charles Gairdner Hospital)H-Index: 1
Last. Frank L. MastagliaH-Index: 62
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This study compared self-reported incidence and prevalence of dysphagia in inflammatory myopathy diseases with instrumental data from videofluoroscopy in a cohort of 18 patients with inflammatory myopathies (inclusion body myositis, polymyositis, and dermatomyositis). We found a high self-report of incidence of dysphagia and demonstrated that symptoms described by the patients follow a characteristic pattern. We conclude that there is a high incidence of dysphagia in all three of the inflammator...
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Aliment Pharmacol Ther 31, 359–365 Summary Background The inflammatory myopathies are a group of acquired diseases characterized by a proximal myopathy caused by an inflammatory infiltrate of the skeletal muscle. The three major diseases are dermatomyositis, polymyositis and inclusion body myositis. Aims To review the gastrointestinal manifestations of myositis. Methods Over 110 articles in the English literature were reviewed. Results Dysphagia to solids and liquids occurs in patients with myos...
40 CitationsSource
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