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      Dysphagia in Patients with Sporadic Inclusion Body Myositis: Management Challenges

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          Abstract

          Dysphagia in inclusion body myositis (IBM) is common and associated with increased mortality and morbidity due to aspiration pneumonia, malnutrition and dehydration. There is currently no consensus on treatment of dysphagia in IBM and outcomes are variable depending on timing of intervention, patient preference and available expertise. There is a paucity of research exploring the pathophysiology of dysphagia in IBM and appropriate investigations. Increased knowledge of the aetiopathogenesis is likely to change the approach to treatment as well as improve the quality of life for patients. This review explores the epidemiology and pathophysiology of dysphagia in IBM and the currently available treatment strategies.

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          Most cited references71

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          Inclusion body myositis: clinical features and pathogenesis

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            Prevalence of sporadic inclusion body myositis and factors contributing to delayed diagnosis.

            The prevalence of sporadic inclusion body myositis (sIBM) is variable in different populations and ethnic groups. A previous survey in Western Australia in 2000 found a prevalence of 9.3 per million population. We have now performed a follow-up survey to determine whether there has since been any change in prevalence. The current prevalence was found to be 14.9 per million population, with a prevalence of 51.3 per million population in people over 50 years of age. This is the highest reported prevalence of sIBM and correlates with a high frequency of HLA-DR3 and the 8.1 major histocompatibility complex ancestral haplotype in this population. Review of a combined cohort of 57 sIBM cases from three Australian centres revealed a high rate of initial misdiagnosis and a mean time to diagnosis of 5.2 years, which suggests that even the latest prevalence figure may be an underestimate, and emphasising the need to increase the level of awareness of the condition among the medical community.
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              Impact of oropharyngeal dysphagia on healthcare cost and length of stay in hospital: a systematic review

              Background Healthcare systems internationally are under an ever-increasing demand for services that must be delivered in an efficient, effective and affordable manner. Several patient-related and organisational factors influence health-care expenditure and utilisation, including oropharyngeal dysphagia. Here, we present a systematic review of the literature and meta-analyses investigating how oropharyngeal dysphagia influences healthcare utilisation through length of stay (LOS) and cost. Methods Using a standardised approach, eight databases were systematically searched for relevant articles reporting on oropharyngeal dysphagia attributable inpatient LOS and healthcare costs through June 2016. Study methodologies were critically appraised and where appropriate, extracted LOS data were analysed in an overall summary statistic. Results Eleven studies reported on cost data, and 23 studies were included reporting on LOS data. Descriptively, the presence of dysphagia added 40.36% to health care costs across studies. Meta-analysis of all-cause admission data from 13 cohort studies revealed an increased LOS of 2.99 days (95% CI, 2.7, 3.3). A subgroup analysis revealed that admission for stroke resulted in higher and more variable LOS of 4.73 days (95% CI, 2.7, 7.2). Presence of dysphagia across all causes was also statistically significantly different regardless of geographical location: Europe (8.42 days; 95% CI, 4.3; 12.5), North America (3.91 days; 95% CI, 3.3, 4.5). No studies included in meta-analysis were conducted in Asia. Conclusions This systematic review demonstrated that the presence of oropharyngeal dysphagia significantly increases healthcare utilisation and cost, highlighting the need to recognise oropharyngeal dysphagia as an important contributor to pressure on healthcare systems. Electronic supplementary material The online version of this article (10.1186/s12913-018-3376-3) contains supplementary material, which is available to authorized users.
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                Author and article information

                Journal
                Int J Gen Med
                Int J Gen Med
                IJGM
                ijgm
                International Journal of General Medicine
                Dove
                1178-7074
                05 December 2019
                2019
                : 12
                : 465-474
                Affiliations
                [1 ]School of Medicine, The University of Notre Dame , Fremantle, Western Australia, Australia
                [2 ]Department of Speech Pathology, Royal Perth Hospital , Perth, Western Australia, Australia
                [3 ]Department of Neurology, Fiona Stanley Hospital , Murdoch, Western Australia, Australia
                [4 ]Institute for Immunology and Infectious Diseases, Murdoch University , Murdoch, Western Australia, Australia
                Author notes
                Correspondence: Merrilee Needham Institute for Immunology and Infectious Diseases, Murdoch University , Building 390 Discovery Way, Murdoch, WA6150, AustraliaTel +61 8 9360 1334Fax +61 8 9360 1380 Email Merrilee.Needham@health.wa.gov.au
                Author information
                http://orcid.org/0000-0001-5641-5852
                http://orcid.org/0000-0003-0863-3956
                http://orcid.org/0000-0002-1077-5569
                Article
                198031
                10.2147/IJGM.S198031
                6901064
                31824189
                cc20c78a-960a-429b-ba77-6536fd36ef58
                © 2019 Mohannak et al.

                This work is published and licensed by Dove Medical Press Limited. The full terms of this license are available at https://www.dovepress.com/terms.php and incorporate the Creative Commons Attribution – Non Commercial (unported, v3.0) License ( http://creativecommons.org/licenses/by-nc/3.0/). By accessing the work you hereby accept the Terms. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed. For permission for commercial use of this work, please see paragraphs 4.2 and 5 of our Terms ( https://www.dovepress.com/terms.php).

                History
                : 11 September 2019
                : 21 November 2019
                Page count
                Figures: 2, References: 76, Pages: 10
                Categories
                Review

                Medicine
                inflammatory myopathies,swallowing,epidemiology,pathophysiology,treatment,diagnosis
                Medicine
                inflammatory myopathies, swallowing, epidemiology, pathophysiology, treatment, diagnosis

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