Audiology and Speech Pathology - Research Publications
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ItemNo Preview AvailableSwallowing function in people with Friedreich's ataxia(WILEY-BLACKWELL, 2013-06)
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ItemNo Preview AvailableTreatment for dysphagia (swallowing difficulties) in hereditary ataxia(WILEY, 2015)BACKGROUND: Hereditary ataxias are a heterogeneous group of disorders resulting in progressive inco-ordination. Swallowing impairment, also known as dysphagia, is a common and potentially life threatening sequel of disease progression. The incidence and nature of dysphagia in these conditions is largely unknown. The loss of an effective and safe swallow can dramatically affect the health and well-being of an individual. Remediation of difficulties of eating and drinking is an important goal in the clinical care of people with hereditary ataxia. OBJECTIVES: To assess the effects of interventions for swallowing impairment (dysphagia) in people with hereditary ataxias. SEARCH METHODS: We searched the Cochrane Neuromuscular Disease Group Specialized Register, the Cochrane Central Register of Controlled Trials (CENTRAL), MEDLINE, EMBASE, CINAHL Plus, PsycINFO, and the Education Resources Information Center (ERIC) on 14 September 2015. We also searched Linguistics and Language Behavior Abstracts (LLBA), Dissertation Abstracts, and Trials Registries on 24 September 2015. SELECTION CRITERIA: We considered all randomised controlled trials (RCTs) and quasi-RCTs that compared treatments for hereditary ataxia with placebo or no treatment. We only included studies measuring dysphagia. DATA COLLECTION AND ANALYSIS: Three review authors (ES, KJ, MK) independently screened all titles and abstracts. In the event of any disagreement or uncertainty over the inclusion of a particular paper, the review authors planned to meet and reach consensus. MAIN RESULTS: We identified no RCTs from the 519 titles and abstracts screened. We excluded papers primarily for not including participants with a hereditary ataxia (that is, being focused on other neurological conditions), being theoretical reviews rather than intervention studies, or being neither randomised nor quasi-randomised trials.We identified five papers of various design that described treatment for dysphagia, or improvement to swallow as a by-product of treatment, in people with hereditary ataxia. None of these studies were RCTs or quasi-RCTs. AUTHORS' CONCLUSIONS: There is an absence of any significant evidence supporting the use of any dysphagia intervention in hereditary ataxia. The lack of evidence highlights the critical need for well-controlled treatment trials in the field.
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ItemDysphagia in Friedreich Ataxia(SPRINGER, 2017-10)The objective of the study was to comprehensively characterise dysphagia in Friedreich ataxia (FRDA) and identify predictors of penetration/aspiration during swallowing. We also investigated the psychosocial impact of dysphagia on individuals with FRDA. Sixty participants with FRDA were screened for dysphagia using a swallowing quality of life questionnaire (Swal-QOL) and case history. Individuals reporting dysphagia underwent a standardised oromotor assessment (Frenchay Dysarthria Assessment, 2, FDA-2) and videofluoroscopic study of swallowing (VFSS). Data were correlated with disease parameters (age at symptom onset, age at assessment, disease duration, FXN intron 1 GAA repeat sizes, and Friedreich Ataxia Rating Scale (FARS) score). Predictors of airway penetration/aspiration were explored using logistic regression analysis. Ninety-eight percent (59/60) of participants reported dysphagia, of whom 35 (58.3%) underwent FDA-2 assessment, and 38 (63.3%) underwent VFSS. Laryngeal, respiratory, and tongue dysfunction was observed on the FDA-2. A Penetration-Aspiration Scale score above 3 (deemed significant airway compromise based on non-clinical groups) was observed on at least one consistency in 13/38 (34.2%) participants. All of those who aspirated (10/38, 26.3%) did so silently, with no overt signs of airway entry such as reflexive cough. Significant correlations were observed between dysphagic symptoms and disease duration and severity. No reliable predictors of penetration or aspiration were identified. Oropharyngeal dysphagia is commonly present in individuals with FRDA and worsens with disease duration and severity. Individuals with FRDA are at risk of aspiration at any stage of the disease and should be reviewed regularly. Instrumental analysis remains the only reliable method to detect aspiration in this population. Dysphagia significantly affects the quality of life of individuals with FRDA.
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ItemA Systematic Review of Self-reported Swallowing Assessments in Progressive Neurological Disorders(SPRINGER, 2015-02)Dysphagia experienced as a consequence of neurodegenerative disease can have severe consequences on a patient's health and well-being. Regular assessment of swallowing function can assist to achieve adequate nutrition and hydration. Here we review subjective swallowing assessments currently available are suitable for use in people with neurodegenerative disease. Measurement properties were reviewed for each tool and coverage of the World Health Organization's International Classification of Functioning, Disability and Health (WHO ICF) was considered. Assessments were identified following a review of the published literature Instruments were reviewed on the basis of reliability and validity, as well as administrative properties, such an interpretability, acceptability, and feasibility. Tools were also evaluated according to the WHO ICF framework. In total, 19 studies were identified for full-text review from 13,315 abstracts. Nine self-reported dysphagia assessment tools suitable for use in progressive neurological disorders were identified. The Swallowing Quality of Life Questionnaire (SWAL-QOL) yields the strongest combination of reliability (including internal consistency and test-retest reliability) and convergent validity while simultaneously covering all WHO ICF domains. Lengthy administration time was identified as a limitation of the SWAL-QOL. The review highlights a relative lack of well-validated self-report questionnaires in dysphagia for people with progressive neurological disease. Additional validation and evaluation of the clinical utility of the tools currently available is required to further promote an informed selection of available assessments.