The aim of this thesis is to explore fear of choking and fear of falling in people with Huntington's disease (HD) and their caregivers. Dysphagia and falls are common in HD and may lead to fear of... Show moreThe aim of this thesis is to explore fear of choking and fear of falling in people with Huntington's disease (HD) and their caregivers. Dysphagia and falls are common in HD and may lead to fear of choking and fear of falling. However, knowledge about this is mostly lacking, as well as knowledge about the relationship between cognitive and emotional factors and these types of fear. The study confirms that fall prevalence (29% over a 30-day period) and prevalence of dysphagia symptoms (90%) are high. However, a prevalence of about 50% is also reported for fear of choking and fear of falling in people with HD. However, their informal caregivers report the most fear. Severity of dysphagia symptoms was a predictor of fear of choking, and anticipatory awareness of fall risks and gender were found to be predictors of fear of falling. In most individuals with HD, a combination of preventive measures was used.Future research can improve both management of dysphagia and fear of choking and management of falls and fear of falling. Because questioning people with HD is sometimes difficult, due to speech and cognitive difficulties, diagnostic tools may be sought to facilitate this, such as wearable electronic devices. Show less
PurposeOropharyngeal dysphagia is a common swallowing impairment post-stroke managed by speech language pathologists (SLP). This article aims to demonstrate a local know-do gap assessment for usual... Show morePurposeOropharyngeal dysphagia is a common swallowing impairment post-stroke managed by speech language pathologists (SLP). This article aims to demonstrate a local know-do gap assessment for usual dysphagia care for patients undergoing inpatient stroke rehabilitation in primary healthcare in Norway, which included an assessment of the functional level of the patients and characteristics and outcomes of treatment.Materials and methodsIn this observational study, we assessed the outcomes and interventions of patients admitted to inpatient rehabilitation following stroke. The patients received usual care from SLPs while the research team administered a dysphagia assessment protocol that included assessment of several swallowing domains including oral intake, swallowing, patient self-reported functional health status and health-related quality of life, and oral health. The treating SLPs documented the treatments provided in a treatment diary.ResultsOf 91 patients who consented, 27 were referred for SLP and 14 received treatment. During the median treatment period of 31.5 days (IQR = 8.8-57.0), patients received 7.0 treatment sessions (IQR = 3.8-13.5) of 60 minutes (IQR = 55-60). The patients who received SLP treatment demonstrated no/minor disorders (n = 7) and moderate/severe disorders (n = 7). Dysphagia treatments primarily included oromotor training and advice on bolus modification and were provided without association to dysphagia severity. Patients with moderate/severe swallowing impairments received slightly more SLP sessions over a longer time.ConclusionsThis study identified gaps between current and best practices and opportunities to improve assessment, decision-making, and implement evidence-based practices. Show less
Background and purpose: A matched comparison of external beam radiotherapy (EBRT) versus brachytherapy recently demonstrated that EBRT appears at least as effective for palliating dysphagia in... Show moreBackground and purpose: A matched comparison of external beam radiotherapy (EBRT) versus brachytherapy recently demonstrated that EBRT appears at least as effective for palliating dysphagia in patients with incurable esophageal cancer. The aim of this analysis was to compare patient-reported outcomes (PROs) after EBRT versus brachytherapy.Materials and methods: In a multicenter prospective cohort study, patients with incurable esophageal cancer requiring palliation of dysphagia were included to undergo EBRT (20 Gy in 5 fractions). This EBRT cohort was compared to the single-dose 12 Gy brachytherapy cohort of the previously reported SIRECtrial. Propensity score matching was applied to adjust for baseline imbalances. The primary endpoint of dysphagia improvement was reported previously. PROs were secondary outcomes and assessed at baseline and 3 months after treatment using EORTC QLQ-C30 and QLQ-OES18 questionnaires.Results: A total of 115 enrolled EBRT patients and 93 brachytherapy patients were eligible. After matching, 69 well-balanced pairs remained. At follow-up, significant deteriorations in functioning (i.e. physical, role, social), pain, appetite loss, and trouble with taste were observed after brachytherapy. In the EBRT group, such deterioration was observed only for role functioning, while significant improvements in trouble with eating and pain were found. Between-group comparison showed mostly comparable PRO changes, but significantly favored EBRT with regard to nausea, vomiting, pain, and appetite loss.Conclusion: Short course EBRT results in similar or better PROs at 3 months after treatment compared to single-dose brachytherapy for the palliation of malignant dysphagia. These findings further support its use and inclusion in clinical practice guidelines. (C) 2020 The Author(s). Published by Elsevier B.V. Show less
Baijens, L.W.J.; Walshe, M.; Aaltonen, L.M.; Arens, C.; Cordier, R.; Cras, P.; ... ; Clave, P. 2020
Purpose To develop a European White Paper document on oropharyngeal dysphagia (OD) in head and neck cancer (HNC). There are wide variations in the management of OD associated with HNC across Europe... Show morePurpose To develop a European White Paper document on oropharyngeal dysphagia (OD) in head and neck cancer (HNC). There are wide variations in the management of OD associated with HNC across Europe. Methods Experts in the management of specific aspects of OD in HNC across Europe were delegated by their professional medical and multidisciplinary societies to contribute to this document. Evidence is based on systematic reviews, consensus-based position statements, and expert opinion. Results Twenty-four sections on HNC-specific OD topics. Conclusion This European White Paper summarizes current best practice on management of OD in HNC, providing recommendations to support patients and health professionals. The body of literature and its level of evidence on diagnostics and treatment for OD in HNC remain poor. This is in the context of an expected increase in the prevalence of OD due to HNC in the near future. Contributing factors to increased prevalence include aging of our European population (including HNC patients) and an increase in human papillomavirus (HPV) related cancer, despite the introduction of HPV vaccination in various countries. We recommend timely implementation of OD screening in HNC patients while emphasizing the need for robust scientific research on the treatment of OD in HNC. Meanwhile, its management remains a challenge for European professional associations and policymakers. Show less
Introduction: Short-course external beam radiotherapy (EBRT) and intraluminal brachytherapy are both accepted treatments for the palliation of dysphagia in patients with incurable esophageal cancer... Show moreIntroduction: Short-course external beam radiotherapy (EBRT) and intraluminal brachytherapy are both accepted treatments for the palliation of dysphagia in patients with incurable esophageal cancer. We compared the effects of both treatments from two prospective studies.Methods: We performed a multicenter prospective cohort study of patients with metastasized or otherwise incurable esophageal cancer requiring palliation of dysphagia from September 2016 to March 2019. Patients were treated with EBRT in five fractions of 4 Gy. Data were compared with all patients treated with a single brachytherapy dose of 12 Gy in the SIREC (Stent or Intraluminal Radiotherapy for inoperable Esophageal Cancer) trial, both between the original cohorts and between 1:1 propensity score-matched cohorts. The primary end point was an improvement of dysphagia at 3 months without reintervention. The secondary end points included toxicity and time-to-effect.Results: A total of 115 patients treated with EBRT and 93 patients who underwent brachytherapy were eligible for analysis. In the original cohorts, dysphagia improved after EBRT in 79% of patients compared with 64% after brachytherapy (p = 0.058). Propensity score matching resulted in 69 patients in each cohort well-balanced at baseline. Improvement of dysphagia was observed in 83% after EBRT versus 64% after brachytherapy (p = 0.048). In responding patients, improvement of dysphagia at 2 weeks was observed in 67% after EBRT compared with 35% after brachytherapy, and the maximum effect was reached after 4 weeks in 55% and 33%, respectively. Severe toxicity occurred in 3% of patients after EBRT compared with 13% after brachytherapy.Conclusions: Short-course EBRT appears at least as effective as brachytherapy in the palliation of dysphagia in patients with esophageal cancer. (C) 2020 International Association for the Study of Lung Cancer. Published by Elsevier Inc. Show less
Fibreoptic Endoscopic Evaluation of Swallowing (FEES) and Videofluoroscopic Swallow Studies (VFSS) are instrumental assessments of dysphagia which provide videos of the internal structures of... Show moreFibreoptic Endoscopic Evaluation of Swallowing (FEES) and Videofluoroscopic Swallow Studies (VFSS) are instrumental assessments of dysphagia which provide videos of the internal structures of swallowing. They are commonly regarded as gold-standard' assessments; however, there is no consensus regarding a gold-standard measure to analyse the video recordings that they produce. Measures require sound psychometric properties to be suitable for clinical or research purposes. To date, no review of psychometric properties of FEES and VFSS measures has been undertaken or formally reported. This review assessed the quality of the psychometric properties of visuoperceptual measures of FEES and VFSS. Electronic databases were searched for studies reporting on psychometric qualities of visuoperceptual measures which are used to analyse recordings from FEES and VFSS. All dates until February 2017 were included. The Consensus-based Standards for the selection of health Measurement Instruments (COSMIN) checklist was used to evaluate the methodical quality of studies. The measures' overall quality was then assessed by combining COSMIN ratings with quality criteria. Forty-five studies, reporting on 39 measures, met the inclusion criteria for this review. Data about the measures' psychometric properties were very limited. Twenty-one measures had information available about reliability only, while 18 had information on up to five of the possible nine psychometric properties categorised within the COSMIN framework. The majority of the FEES and VFSS measures' psychometric properties were rated as indeterminate' overall, due to the small number of studies, issues with design, statistical analyses, and reporting practices of extant studies. There is insufficient evidence to recommend any individual measure included in this review as valid and reliable to interpret VFSS and FEES recordings. Further research, which utilises robust methodological design and reporting, is needed to examine the psychometric properties of measures for FEES and VFSS. Show less
The scope of this thesis spanned several issues in the measurement and evaluation of OD. The screening, assessment, and treatment effect for OD have been covered,with a special emphasis on... Show moreThe scope of this thesis spanned several issues in the measurement and evaluation of OD. The screening, assessment, and treatment effect for OD have been covered,with a special emphasis on patient self-evaluation. Show less
The Swallowing Quality of Life questionnaire (SWAL-QOL) is widely used clinically and in research to evaluate quality of life related to swallowing difficulties. It has been described as a valid... Show moreThe Swallowing Quality of Life questionnaire (SWAL-QOL) is widely used clinically and in research to evaluate quality of life related to swallowing difficulties. It has been described as a valid and reliable tool, but was developed and tested using classic test theory. This study describes the reliability and validity of the SWAL-QOL using item response theory (IRT; Rasch analysis). SWAL-QOL data were gathered from 507 participants at risk of oropharyngeal dysphagia (OD) across four European countries. OD was confirmed in 75.7% of participants via videofluoroscopy and/or fiberoptic endoscopic evaluation, or a clinical diagnosis based on meeting selected criteria. Patients with esophageal dysphagia were excluded. Data were analysed using Rasch analysis. Item and person reliability was good for all the items combined. However, person reliability was poor for 8 subscales and item reliability was poor for one subscale. Eight subscales exhibited poor person separation and two exhibited poor item separation. Overall item and person fit statistics were acceptable. However, at an individual item fit level results indicated unpredictable item responses for 28 items, and item redundancy for 10 items. The item-person dimensionality map confirmed these findings. Results from the overall Rasch model fit and Principal Component Analysis were suggestive of a second dimension. For all the items combined, none of the item categories were 'category', 'threshold' or 'step' disordered; however, all subscales demonstrated category disordered functioning. Findings suggest an urgent need to further investigate the underlying structure of the SWAL-QOL and its psychometric characteristics using IRT. Show less
As early diagnosis of swallowing and feeding difficulties in infants and children is of utmost importance, there is a need to evaluate the quality of the psychometric properties of pediatric... Show moreAs early diagnosis of swallowing and feeding difficulties in infants and children is of utmost importance, there is a need to evaluate the quality of the psychometric properties of pediatric assessments of swallowing and feeding. A systematic review was performed summarizing the psychometric properties of non-instrumental assessments for swallowing and feeding difficulties in pediatrics; no data were identified for the remaining twelve assessments. The COSMIN taxonomy and checklist were used to evaluate the methodological quality of 23 publications on psychometric properties. For each assessment, an overall quality score for each measurement property was determined. As psychometric data proved incomplete, conflicting or indeterminate for all assessments, only preliminary conclusions could be drawn; the most robust assessment based on current data is the dysphagia disorder survey (DDS). However, further research is needed to provide additional information on all psychometric properties for all assessments. Show less
Heijnen, B.J.; Speyer, R.; Bulow, M.; Kuijpers, L.M.F. 2016
Huntington__s disease (HD) is a progressive neurodegenerative disease with an autosomal, dominant mode of inheritance. Patients with HD suffer from dysphagia which can have serious consequences,... Show moreHuntington__s disease (HD) is a progressive neurodegenerative disease with an autosomal, dominant mode of inheritance. Patients with HD suffer from dysphagia which can have serious consequences, such as weight loss, dehydration, and pneumonia leading to death. Many patients with HD die of aspiration pneumonia. In order to start monitoring dysphagia in HD patients systematically during different stages of the disease, a new measurement instrument was needed. We developed the Huntington__s Disease Dysphagia Scale. The availability of this new scale now allows prospective monitoring of dysphagia in patients with HD. Another aim was to investigate specific dysphagia features in HD using videofluoroscopy. We found that 78% of our patients suffered from dysphagia. Swallowing difficulties start already in the first stage of HD, and become more severe as the disease progresses. The final aim was to initiate intervention studies on dysphagia in HD. We began by developing a guideline based on best practice. The guideline describes dysphagia problems per stage of HD, and makes recommendations for intervention and clinical advice. One of the recommendations in the European guideline is the chin tuck position while swallowing. As there is no evidence for improvement with this intervention for HD patients, we carried out a videofluoroscopic swallowing test with and without the chin tuck test procedure. No significant differences were found for spilling, aspiration or residue in swallowing with the head in normal upright position or the chin tuck posture. We therefore recommend that the chin tuck intervention should not be used in HD patients. Show less
Sporadische inclusion body myositis (IBM) is een van de meest voor voorkomende verworven spierziekte die ontstaat na het 50e levensjaar. In dit proefschrift worden de klinische aspecten van... Show moreSporadische inclusion body myositis (IBM) is een van de meest voor voorkomende verworven spierziekte die ontstaat na het 50e levensjaar. In dit proefschrift worden de klinische aspecten van sporadische IBM beschreven. Uit de studie met betrekking tot het natuurlijk beloop blijkt dat de ziekte niet levensverkortend is, maar dat de doodsoorzaken bij sporadische IBM wel verschillen ten opzichte van een voor de leeftijd gecorrigeerde populatie. De aard en frequentie van slikstoornissen worden beschreven, alsmede de potentiele betrokkenheid van het hart. Door middel van MRI's van skeletspieren is een voor sporadische IBM specifiek patroon van afwijkingen beschreven. Ten slotte is aangetoond dat TREX1 mutaties geen rol spelen in het ontstaan van de ziekte. Show less
Molen, L. van der; Rossum, M.A. van; Rasch, C.R.N.; Smeele, L.E.; Hilgers, F.J.M. 2014
The objective of the study was the assessment of the results of a prospective clinical trial with two preventive swallowing rehabilitation programs on the long-term side effects of... Show moreThe objective of the study was the assessment of the results of a prospective clinical trial with two preventive swallowing rehabilitation programs on the long-term side effects of chemoradiotherapy (CCRT) in advanced head and neck cancer patients. The study cohort consisted of 29 patients, randomized in two exercise groups: a standard (S) group receiving routine swallowing exercises (N = 14), and an experimental (E) group receiving swallowing exercises based on the TheraBiteA (R) Jaw Motion Rehabilitation System (TM) (N = 15). Assessment of functional changes was carried out with multidimensional outcome measures (e.g., videofluoroscopy, study-specific questionnaires) at four time points (pre-treatment, at 10 weeks, 1 year, and 2 years post-treatment). Overall, in the first year post-treatment many initial tumor- and treatment-related problems diminished significantly, except xerostomia (59 %). The only additional improvement at 2 years was that the overall weight significantly further increased (p = 0.000), however, without regaining baseline value. In the subgroup analysis according to exercise group, this difference was significant in the E-group only (p = 0.002). The same was the case for the subgroup analysis according to site of disease, with a significant weight gain in the 'below the hyoid bone' group only. This study shows that the overall functional problems at 1 and 2 years post-CCRT are limited. Both rehabilitation programs produce similar results, with a slight but significant benefit for the E-group in weight gain at 2 years, as also seen in the 'below the hyoid bone' group. Both rehabilitation programs applied are feasible and show good compliance despite the burdensome CCRT. Show less
Molen, L. van der; Heemsbergen, W.D.; Jong, R. de; Rossum, M.A. van; Smeele, L.E.; Rasch, C.R.N.; Hilgers, F.J.M. 2013
Background and purpose: Prospective assessment of dysphagia and trismus in chemo-IMRT head and neck cancer patients in relation to dose-parameters of structures involved in swallowing and... Show moreBackground and purpose: Prospective assessment of dysphagia and trismus in chemo-IMRT head and neck cancer patients in relation to dose-parameters of structures involved in swallowing and mastication.Material and methods: Assessment of 55 patients before, 10-weeks (N = 49) and 1-year post-treatment (N = 37). Calculation of dose-volume parameters for swallowing (inferior (IC), middle (MC), and superior constrictors (SC)), and mastication structures (e.g. masseter). Investigation of relationships between dose-parameters and endpoints for swallowing problems (videofluoroscopy-based laryngeal Penetration-Aspiration Scale (PAS), and study-specific structured questionnaire) and limited mouth-opening (measurements and questionnaire), taking into account baseline scores.Results: At 10-weeks, volume of IC receiving >= 60 Gy (V60) and mean dose IC were significant predictors for PAS. One-year post-treatment, reported problems with swallowing solids were significantly related to masseter dose-parameters (mean, V20, V40 and V60) and an inverse relationship (lower dose related to a higher probability) was observed for V60 of the IC. Dose-parameters of masseter and pterygoid muscles were significant predictors of trismus at 10-weeks (mean, V20, and V40). At 1-year, dose-parameters of all mastication structures were strong predictors for subjective mouth-opening problems (mean, max, V20, V40, and V60).Conclusions: Dose-effect relationships exist for dysphagia and trismus. Therefore treatment plans should be optimized to avoid these side effects. (C) 2013 Elsevier Ireland Ltd. All rights reserved. Show less
