Evaluation of dysphagia by novel real-time MRI

Olthoff, Arno; Carstens, Per-Ole; Zhang, Shuo; Fintel, Eva von; Friede, Tim; Lotz, Joachim; Frahm, Jens; Schmidt, Jens

doi:10.1212/wnl.0000000000003337

Cited by 68 publications

(92 citation statements)

References 31 publications

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“…3 The IBMFRS has been extensively used in a number of cross-sectional, natural history, and drug trials. [4][5][6][7][8][9][10][11][12] It is also the primary endpoint of the phase 2/3 study of arimoclomol in IBM that started recently (NCT02753530). There is still value in optimizing the data obtained from the scale to improve the analysis of current and future trials and allow improved comparison with previous intervention and nonintervention studies.…”

Section: Accepted 14 May 2019mentioning

confidence: 99%

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Investigation of the psychometric properties of the inclusion body myositis functional rating scale with rasch analysis

et al. 2019

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Introduction The Inclusion Body Myositis Functional Rating Scale (IBMRFS) is a 10‐item clinician‐rated ordinal scale developed for people with inclusion body myositis. Methods Single observations of the IBMFRS were collected from 132 patients. After Rasch analysis, modifications were made to the scale to optimize fit to the Rasch model while maintaining clinical validity and utility. Results The original IBMFRS did not fit the assumptions of the Rasch model because of multidimensionality of the scale. Items assessed local dependence, disordered step thresholds, and differential item functioning. Deconstructing the scale into upper limb (IBMFRS‐UL) and lower limb (IBMFRS‐LL) scales improved fit to the Rasch model. A 9‐item scale with the swallowing item removed (IBMFRS‐9) remained multidimensional but demonstrated the ability to discriminate patients along the severity continuum. IBMFRS‐UL, IBMFRS‐LL, and IBMFRS‐9 scores were transformed to a 0–100 scale for comparability. Discussion This analysis has led to the development of 3 optimized versions of the IBMFRS. Muscle Nerve 60: 161–168, 2019

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Section: Accepted 14 May 2019mentioning

confidence: 99%

“…The IBMFRS has been extensively used in a number of cross‐sectional, natural history, and drug trials . It is also the primary endpoint of the phase 2/3 study of arimoclomol in IBM that started recently (NCT02753530).…”

mentioning

confidence: 99%

Investigation of the psychometric properties of the inclusion body myositis functional rating scale with rasch analysis

et al. 2019

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“…Comparisons between control and diseased/treatment groups are crucial for establishing valid VFSS markers, but due to radiation problems we limited the number of healthy controls compared with the number of SBMA patients. More recently, real‐time MRI was reported to display dysphagia equally as well as VFSS 39. Our study results will help select quantitative measures for real‐time MRI evaluations.…”

Section: Discussionmentioning

confidence: 62%

Swallowing markers in spinal and bulbar muscular atrophy

Banno

Katsuno

Suzuki

et al. 2017

Ann Clin Transl Neurol

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ObjectiveWe examined the characteristics of dysphagia in spinal and bulbar muscular atrophy, a hereditary neuromuscular disease causing weakness of limb, facial, and oropharyngeal muscles via a videofluoroscopic swallowing study, and investigated the plausibility of using these outcome measures for quantitative analysis.MethodsA videofluoroscopic swallowing study was performed on 111 consecutive patients with genetically confirmed spinal and bulbar muscular atrophy and 53 age‐ and sex‐matched healthy controls. Swallowing of 3‐mL liquid barium was analyzed by the Logemann's Videofluorographic Examination of Swallowing worksheet.ResultsOf more than 40 radiographic findings, the most pertinent abnormal findings in patients with spinal and bulbar muscular atrophy, included vallecular residue after swallow (residue just behind the tongue base), nasal penetration, and insufficient tongue movement (P < 0.001 for each) compared with healthy controls. Quantitative analyses showed that pharyngeal residue after initial swallowing, oral residue after initial swallowing, multiple swallowing sessions, and the penetration–aspiration scale were significantly worse in these patients (P ≤ 0.005 for each) than in controls. In patients with spinal and bulbar muscular atrophy, laryngeal penetration was observed more frequently in those without subjective dysphagia.InterpretationDysphagia of spinal and bulbar muscular atrophy was characterized by impaired tongue movement in the oral phase and nasal penetration followed by pharyngeal residues, which resulted in multiple swallowing sessions and laryngeal penetration. Although major limitations of reproducibility and radiation exposure still exist with videofluoroscopy, pharyngeal residue after initial swallowing and the penetration–aspiration scale might serve as potential outcome measures in clinical studies.

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“…Paraspinal and axial muscles may be affected, resulting in head drop and camptocormia 12. Depending on the study, oropharyngeal dysphagia is reported in up to 40–86% of IBM cases, mostly due to upper esophageal sphincter dysfunction 13, 14, 15, 16. It develops insidiously, leading to frequent choking episodes and is, alongside pneumonia as a result of immobility, considered to be a potentially fatal complication of IBM.…”

Section: Clinical Presentationmentioning

confidence: 99%

Immune and myodegenerative pathomechanisms in inclusion body myositis

Keller

Schmidt

Lünemann

2017

Ann Clin Transl Neurol

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Inclusion Body Myositis (IBM) is a relatively common acquired inflammatory myopathy in patients above 50 years of age. Pathological hallmarks of IBM are intramyofiber protein inclusions and endomysial inflammation, indicating that both myodegenerative and inflammatory mechanisms contribute to its pathogenesis. Impaired protein degradation by the autophagic machinery, which regulates innate and adaptive immune responses, in skeletal muscle fibers has recently been identified as a potential key pathomechanism in IBM. Immunotherapies, which are successfully used for treating other inflammatory myopathies lack efficacy in IBM and so far no effective treatment is available. Thus, a better understanding of the mechanistic pathways underlying progressive muscle weakness and atrophy in IBM is crucial in identifying novel promising targets for therapeutic intervention. Here, we discuss recent insights into the pathomechanistic network of mutually dependent inflammatory and degenerative events during IBM.

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Evaluation of dysphagia by novel real-time MRI

Cited by 68 publications

References 31 publications

Investigation of the psychometric properties of the inclusion body myositis functional rating scale with rasch analysis

Investigation of the psychometric properties of the inclusion body myositis functional rating scale with rasch analysis

Swallowing markers in spinal and bulbar muscular atrophy

Immune and myodegenerative pathomechanisms in inclusion body myositis

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