the relationship between muscle characteristics and speech ... · resume in dmd: • tongue is...
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The relationship between muscle characteristics and speech in Duchenne musculardystrophy, using ultrasound.
Dr. Simone Knuijt, speech-language pathologist and researcher
Radboudumc, Nijmegen
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Outline
• Duchenne muscular dystrophy
• Previous work• Muscle characteristics related to speech• Current research
Lenie van den Engel-Hoek, Marloes Lagarde, Simone Knuijt, Janna Maas
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Duchenne muscular dystrophy (DMD)
• Severe hereditary muscle disease, progressive
• Recessive, linked to X-chromosome• Women are carrier, boys are affected
• Mutations and deletions in the dystrophin gene
• Muscle fibers are interspersed with fibrosis and fat
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Stages of DMD
• Presymptomatic
Jungyoon Kim et al. A New Functional Scale and Ambulatory Functional Classification of Duchenne Muscular Dystrophy: Scale Development and Preliminary Analyses of Reliability and Validity. Annals of Rehabilitation Medicine 2018;42(5):690-701.
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Stages of DMD
• Presymptomatic
• Early ambulatory stage (EAS)
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Stages of DMD
• Presymptomatic
• Early ambulatory stage (EAS)• Late ambulatory stage (LAS)
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Stages of DMD
• Presymptomatic
• Early ambulatory stage (EAS)• Late ambulatory stage (LAS)• Early non-ambulatory stage (ENAS)
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Stages of DMD
• Presymptomatic
• Early ambulatory stage (EAS)• Late ambulatory stage (LAS)• Early non-ambulatory stage (ENAS)
• Late non-ambulatory stage (LNAS)
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Research in the past (in Radboudumc)
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Oral muscles in DMD (using quantitative ultrasound - QMUS)• Increased echogenicity
• Enlargement of muscles
S. Pillen, I. Arts, and M. Zwarts, 2008, Muscle Ultrasound in Neuromuscular Disorders, Muscle & Nerve, 37, p. 680.
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Longitudinal measurements
M .L.J. Lagarde, S. Knuijt, J.T. Groothuis, I.J.M. de Groot, L. van den Engel- Hoek. Longitudinal changes in oral and masticatory muscles in Duchenne Muscular Dystrophy: a disturbed balance. Poster ESSD
T1 (EAS) and T2 (EAS) T1 (EAS) and T2 (LAS)
T1 (ENAS) and T2 (ENAS) T1 (LNAS) and T2 (LNAS)
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QMUS of the flour of the mouth
Healthy DMD
Digastricmuscles
Geniohyoidmuscles
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QMUS of masseter muscle
DMD
?
Healthy
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QMUS of temporal muscle
Healthy DMD
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QMUS of the tongue
Healthy DMD
Superior longitudinalmuscle
Transversemuscle
?
?
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QMUS of tongue thickness
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Resume
In DMD:
• Tongue is affected (echogenicity and thickness).• Problems with mouth opening, chewing, swallowing, speech.• Relations between QMUS, chewing problems and dysphagia.
• What about the relationship between QMUS and speech?
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Hypothesis
• The dysarthria is expected to be more severe when the QMUS of the
tongue muscles are more affected.• There is a correlation between the acoustic characteristics and the QMUS
of the tongue muscles.
• Dysarthria severity increases when the respiratory function decreases.
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Measurements
• Radboud Dysarthria Assessment (Knuijt et al., 2014)
• Dysarthria severity (0-5)• Maximum phonation volume• Maximum phonation time
• Fundamental frequency range• Maximum repetition rate
• Articulation rate during reading
• Sentence intelligibility (NSVO-Z, Martens et al., 2010)• Quantitative muscle ultrasound (QMUS)• Mouth opening
• Respiratory function• Forced vital capacity (FVC)• Peak cough flow (PCF)
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Methods
• N=16 (mean age 24 y, range 19-32)
• 13 x LNAS, 3x intermediate type
• Descriptive statistics
• Correlations• Principal component analysis (PCA) to generate a:
• QMUS component (tongue thickness, echogenicity of superior
longitudinal muscle and transverse muscle)• Acoustic component (articulation rate, /ta/ and /ka/)
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Results – descriptives
• Dysarthria severity score 2 (mild dysarthria)
• Reduced maximum phonation volume (92 dB, norm is 100 dB*)
• Reduced maximum phonation time (7 sec, norm is 22 sec*)
• Reduced fundamental frequency range (24 semitones, norm 31*)
• Reduced speech intelligibility (80%, norm is 98%)
*Normative values derived from: Knuijt et al, Reference values of maximum performance tests of speech production. 2019 Int J Speech Lang Pathol. 21:1
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Results – descriptives
N M(SD) RangeEchogenicity superior longitudinal muscle(z-scores)
14 3.15 (2.02) -.08 – 6.28
Echogenicity transverse muscle(z-scores)
13 2.30 (1.84) .30 – 5.29
Thickness of the tongue(z-scores)
14 8.19 (3.02) 3.31 – 13.13
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Results – significant correlations
• Maximum phonation time with FVC: p = .001
• Maximum phonation volume with PCF: p = .026
• Dysarthria severity with:• QMUS: p = .031• FVC: p = .001
• PCF: p = .007
NB No significant correlations with intelligibility
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Results – components
• Speech acoustics with QMUS: significant (p = .001)
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Conclusion
• Mild dysarthria, characterized by reduced MPT, MPV, (FFR) and
intelligibility.Respiratory function
• But: QMUS correlated with acoustic component and dysarthria severity.Reduced articulatory distances?
• Additional analyses:• Coefficient of variance in DDK
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Current research
Philips Affinity 70
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Current research
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Current research
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“Ultrasound is a tool that is limited only by a
researcher's creativity” (Stone 1995)