Reliability and validity of speech & pause measures during passage reading in ALS

Carolina Barnett, Jordan R Green, Reeman Marzouqah, Kaila L Stipancic, James D Berry, Lawrence Korngut, Angela Genge, Christen Shoesmith, Hannah Briemberg, Agessandro Abrahao, Sanjay Kalra, Lorne Zinman, Yana Yunusova, Carolina Barnett, Jordan R Green, Reeman Marzouqah, Kaila L Stipancic, James D Berry, Lawrence Korngut, Angela Genge, Christen Shoesmith, Hannah Briemberg, Agessandro Abrahao, Sanjay Kalra, Lorne Zinman, Yana Yunusova

Abstract

Objective: The use of speech measures is becoming a common practice in the assessment of bulbar disease progression in amyotrophic lateral sclerosis (ALS). This study aimed to establish psychometric properties (e.g. reliability, validity, sensitivity, specificity) of speech and pause timing measures during a standardized passage. Methods: A large number of passage recordings (ALS N = 775; Neurotypical controls N = 323) was analyzed using a semi-automatic method (Speech and Pause Analysis, SPA). Results: The results revealed acceptable reliability of the speech and pause measures across repeated recording by the control participants. Strong construct validity was established via significant group differences between patients and controls and correlation statistics with clinical measures of overall ALS and bulbar disease severity. Speaking rate, pause events, and mean pause duration were able to detect ALS participants at the presymptomatic stage of bulbar disease with a good discrimination ability (AUC 0.81). Conclusions: Based on the current psychometric evaluation, performing passage recording and speech and pause timing analysis was deemed useful for detecting early and progressive changes associated with bulbar ALS.

Keywords: Bulbar ALS; passage reading; pauses; reliability; speaking rate; validity.

Conflict of interest statement

Disclosure of interest: The authors report no conflict of interest.

Figures

Figure 1.
Figure 1.
The acoustic waveform recorded from a control participant with pauses marked in grey.

References

    1. Hecht M, Hillemacher T, Grasel E, Tigges S, Winterholler M, Heuss D, et al. Subjective experience and coping in ALS. ALS Other Mot Neuron Disord. 2002;3:225–32.
    1. Benatar M, Boylan K, Jeromin A, Rutkove SB, Berry J, Atassi N, et al. ALS biomarkers for therapy development: State of the field and future directions. Muscle and Nerve. 2016;53(2):169–82.
    1. Yunusova Y, Plowman EK, Green JR, Barnett C, Bede P. Clinical measures of bulbar dysfunction in ALS. Front Neurol. 2019;10:1–11.
    1. Yunusova Y, Ansari J, Ramirez J, Shellikeri S, Stanisz GJ, Black SE, et al. Frontal anatomical correlates of cognitive and speech motor deficits in amyotrophic lateral sclerosis. Behav Neurol. 2019;2019:1–11.
    1. Cedarbaum JM, Stambler N, Malta E, Fuller C, Hilt D, Thurmond B, et al. The ALSFRS-R: A revised ALS functional rating scale that incorporates assessments of respiratory function. J Neurol Sci. 1999;169(1–2):13–21.
    1. Plowman EK, Tabor LC, Wymer J, Pattee G. The evaluation of bulbar dysfunction in amyotrophic lateral sclerosis: Survey of clinical practice patterns in the United States. Amyotroph Lateral Scler Front Degener. 2017;18(5–6):351–7.
    1. Allison K, Yunusova Y, Campbell T, Wang J, Berry J, Green JR. The diagnostic utility of patient-report and speech-language pathologists’ ratings for detecting the early onset of bulbar symptoms due to ALS. Amyotroph Lateral Scler Frontotemporal Degener. 2017;18(5–6):358–66.
    1. Yorkston KM, Strand EA, Miller R, Hillel A, Smith K. Speech deterioration in amyotrophic lateral sclerosis: Implications for the timing of intervention. J Med Speech Lang Pathol. 1993;1:35–46.
    1. Ball, Laura J and Willis, Amy and Beukelman, David R and Pattee GL. A protocol for identification of early bulbar signs in amyotrophic lateral sclerosis. J Neurol Sci. 2001;191:43–53.
    1. Yorkston KM, Beukelman D, Hakel MDM. Sentence Intelligibility Test. Lincoln, Nebraska,: Madonna Rehabilitation Hospital,; 2007.
    1. Green JR, Yunusova Y, Kuruvilla MS, Wang J, Pattee GL, Synhorst L, et al. Bulbar and speech motor assessment in ALS: Challenges and future directions. Amyotroph Lateral Scler Frontotemporal Degener. 2013;14(7–8):494–500.
    1. Wang J, Kothalkar PV, Kim M, Bandini A, Cao B, Yunusova Y, et al. Automatic prediction of intelligible speaking rate for individuals with ALS from speech acoustic and articulatory samples. Int J Speech Lang Pathol. 2018. October;20(6):669–79.
    1. Green JR, Beukelman DR, Ball LJ. Algorithmic estimation of pauses in extended speech samples of dysarthric and typical speech. J Med Speech Lang Pathol. 2004;12(4):149–54.
    1. Yunusova Y, Graham NL, Shellikeri S, Phuong K, Kulkarni M, Rochon E, et al. Profiling speech and pausing in amyotrophic lateral sclerosis (ALS) and frontotemporal dementia (FTD). PLoS One. 2016;11(1):1–19.
    1. Green JR, Allison KM, Cordella C, Richburg BD, Pattee GL, Berry JD, et al. Additional evidence for a therapeutic effect of dextromethorphan/quinidine on bulbar motor function in patients with amyotrophic lateral sclerosis: A quantitative speech analysis. Br J Clin Pharmacol. 2018;84:2849–56.
    1. Smith R, Pioro E, Myers K, Sirdofsky M, Goslin K, Meekins G, et al. Enhanced bulbar function in amyotrophic lateral sclerosis: The Nuedexta treatment trial. Neurotherapeutics. 2017;14:762–72.
    1. Stipancic KL, Yunusova Y, Berry JD, Green JR. Minimally detectable change and minimal clinically important difference of a decline in sentence intelligibility and speaking rate for individuals with amyotrophic lateral sclerosis. J Speech, Lang Hear Res. 2018;61(11):2757–71.
    1. Brooks BR, Miller RG, Swash M, Munsat TL. El Escorial revisited: Revised criteria for the diagnosis of amyotrophic lateral sclerosis. Amyotroph Lateral Scler. 2000;1(5):293–9.
    1. Wang Y-T, Green JR, Nip ISB, Kent RD, Kent JF, and Ullman C, Accuracy of perceptually based and acoustically based inspiratory loci in reading. Behavior Research Methods, Instruments & Computers, 2010. 43(3): p. 791–797.
    1. Portney Leslie Gross MPW. Foundations of Clincial Research. Prentice Hall; 2009. 892.
    1. Bland JM, Altman DG. Statistical methods for assessing agreement between two methods of clinical measurement. Lancet. 1986;1(8476):307–10.
    1. Beaton DE, Bombardier C, Katz JN, Wright JG. A taxonomy for responsiveness. J Clin Epidemiol. 2001. December;54(12):1204–17.
    1. Bakdash JZ, Marusich LR. Repeated measures correlation. Front Psychol. 2017;8:1–13.
    1. Harrell F Regression modeling strategies: with applications to linear models, logistic regression, and survival analysis. J Am Stat Assoc. 2001;
    1. Team RC. R: A language and environment for statistical computing. R Found Stat Comput; 2013;
    1. Yorkston K, Beukelman D, Hakel M. Speech Intelligibility Test (SIT) for Windows [computer software]. Lincoln, Nebraska, USA: Madonna Rehabilitation Hospital; 2007.
    1. Mitchell HL, Hoit JD, Watson PJ. Cognitive-linguistic demands and speech breathing. J Speech, Lang Hear Res. 1996;39(1):93–104.

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