Understanding speech and swallowing difficulties in individuals with Huntington disease: Validation of the HDQLIFE Speec
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Understanding speech and swallowing difficulties in individuals with Huntington disease: Validation of the HDQLIFE Speech Difficulties and Swallowing Difficulties Item Banks Noelle E. Carlozzi1 · Nicholas R. Boileau1 · Angela Roberts2 · Praveen Dayalu3 · Dana L. Hanifan4 · Jennifer A. Miner1 · Daniel Claassen5 · Emily Mower Provost6 Accepted: 7 August 2020 © Springer Nature Switzerland AG 2020
Abstract Purpose As Huntington disease (HD) progresses, speech and swallowing difficulties become more profound. These difficulties have an adverse effect on health-related quality of life (HRQOL), thus psychometrically robust measures of speech and swallowing are needed to better understand the impact of these domains across the course of the disease. Therefore, the purpose of this study is to establish the clinical utility of two new patient-reported outcome measures (PROs), HDQLIFE Speech Difficulties and HDQLIFE Swallowing Difficulties. Methods Thirty-one participants with premanifest or manifest HD, and 31 age- and sex-matched healthy control participants were recruited for this study. Participants completed several PROs [HDQLIFE Speech Difficulties, HDQLIFE Swallowing Difficulties, Communication Participation Item Bank (CPIB)], as well as several clinician-rated assessments of speech and functioning. A computational algorithm designed to detect features of spoken discourse was also examined. Analyses were focused on establishing the reliability and validity of these new measures. Results Internal consistency was good for Swallowing (Cronbach’s alpha = 0.89) and excellent for Speech and the CPIB (both Cronbach’s alpha ≥ 0.94), and convergent/discriminant validity was supported. Known groups validity for the PROs was supported by significant group differences among control participants and persons with different stages of HD (all p 35 and a diagnostic confidence rating of 0.85 (for each individual behavior coded) before independently coding the data. Orthographic transcriptions were coded for speech fluency disruptions using CHAT discourse coding conventions [69]. Each research assistant independently coded all transcriptions (blinded to HD status); initial ICCs were 85% (without coding for dialect; 84% with coding for dialect). All discrepancies were resolved through consensus agreement. The data were automatically transcribed using speech recognition systems trained on speech patterns from individuals with HD. These automatic transcripts were used in conjunction with the recorded audio to extract speech
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Clinician-Rated Assessments of Functioning UHDRS Motor Assessment [30]
World Health Organization Disability Assessment Schedule (WHODAS 2.0) [59, 60]
EQ5D Index Scale [50]
Communicative Participation Impairment Bank (CPIB) [49]
HDQLIFE Swallowing Difficulties [6, 36]
Patient-Reported Outcome Measures HDQLIFE Speech Difficulties [6, 36]
Counting from 1 to 75 [46]
Sustained/a/[45]**
Objective motor speech and spoken language tasks Standardized Reading Passage (The Grandfather Passage [42,
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