TY - JOUR
T1 - Understanding speech and swallowing difficulties in individuals with Huntington disease
T2 - Validation of the HDQLIFE Speech Difficulties and Swallowing Difficulties Item Banks
AU - Carlozzi, Noelle E.
AU - Boileau, Nicholas R.
AU - Roberts, Angela
AU - Dayalu, Praveen
AU - Hanifan, Dana L.
AU - Miner, Jennifer A.
AU - Claassen, Daniel
AU - Provost, Emily Mower
N1 - Funding Information:
Convergent and discriminant validity correlation coefficients are reported in Table . Correlations among the PRO measures were large supporting convergent validity. Discriminant validity was supported by moderate correlations among the HDQLIFE PROs and other measures of clinician-rated speech and the Algorithm-based features of spoken discourse.
Funding Information:
Convergent and discriminant validity of the HDQLIFE PROs were also supported by our findings. Specifically, convergent validity was supported by moderate to strong associations (all ≥ 0.44) among the three PROs. Furthermore, discriminant validity was supported by moderate correlations (generally between 0.40 and 0.60) among the PROs and other measures of clinician-rated speech and the algorithm-based features of spoken discourse. This pattern of findings was consistent with our proposed hypotheses and provides support for the validity of the HDQLIFE Speech and Swallowing Difficulties PROs in persons with HD.
Funding Information:
We thank the Investigators and Coordinators of this study, the study participants, the Huntington Study Group, and the Huntington?s Disease Society of America.
Publisher Copyright:
© 2020, Springer Nature Switzerland AG.
PY - 2021/1
Y1 - 2021/1
N2 - 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 < 0.0001). All PROs were able to distinguish those with and without clinician-rated dysarthria (likelihood ratios far exceeded the threshold for clinical decision making [all ≥ 3.28]). Conclusions: Findings support the clinical utility of the HDQLIFE Speech and Swallowing PROs and the CPIB for use across the HD disease spectrum. These PROs also have the potential to be clinically useful in other populations.
AB - 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 < 0.0001). All PROs were able to distinguish those with and without clinician-rated dysarthria (likelihood ratios far exceeded the threshold for clinical decision making [all ≥ 3.28]). Conclusions: Findings support the clinical utility of the HDQLIFE Speech and Swallowing PROs and the CPIB for use across the HD disease spectrum. These PROs also have the potential to be clinically useful in other populations.
KW - Health-related quality of life
KW - Huntington disease
KW - Patient-reported outcome measures
KW - Speech difficulties
KW - Swallowing difficulties
UR - http://www.scopus.com/inward/record.url?scp=85089782502&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85089782502&partnerID=8YFLogxK
U2 - 10.1007/s11136-020-02608-0
DO - 10.1007/s11136-020-02608-0
M3 - Article
C2 - 32839864
AN - SCOPUS:85089782502
SN - 0962-9343
VL - 30
SP - 251
EP - 265
JO - Quality of Life Research
JF - Quality of Life Research
IS - 1
ER -
