TY - JOUR
T1 - Validation of the first quality-of-life measurement for patients with Huntington's disease
T2 - The Huntington quality of life instrument
AU - Clay, Emilie
AU - De Nicola, Annunziata
AU - Dorey, Julie
AU - Squitieri, Ferdinando
AU - Aballéa, Samuel
AU - Martino, Tiziana
AU - Tedroff, Joakim
AU - Zielonka, Daniel
AU - Auquier, Pascal
AU - Verny, Christophe
AU - Toumi, Mondher
PY - 2012/7
Y1 - 2012/7
N2 - Health-related quality-of-life instruments are critical for assessing disease burdens. Generic tools allow comparison between diseases but do not discriminate between disease severities. Specific tools also tend to be more sensitive. No specific tool is available to assess quality of life in patients with Huntington's disease (HD). In the context of the European study on HD burden, a specific tool was created: the Huntington Quality of Life Instrument (H-QoL-I). The aim of this study was to optimize the content and validate the H-QoL-I. After a semistructured interview with patients, caregivers and HD specialists, we conducted a patient focus group. A self-reported questionnaire was then developed in French and Italian. A total of 252 patients were recruited to answer the questionnaire. Face, internal and external validities were examined using a variety of methods. The shortened H-QoL-I that resulted from the successive analyses comprises 11 items, which are divided into three dimensions: motor functioning (four items), psychology (four items) and socializing (three items). These three domains were identified as being essential to cover the full domain of the quality of life for patients affected by HD. The H-QoL-I showed an acceptable reliability (Cronbach's α>0.84). Factor analyses demonstrated satisfactory construct validity. Moreover, the item internal consistency and item discriminant validity criteria were fulfilled. No differential item functioning was detected. External validity supported the scale's robustness. These data support the validity of the H-QoL-I in patients with HD.
AB - Health-related quality-of-life instruments are critical for assessing disease burdens. Generic tools allow comparison between diseases but do not discriminate between disease severities. Specific tools also tend to be more sensitive. No specific tool is available to assess quality of life in patients with Huntington's disease (HD). In the context of the European study on HD burden, a specific tool was created: the Huntington Quality of Life Instrument (H-QoL-I). The aim of this study was to optimize the content and validate the H-QoL-I. After a semistructured interview with patients, caregivers and HD specialists, we conducted a patient focus group. A self-reported questionnaire was then developed in French and Italian. A total of 252 patients were recruited to answer the questionnaire. Face, internal and external validities were examined using a variety of methods. The shortened H-QoL-I that resulted from the successive analyses comprises 11 items, which are divided into three dimensions: motor functioning (four items), psychology (four items) and socializing (three items). These three domains were identified as being essential to cover the full domain of the quality of life for patients affected by HD. The H-QoL-I showed an acceptable reliability (Cronbach's α>0.84). Factor analyses demonstrated satisfactory construct validity. Moreover, the item internal consistency and item discriminant validity criteria were fulfilled. No differential item functioning was detected. External validity supported the scale's robustness. These data support the validity of the H-QoL-I in patients with HD.
KW - disease burden
KW - health-related quality of life
KW - Huntingtona's disease
KW - specific instrument
KW - validation
UR - http://www.scopus.com/inward/record.url?scp=84862122854&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=84862122854&partnerID=8YFLogxK
U2 - 10.1097/YIC.0b013e3283534fa9
DO - 10.1097/YIC.0b013e3283534fa9
M3 - Article
C2 - 22508443
AN - SCOPUS:84862122854
VL - 27
SP - 208
EP - 214
JO - International Clinical Psychopharmacology
JF - International Clinical Psychopharmacology
SN - 0268-1315
IS - 4
ER -