Survival in Huntington's disease and other young-onset dementias

被引:4
作者
Loi, Samantha M. [1 ,2 ,7 ]
Tsoukra, Paraskevi [3 ]
Sun, Emily [1 ]
Chen, Zhibin [4 ]
Wibawa, Pierre [1 ]
di Biase, Maria [2 ]
Farrand, Sarah [1 ]
Eratne, Dhamidhu [1 ,2 ,5 ]
Kelso, Wendy [1 ]
Evans, Andrew [6 ]
Walterfang, Mark [1 ,2 ,5 ]
Velakoulis, Dennis [1 ,2 ]
机构
[1] Royal Melbourne Hosp, Neuropsychiat, NorthWestern Mental Hlth, Melbourne Hlth, Parkville, Vic, Australia
[2] Univ Melbourne, Dept Psychiat, Parkville, Vic, Australia
[3] Evaggelismos Hosp, Dept Neurol, Athens, Greece
[4] Monash Univ, Sch Publ Hlth & Prevent Med, Clayton, Vic, Australia
[5] Florey Inst Neurosci & Mental Hlth, Parkville, Vic, Australia
[6] Royal Melbourne Hosp, Dept Med, Parkville, Vic, Australia
[7] Royal Melbourne Hosp, Neuropsychiat, John Cade Level 2, Parkville, Vic 3050, Australia
基金
英国医学研究理事会;
关键词
dementia; early-onset dementia; epidemiology; Huntington's disease; mortality; prognosis; risk factors; survival; young-onset dementia; FRONTOTEMPORAL DEMENTIA; BEHAVIORAL VARIANT; DEATH; DIAGNOSIS; C9ORF72; AGE; EXPANSION; DURATION; MUTATION;
D O I
10.1002/gps.5913
中图分类号
R592 [老年病学]; C [社会科学总论];
学科分类号
03 ; 0303 ; 100203 ;
摘要
Objectives: To compare survival and risk factors associated with mortality in common young-onset dementias (YOD) including Huntington's disease.Methods: This retrospective cohort study included inpatients from an Australian specialist neuropsychiatry service, over 20 years. Dementia diagnoses were based on consensus criteria and Huntington's disease (HD) was confirmed genetically. Mortality and cause of death were determined using linkage to the Australian Institute of Health and Welfare National Death Index.Results: There were 386 individuals with YOD included. The dementia types included frontotemporal dementia (FTD) (24.5%), HD (21.2%) and Alzheimer's disease (AD) (20.5%). 63% (n = 243) individuals had died. The longest median survival was for those who had HD, 18.8 years from symptom onset and with a reduced mortality risk compared to AD and FTD (hazard ratio 0.5). Overall, people with YOD had significantly increased mortality, of 5-8 times, compared to the general population. Females with a YOD had higher standardised mortality ratio compared to males (9.3 vs. 4.9) overall. The most frequent cause of death in those with HD was reported as HD, with other causes of death in the other YOD-subtypes related to dementia and mental/behavioural disorders.Discussion: This is the first Australian study to investigate survival and risk factors of mortality in people with YOD. YOD has a significant risk of death compared to the general population. Our findings provide useful clinical information for people affected by YOD as well as future planning and service provision.
引用
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页数:10
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