Epidemiology and economic burden of Huntington’s disease: a Canadian provincial public health system perspective

Shaw, Eileen; Mayer, Michelle L.; Ekwaru, Paul; McMullen, Suzanne; Graves, Erin; Wu, Jiao; Budd, Nathalie; Maturi, B; Cowling, Thomas E; Mestre, Tiago

doi:10.1080/13696998.2022.2033493

Cited by 11 publications

(14 citation statements)

References 28 publications

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“…A total of 18 studies examined the incidence of HD, with geographic representation across 12 countries (Canada, 12 Cyprus, 10 England, 9 , 22 , 23 Iceland, 24 Israel, 25 Italy, 26 , 27 , 28 Germany, 29 Greece, 30 South Korea, 31 Spain, 32 , 33 Sweden, 34 and the United States 35 , 36 ) (see Table S1 for study details).…”

Section: Resultsmentioning

confidence: 99%

“…A total of 27 studies explored the prevalence of HD, representing 21 countries (Brazil, 37 , 38 Cameroon, 39 Canada, 7 , 12 Cyprus, 10 Denmark, 40 England, 5 , 22 Finland, 41 Iceland, 24 Iran, 21 Israel, 25 Italy, 8 , 26 , 42 Germany, 29 Greece, 30 Northern Ireland, 43 Scotland, 11 South Africa, 6 South Korea, 31 Spain, 33 Sultanate of Oman, 44 Sweden, 45 and the United States 35 , 36 ) (see Table S2 for study details).…”

Section: Resultsmentioning

confidence: 99%

“…The pooled incidence of HD based on studies published from 1985 to 2010 has been estimated as 0.38 per 100,000 person‐years (95% confidence interval [CI], 0.16–0.94), with a global overall prevalence of 2.71 per 100,000 (95% CI, 1.55–4.72) 4 . More recent studies highlight that there is an increment in prevalence in some regions 5–12 . Although there are plausible explanations for variations and increased rates across populations, such as the identification of different HTT gene haplotypes, health care accessibility, attitudes that differ on illness‐related stigma, migration, and the identification of HD cluster regions, specific determinants remain to be elucidated.…”

mentioning

confidence: 99%

“… 4 More recent studies highlight that there is an increment in prevalence in some regions. 5 , 6 , 7 , 8 , 9 , 10 , 11 , 12 Although there are plausible explanations for variations and increased rates across populations, such as the identification of different HTT gene haplotypes, health care accessibility, attitudes that differ on illness‐related stigma, migration, and the identification of HD cluster regions, specific determinants remain to be elucidated.…”

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confidence: 99%

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Prevalence and Incidence of Huntington's Disease: An Updated Systematic Review and Meta‐Analysis

et al. 2022

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The incidence and prevalence of Huntington's disease (HD) based on a systematic review and meta‐analysis of 20 studies published from 1985 to 2010 was estimated at 0.38 per 100,000 person‐years (95% confidence interval [CI], 0.16–0.94) and 2.71 per 100,000 persons (95% CI, 1.55–4.72), respectively. Since 2010, there have been many new epidemiological studies of HD. We sought to update the global estimates of HD incidence and prevalence using data published up to February 2022 and perform additional analyses based on study continent. Medline and Embase were searched for epidemiological studies of HD published between 2010 and 2022. Risk of bias was assessed using a quality assessment tool. Estimated pooled prevalence or incidence was calculated using a random‐effects meta‐analysis. A total of 33 studies published between 2010 and 2022 were included. Pooled incidence was 0.48 cases per 100,000 person‐years (95% CI, 0.33–0.63). Subgroup analysis by continent demonstrated a significantly higher incidence of HD in Europe and North America than in Asia. Pooled prevalence was 4.88 per 100,000 (95% CI, 3.38–7.06). Subanalyses by continent demonstrated that the prevalence of HD was significantly higher in Europe and North America than in Africa. The minor increase in prevalence (more so than incidence) demonstrated in this updated review could relate to the enhanced availability of molecular testing, earlier diagnosis, increased life expectancy, and de novo mutations. Limitations include variable case ascertainment methods and lacking case validation data. © 2022 Her Majesty the Queen in Right of Canada. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society. Reproduced with the permission of the Minister of Public Health Agency of Canada.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

mentioning

confidence: 99%

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confidence: 99%

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Prevalence and Incidence of Huntington's Disease: An Updated Systematic Review and Meta‐Analysis

et al. 2022

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“…In addition to being part of the behavioral neuropathology of HD, the disease also has its own specific life stressors due to individuals struggling with relatives affected by the illness and the issues related to presymptomatic genetic testing [16]. Several studies have described the burden of HD in a routine care setting; however, these studies have typically placed an emphasis on healthcare resource use and associated economic burden, often over shorter time periods, with less emphasis on the elevated incidence of other comorbidities and symptoms associated with HD [17][18][19][20][21][22].…”

Section: Introductionmentioning

confidence: 99%

Comorbidities and clinical outcomes in adult- and juvenile-onset Huntington’s disease: a study of linked Swedish National Registries (2002–2019)

et al. 2022

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Background Huntington’s disease (HD) is a rare, neurodegenerative disease and its complex motor, cognitive and psychiatric symptoms exert a lifelong clinical burden on both patients and their families. Objective To describe the clinical burden and natural history of HD. Methods This longitudinal cohort study used data from the linked Swedish national registries to describe the occurrence of comorbidities (acute and chronic), symptomatic treatments and mortality in an incident cohort of individuals who either received the first diagnosis of HD above (adult onset HD; AoHD) or below (juvenile-onset HD; JoHD) 20 years of age, compared with a matched cohort without HD from the general population. Disease burden of all individuals alive in Sweden was described during a single calendar year (2018), including the occurrence of key symptoms, treatments and hospitalizations. Results The prevalence of HD in 2018 was approximately 10.2 per 100,000. Of 1492 individuals with a diagnosis of HD during 2002 and 2018, 1447 had AoHD and 45 had JoHD. Individuals with AoHD suffered a higher incidence of obsessive–compulsive disorder, acute psychotic episodes, pneumonia, constipation and fractures compared with matched controls. Individuals with JoHD had higher incidence rates of epilepsy, constipation and acute respiratory symptoms. Median time to all-cause mortality in AoHD was 12.1 years from diagnosis. Patients alive with HD in Sweden in 2018 displayed a pattern of increased clinical burden for a number of years since diagnosis. Conclusions This study demonstrates the significant and progressive clinical burden in individuals with HD and presents novel insights into the natural history of JoHD.

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Epidemiology of Huntington's Disease in Latin America: A Systematic Review and Meta‐Analysis

Medina Escobar,

Pringsheim,

Gautreau

et al. 2024

Movement Disorders

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BackgroundLatin America has played a crucial role in advancing our understanding of Huntington's disease (HD). However, previous global reviews include limited data from Latin America. It is possible that English‐based medical search engines may not capture all the relevant studies.MethodsWe searched databases in Spanish, Portuguese, and English. The names of every country in Latin America in English‐based search engines were used to ensure we found any study that had molecular ascertainment and provided general epidemiological information or subpopulation data. Additionally, we contacted experts across the region.ResultsThe search strategy yielded 791 citations; 24 studies met inclusion criteria, representing 12 of 36 countries. The overall pooled prevalence was 0.64 per 100,000 (prediction interval, 0.06–7.22); for cluster regions, it was 54 per 100,000 (95% CI, 34.79–84.92); for juvenile HD, it was 8.7% (prediction interval, 5.12–14.35), and 5.9% (prediction interval, 2.72–13.42) for late‐onset HD. The prevalence was higher for Mexico, Peru, and Brazil. However, there were no significant differences between Central America and the Caribbean versus South America.ConclusionThe prevalence of HD appears to be similar across Latin America. However, we infer that our findings are underestimates, in part because of limited research and underdiagnosis of HD because of limited access to molecular testing and the availability of neurologists and movement disorders specialists. Future research should focus on identifying pathways to improve access to molecular testing and education and understanding differences among different ancestral groups in Latin America. © 2024 International Parkinson and Movement Disorder Society.

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Epidemiology and economic burden of Huntington’s disease: a Canadian provincial public health system perspective

Cited by 11 publications

References 28 publications

Prevalence and Incidence of Huntington's Disease: An Updated Systematic Review and Meta‐Analysis

Prevalence and Incidence of Huntington's Disease: An Updated Systematic Review and Meta‐Analysis

Comorbidities and clinical outcomes in adult- and juvenile-onset Huntington’s disease: a study of linked Swedish National Registries (2002–2019)

Epidemiology of Huntington's Disease in Latin America: A Systematic Review and Meta‐Analysis

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