Avery Shuei-He Yang, Daniel Hsiang-Te Tsai, Li-Wen Chen, Wen-Mei Cheng, Chu Chiao Wang, Shih-Chieh Shao, Wendy Wenjie Song, Mahsa H Kouhkamari, Wei-Ching Huang, Sally Bowditch, Edward Chia-Cheng Lai
{"title":"Epidemiology and Burden of Illness of Lennox-Gastaut Syndrome in Taiwan: A Retrospective Cohort Study.","authors":"Avery Shuei-He Yang, Daniel Hsiang-Te Tsai, Li-Wen Chen, Wen-Mei Cheng, Chu Chiao Wang, Shih-Chieh Shao, Wendy Wenjie Song, Mahsa H Kouhkamari, Wei-Ching Huang, Sally Bowditch, Edward Chia-Cheng Lai","doi":"10.2147/RMHP.S519367","DOIUrl":null,"url":null,"abstract":"<p><strong>Purpose: </strong>Examine the epidemiology and burden of illness of patients with Lennox-Gastaut syndrome (LGS) in Taiwan.</p><p><strong>Methods: </strong>The National Health Insurance Database (NHID) was used, complemented by the Chang Gung Research Database (CGRD). Confirmed LGS was defined by International Classification of Diseases-10 (ICD-10) LGS codes or ≥1 rufinamide prescription; probable LGS was defined as patients aged ≤10 years when receiving ≥3 antiseizure medications (ASMs), with ICD-9/10 codes for developmental delay. Independent clinical review/validation of all LGS cases in the CGRD was conducted. Prevalence, incidence, time to treatment/LGS diagnosis, hospitalizations, costs (US dollars), ASM usage, and mortality were assessed. For the NHID, a positive predicted value (PPV) was calculated from the CGRD validation step to adjust prevalence/incidence estimates.</p><p><strong>Results: </strong>In the NHID, 190 patients with confirmed LGS were identified. In 2018, PPV-adjusted prevalence was 2.4, 0.6, and 10.2 per 100,000 people and PPV-adjusted incidence was 0.6, 0.2, and 2.4 per 100,000 person-years in the total, adult, and pediatric populations, respectively. Of 92 (48%) hospitalized patients, 22 (24%) had ≥3 hospitalizations/year. Mean (standard deviation [SD]) time to treatment and LGS diagnosis were 12.3 (26.5) and 110.1 (54.7) months. Mean length of stay was around 12 days. Most hospitalizations (96%) were epilepsy related. Mean (SD) hospitalization cost was $237 ($216) per day. Mean (SD) total inpatient and outpatient costs were $5800 ($817) and $2667 ($132), respectively, per patient per year (PPPY); medication in hospital and ASM costs were $1910 ($108) and $1614 ($93) PPPY. Most prescribed ASMs were valproate (89%), levetiracetam (83%), clonazepam (69%), clobazam (68%), and topiramate (65%). Mortality was 0.01 deaths per 100,000 people in 2018.</p><p><strong>Conclusion: </strong>Although prevalence and incidence of LGS in Taiwan were lower than in other countries, the multifaceted burden of illness in LGS is highlighted herein. Reduced hospitalizations through better epilepsy control may reduce LGS expenditure.</p>","PeriodicalId":56009,"journal":{"name":"Risk Management and Healthcare Policy","volume":"18 ","pages":"3153-3166"},"PeriodicalIF":2.0000,"publicationDate":"2025-09-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12459616/pdf/","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Risk Management and Healthcare Policy","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.2147/RMHP.S519367","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/1/1 0:00:00","PubModel":"eCollection","JCR":"Q2","JCRName":"HEALTH CARE SCIENCES & SERVICES","Score":null,"Total":0}
引用次数: 0
Abstract
Purpose: Examine the epidemiology and burden of illness of patients with Lennox-Gastaut syndrome (LGS) in Taiwan.
Methods: The National Health Insurance Database (NHID) was used, complemented by the Chang Gung Research Database (CGRD). Confirmed LGS was defined by International Classification of Diseases-10 (ICD-10) LGS codes or ≥1 rufinamide prescription; probable LGS was defined as patients aged ≤10 years when receiving ≥3 antiseizure medications (ASMs), with ICD-9/10 codes for developmental delay. Independent clinical review/validation of all LGS cases in the CGRD was conducted. Prevalence, incidence, time to treatment/LGS diagnosis, hospitalizations, costs (US dollars), ASM usage, and mortality were assessed. For the NHID, a positive predicted value (PPV) was calculated from the CGRD validation step to adjust prevalence/incidence estimates.
Results: In the NHID, 190 patients with confirmed LGS were identified. In 2018, PPV-adjusted prevalence was 2.4, 0.6, and 10.2 per 100,000 people and PPV-adjusted incidence was 0.6, 0.2, and 2.4 per 100,000 person-years in the total, adult, and pediatric populations, respectively. Of 92 (48%) hospitalized patients, 22 (24%) had ≥3 hospitalizations/year. Mean (standard deviation [SD]) time to treatment and LGS diagnosis were 12.3 (26.5) and 110.1 (54.7) months. Mean length of stay was around 12 days. Most hospitalizations (96%) were epilepsy related. Mean (SD) hospitalization cost was $237 ($216) per day. Mean (SD) total inpatient and outpatient costs were $5800 ($817) and $2667 ($132), respectively, per patient per year (PPPY); medication in hospital and ASM costs were $1910 ($108) and $1614 ($93) PPPY. Most prescribed ASMs were valproate (89%), levetiracetam (83%), clonazepam (69%), clobazam (68%), and topiramate (65%). Mortality was 0.01 deaths per 100,000 people in 2018.
Conclusion: Although prevalence and incidence of LGS in Taiwan were lower than in other countries, the multifaceted burden of illness in LGS is highlighted herein. Reduced hospitalizations through better epilepsy control may reduce LGS expenditure.
期刊介绍:
Risk Management and Healthcare Policy is an international, peer-reviewed, open access journal focusing on all aspects of public health, policy and preventative measures to promote good health and improve morbidity and mortality in the population. Specific topics covered in the journal include:
Public and community health
Policy and law
Preventative and predictive healthcare
Risk and hazard management
Epidemiology, detection and screening
Lifestyle and diet modification
Vaccination and disease transmission/modification programs
Health and safety and occupational health
Healthcare services provision
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Health economic evaluations and resource management
Risk Management and Healthcare Policy focuses on human interventional and observational research. The journal welcomes submitted papers covering original research, clinical and epidemiological studies, reviews and evaluations, guidelines, expert opinion and commentary, and extended reports. Case reports will only be considered if they make a valuable and original contribution to the literature. The journal does not accept study protocols, animal-based or cell line-based studies.