Economic burden, work, and school productivity in individuals with tuberous sclerosis and their families

Anne M. Skalicky, Anne M. Rentz, Zhimei Liu, Qayyim Said, Jo Anne Nakagawa, Michael D. Frost, James W. Wheless, David Dunn

Research output: Contribution to journalArticle

Abstract

Aims: Tuberous sclerosis complex (TSC) is a multi-organ autosomal-dominant, genetic disorder with incomplete penetrance. The multiple manifestations of TSC and impacts to numerous organ systems represent significant disease, healthcare, and treatment burden. The economic and employment burden of the disease on individuals and their families is poorly understood. This study assessed the cost of illness and work and school productivity burden associated with TSC in a cross-sectional web-survey sample. Materials and methods: Eligible TSC individuals and caregivers were invited through the Tuberous Sclerosis Alliance advocacy group to complete a web-based survey about illness characteristics, treatment, disease burden, direct and indirect healthcare costs, work and school impairment. Results: Data from 609 TSC adults or caregiver respondents with no cognitive impairments were analyzed. TSC adults (>18 years of age) had significantly higher direct out-of-pocket costs for ER visits, expenses for medical tests and procedures, alternative treatments, medications and lifetime cost of surgeries compared to TSC pediatric individuals. Both TSC adults and TSC caregivers reported work and school absenteeism and presenteeism; however, adults reported significantly higher absenteeism and presenteeism and overall activity impairment due to TSC, as might be expected, compared to TSC caregivers. TSC adults had significantly higher absenteeism and presenteeism rates compared to adults with moderate-to-severe plaque psoriasis and muscular sclerosis. Conclusions: TSC results in considerable direct out-of-pocket medical costs and impairment to work productivity, especially for adults. Future studies should include the comparator group and examine direct cost burden in the US using electronic medical records and insurance databases.

Original languageEnglish (US)
Pages (from-to)1-7
Number of pages7
JournalJournal of Medical Economics
DOIs
StateAccepted/In press - Jun 28 2018

Fingerprint

Tuberous Sclerosis
Economics
Efficiency
Absenteeism
Caregivers
Cost of Illness
Health Expenditures
Costs and Cost Analysis
Inborn Genetic Diseases
Penetrance
Electronic Health Records
Sclerosis
Insurance
Psoriasis
Health Care Costs

Keywords

  • direct healthcare costs
  • Economic burden
  • tuberous sclerosis complex
  • work productivity and activity impairment (WPAI)

ASJC Scopus subject areas

  • Health Policy

Cite this

Economic burden, work, and school productivity in individuals with tuberous sclerosis and their families. / Skalicky, Anne M.; Rentz, Anne M.; Liu, Zhimei; Said, Qayyim; Nakagawa, Jo Anne; Frost, Michael D.; Wheless, James W.; Dunn, David.

In: Journal of Medical Economics, 28.06.2018, p. 1-7.

Research output: Contribution to journalArticle

Skalicky, Anne M. ; Rentz, Anne M. ; Liu, Zhimei ; Said, Qayyim ; Nakagawa, Jo Anne ; Frost, Michael D. ; Wheless, James W. ; Dunn, David. / Economic burden, work, and school productivity in individuals with tuberous sclerosis and their families. In: Journal of Medical Economics. 2018 ; pp. 1-7.
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