Leber hereditary optic neuropathy in Czechia and Slovakia: Quality of life and costs from patient perspective.

Autor:	Bušányová B; Department of Paediatric Ophthalmology of the Faculty of Medicine, Comenius University Bratislava, And the National Institute of Children's Diseases in Bratislava, Slovakia., Vajter M; Department of Ophthalmology, First Faculty of Medicine, Charles University and General University Hospital in Prague, Czech Republic., Kelifová S; Department of Paediatrics and Inherited Metabolic Disorders, First Faculty of Medicine, Charles University and General University Hospital in Prague, Czech Republic., Lišková P; Department of Ophthalmology, First Faculty of Medicine, Charles University and General University Hospital in Prague, Czech Republic.; Department of Paediatrics and Inherited Metabolic Disorders, First Faculty of Medicine, Charles University and General University Hospital in Prague, Czech Republic., Miková H; Ophthalmological Outpatient Clinic, University Hospital - St. Michael's Hospital, Bratislava, Slovakia., Breciková K; CEEOR s.r.o, Prague, Czech Republic., Žigmond J; Department of Biomedical Technology, Faculty of Biomedical Engineering, Czech Technical University in Prague, Kladno, Czech Republic., Rogalewicz V; Department of Biomedical Technology, Faculty of Biomedical Engineering, Czech Technical University in Prague, Kladno, Czech Republic., Tichopád A; Department of Biomedical Technology, Faculty of Biomedical Engineering, Czech Technical University in Prague, Kladno, Czech Republic., Višňanský M; Department of Pharmacy and Social Pharmacy, University of Veterinary Medicine and Pharmacy in Košice, Slovakia.; Department of Public Economics, Faculty of Economics and Administration, Masaryk University in Brno, Czech Republic., Šarkanová I; Department of Biomedical Technology, Faculty of Biomedical Engineering, Czech Technical University in Prague, Kladno, Czech Republic.
Jazyk:	angličtina
Zdroj:	Heliyon [Heliyon] 2024 May 31; Vol. 10 (11), pp. e32296. Date of Electronic Publication: 2024 May 31 (Print Publication: 2024).
DOI:	10.1016/j.heliyon.2024.e32296
Abstrakt:	Introduction: Leber hereditary optic neuropathy (LHON) is the most frequent mitochondrial disease causing dyschromatopsia and progressive central visual loss that is subacute in progression and painless. Several studies have been published assessing QoL in patients with LHON, but no estimate of the economic burden has been reported to date. This study aims to quantify direct non-medical and indirect costs (productivity loss) incurred by LHON patients and their informal caregivers in Czechia and Slovakia, as well as to assess their quality of life. Methods: The study was performed in 27 adults and children with LHON. To determine the socioeconomic burden of LHON, separate questionnaires for adults, children, and their parents were developed, including demographic and socioeconomic data. The following data were collected: age, education, family size, severity of LHON, non-medical direct and indirect costs of LHON. Results: The mean age of adult respondents was 36.1 years (SD 13.1; n = 21). The total cost of absenteeism was EUR 1003 per person/year in adult employees, and EUR 2711 per person/year in children's parents. The productivity loss as a consequence of LHON due to combined relative absenteeism and relative presenteeism was estimated at EUR 9840 per an adult patient/year, and EUR 6298 per a parent/year, respectively. The mean cost of informal care was estimated at EUR 4502 (SD 4772; n = 6) per person/year. The mean VFQ-25 score for adult patients with LHON was 43.47 (SD 15.86). Conclusion: The results of this study clearly show that patients with LHON and their families face an extensive socioeconomic burden related to this rare disease. Early, timely and appropriate access to diagnosis, treatment, and reimbursement decisions, but also to psychological counselling and services may help the patients and their relatives adapt and cope with the challenging aspects of vision loss and life with the disease. Competing Interests: The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper. (© 2024 The Authors.)
Databáze:	MEDLINE
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