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

Kód výsledku v IS VaVaI

<a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F00216224%3A14560%2F24%3A00140058" target="_blank" >RIV/00216224:14560/24:00140058 - isvavai.cz</a>

Nalezeny alternativní kódy

RIV/68407700:21460/24:00366384 RIV/00216208:11110/24:10483306 RIV/00064165:_____/24:10483306

Výsledek na webu

<a href="https://www.cell.com/heliyon/fulltext/S2405-8440(24)08327-0?_returnURL=https%3A%2F%2Flinkinghub.elsevier.com%2Fretrieve%2Fpii%2FS2405844024083270%3Fshowall%3Dtrue" target="_blank" >https://www.cell.com/heliyon/fulltext/S2405-8440(24)08327-0?_returnURL=https%3A%2F%2Flinkinghub.elsevier.com%2Fretrieve%2Fpii%2FS2405844024083270%3Fshowall%3Dtrue</a>

DOI - Digital Object Identifier

<a href="http://dx.doi.org/10.1016/j.heliyon.2024.e32296" target="_blank" >10.1016/j.heliyon.2024.e32296</a>

Jazyk výsledku

angličtina

Název v původním jazyce

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

Popis výsledku v původním jazyce

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

Název v anglickém jazyce

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

Popis výsledku anglicky

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

Druh

J_imp - Článek v periodiku v databázi Web of Science

CEP obor

—

OECD FORD obor

30300 - Health sciences

Projekt

<a href="/cs/project/NU22-07-00614" target="_blank" >NU22-07-00614: Mitochondriální optické neuropatie: komplexní genetická a metabolomická analýza k určení vzácných příčin onemocnění a hledání nových biomarkerů</a><br>

Návaznosti

I - Institucionalni podpora na dlouhodoby koncepcni rozvoj vyzkumne organizace

Rok uplatnění

2024

Kód důvěrnosti údajů

S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů

Název periodika

Heliyon

ISSN

2405-8440

e-ISSN

2405-8440

Svazek periodika

10

Číslo periodika v rámci svazku

11

Stát vydavatele periodika

GB - Spojené království Velké Británie a Severního Irska

Počet stran výsledku

8

Strana od-do

1-8

Kód UT WoS článku

001274230800001

EID výsledku v databázi Scopus

2-s2.0-85195218775