TY - JOUR

T1 - The burden of spinal muscular atrophy on informal caregivers

AU - Aranda-Reneo, Isaac

AU - Peña-Longobardo, Luz María

AU - Oliva-Moreno, Juan

AU - Litzkendorf, Svenja

AU - Durand-Zaleski, Isabelle

AU - Tizzano, Eduardo F.

AU - López-Bastida, Julio

N1 - Funding Information: Funding: This study was supported by an unrestricted grant from Biogen Idec, Inc., awarded to the University of Castilla–La Mancha (UCTR140345). I.AR., LM.PL., J.OM. and J.LB are employees of the University of Castilla– La Mancha, and part of this grant was distributed to their salaries for their time dedicated to developing the research. The specific roles of these authors are articulated in the author contributions section. The funder also paid the publication fee but had no role in the study design, data collection and analysis, decision to publish or preparation of the manuscript. Patients associations assisted with contacting patients, though neither the funder nor authors charged nor made payment.

PY - 2020/12/2

Y1 - 2020/12/2

N2 - Spinal muscular atrophy (SMA) is one of the most common severe hereditary diseases of infancy and early childhood. The progression of this illness causes a high degree of disability; hence, a significant burden is experienced by individuals with this disease and their families. We analyzed the time taken to care for patients suffering from SMA in European countries and the burden on their informal caregivers. We designed a cross-sectional study recording data from France, Germany, Spain and the United Kingdom. The primary caregivers completed a self-administered questionnaire that included questions about the time of care, The Zarit Burden Interview, type of SMA and socio-demographic characteristics. Multivariate analyses were used to study the associations between the type of SMA, time of care and burden supported by informal caregivers. The caregivers provided 10.0 h (SD = 6.7) per day of care (the principal caregivers provided 6.9 h, SD = 4.6). The informal caregivers of patients with type I SMA had a 36.3 point higher likelihood (p < 0.05) of providing more than 10 hours of care per day in comparison with caregivers of patients with type III SMA. The severity of the disease was associated with more time of care and a higher burden on the caregivers.

AB - Spinal muscular atrophy (SMA) is one of the most common severe hereditary diseases of infancy and early childhood. The progression of this illness causes a high degree of disability; hence, a significant burden is experienced by individuals with this disease and their families. We analyzed the time taken to care for patients suffering from SMA in European countries and the burden on their informal caregivers. We designed a cross-sectional study recording data from France, Germany, Spain and the United Kingdom. The primary caregivers completed a self-administered questionnaire that included questions about the time of care, The Zarit Burden Interview, type of SMA and socio-demographic characteristics. Multivariate analyses were used to study the associations between the type of SMA, time of care and burden supported by informal caregivers. The caregivers provided 10.0 h (SD = 6.7) per day of care (the principal caregivers provided 6.9 h, SD = 4.6). The informal caregivers of patients with type I SMA had a 36.3 point higher likelihood (p < 0.05) of providing more than 10 hours of care per day in comparison with caregivers of patients with type III SMA. The severity of the disease was associated with more time of care and a higher burden on the caregivers.

KW - Burden of disease

KW - Informal care

KW - Spinal muscular atrophy

UR - http://www.scopus.com/inward/record.url?scp=85097171746&partnerID=8YFLogxK

U2 - 10.3390/ijerph17238989

DO - 10.3390/ijerph17238989

M3 - Article

C2 - 33276656

AN - SCOPUS:85097171746

VL - 17

SP - 1

EP - 12

JO - International Journal of Environmental Research and Public Health

JF - International Journal of Environmental Research and Public Health

SN - 1661-7827

IS - 23

M1 - 8989

ER -