TY  - JOUR
A1  - Wohnrade, Camilla
A1  - Velling, Ann-Kathrin
A1  - Mix, Lucas
A1  - Wurster, Claudia D.
A1  - Cordts, Isabell
A1  - Stolte, Benjamin
A1  - Zeller, Daniel
A1  - Uzelac, Zeljko
A1  - Platen, Sophia
A1  - Hagenacker, Tim
A1  - Deschauer, Marcus
A1  - Lingor, Paul
A1  - Ludolph, Albert C.
A1  - Lulé, Dorothée
A1  - Petri, Susanne
A1  - Osmanovic, Alma
A1  - Schreiber-Katz, Olivia
T1  - Health-related quality of life in spinal muscular atrophy patients and their caregivers — a prospective, cross-sectional, multi-center analysis
JF  - Brain Sciences
N2  - Spinal muscular atrophy (SMA) is a disabling disease that affects not only the patient’s health-related quality of life (HRQoL), but also causes a high caregiver burden (CGB). The aim of this study was to evaluate HRQoL, CGB, and their predictors in SMA. In two prospective, cross-sectional, and multi-center studies, SMA patients (n = 39) and SMA patient/caregiver couples (n = 49) filled in the EuroQoL Five Dimension Five Level Scale (EQ-5D-5L) and the Short Form Health Survey 36 (SF-36). Caregivers (CGs) additionally answered the Zarit Burden Interview (ZBI) and the Hospital Anxiety and Depression Scale (HADS). Patients were clustered into two groups with either low or high HRQoL (EQ-5D-5L index value <0.259 or >0.679). The latter group was mostly composed of ambulatory type III patients with higher motor/functional scores. More severely affected patients reported low physical functioning but good mental health and vitality. The CGB (mean ZBI = 22/88) correlated negatively with patients’ motor/functional scores and age. Higher CGB was associated with a lower HRQoL, higher depression and anxiety, and more health impairments of the CGs. We conclude that patient and CG well-being levels interact closely, which highlights the need to consider the health of both parties while evaluating novel treatments.
KW  - caregiver
KW  - caregiver burden
KW  - mental health
KW  - quality of life
KW  - spinal muscular atrophy
KW  - patient reported outcome measures
Y1  - 2023
U6  - http://nbn-resolving.de/urn/resolver.pl?urn:nbn:de:bvb:20-opus-305048
SN  - 2076-3425
VL  - 13
IS  - 1
ER  -