Erik Landfeldt1,2, Josefin Edström3, Filippo Buccella4, Janbernd Kirschner5, Hanns Lochmüller5. 1. Institute of Environmental Medicine, Karolinska Institutet, Stockholm, Sweden. 2. Mapi Group, Stockholm, Sweden. 3. Saco, Stockholm, Sweden. 4. Parent Project Onlus, Rome, Italy. 5. Department of Neuropediatrics and Muscle Disorders, Faculty of Medicine, Medical Center - University of Freiburg, Freiburg, Germany.
Abstract
AIM: To conduct a systematic literature review of caregiver burden in Duchenne muscular dystrophy (DMD). METHOD: We searched Embase, Web of Science, and PubMed for full-text articles reporting results from studies of caregiver burden in DMD. RESULTS: We identified 483 unique publications. Of these, 450 were excluded after title and abstract screening, and 12 after full-text review. A total of 21 articles were included for data synthesis. Results encompassing more than 15 aspects of caregiver burden, investigated through surveys and/or interviews across 15 countries, were identified in the literature. Caregiving in DMD was frequently associated with impaired health-related quality of life, poor sleep quality, reduced family function, depression, pain, stress, sexual dysfunction, and/or lower self-esteem, as well as a considerable impact on work life and productivity. INTERPRETATION: Providing informal care to a patient with DMD can be associated with a substantial burden. Yet, more research is needed to better understand the clinical implications of caregiving in DMD and the relationship between caregiver burden and the progression of the disease. Our data synthesis should be helpful in informing clinical and social support programmes directed to families caring for a patient with DMD. WHAT THIS PAPER ADDS: A substantial body of evidence describes caregiver burden in Duchenne muscular dystrophy. Little is known of the family burden beyond caregivers' self-assessments.
AIM: To conduct a systematic literature review of caregiver burden in Duchenne muscular dystrophy (DMD). METHOD: We searched Embase, Web of Science, and PubMed for full-text articles reporting results from studies of caregiver burden in DMD. RESULTS: We identified 483 unique publications. Of these, 450 were excluded after title and abstract screening, and 12 after full-text review. A total of 21 articles were included for data synthesis. Results encompassing more than 15 aspects of caregiver burden, investigated through surveys and/or interviews across 15 countries, were identified in the literature. Caregiving in DMD was frequently associated with impaired health-related quality of life, poor sleep quality, reduced family function, depression, pain, stress, sexual dysfunction, and/or lower self-esteem, as well as a considerable impact on work life and productivity. INTERPRETATION: Providing informal care to a patient with DMD can be associated with a substantial burden. Yet, more research is needed to better understand the clinical implications of caregiving in DMD and the relationship between caregiver burden and the progression of the disease. Our data synthesis should be helpful in informing clinical and social support programmes directed to families caring for a patient with DMD. WHAT THIS PAPER ADDS: A substantial body of evidence describes caregiver burden in Duchenne muscular dystrophy. Little is known of the family burden beyond caregivers' self-assessments.
Authors: Jamie L Jackson; Christina X Korth; Carine E Leslie; Jennifer Cotto; May Ling Mah; Kan Hor; Linda Cripe; Samiah Al-Zaidy; Eric M Camino; Kathleen Church; Kelly J Lehman; Victoria Shay; Jerry R Mendell Journal: J Child Neurol Date: 2020-10-09 Impact factor: 1.987
Authors: R Quinlivan; B Messer; P Murphy; R Astin; R Mukherjee; J Khan; A Emmanuel; S C Wong; R Kulshresha; T Willis; J Pattni; D Willis; A Morgan; K Savvatis; R Keen; J Bourke; C Marini Bettolo; C Hewamadduma Journal: J Neuromuscul Dis Date: 2021