title: The impact of informal care for patients with Pompe disease: an application of the CarerQol instrument.
abstract: BACKGROUND
  Patients with Pompe disease, a rare progressive neuromuscular disorder, receive a considerable amount of informal care. In this study, we examined the impact of providing informal care to patients with Pompe disease.
  
  
  METHODS
  Caregivers were administered various instruments, which measured the (impact of) informal care in the context of Pompe disease. Patients' quality of life and use of a wheelchair and respiratory support were used to investigate the impact of disease severity on the burden and well-being of caregivers.
  
  
  RESULTS
  Of all Dutch patients with Pompe disease, 88 indicated to receive informal care, of which 67 (76%; 67 caregivers) participated in this study. On average, caregivers provided 17.7 hours of informal care per week. Higher disease burden was associated with more hours of informal care. Caregivers experienced burden due to caregiving. Half of the informal caregivers reported mental health problems and problems with daily activities due to providing informal care. Physical health problems occurred in 40% of informal caregivers. Caregiver burden was higher for patients with a lower quality of life and for wheelchair dependent patients. Burden was not associated with respiratory support. Caregivers reported deriving personal fulfillment from caregiving and, on average, would become unhappier if someone else were to take over their care activities.
  
  
  CONCLUSIONS
  The provision of informal care causes burden to caregivers. However, caregivers also value caring for their loved ones themselves. The study may help physicians and policy makers to design measures to support informal caregivers.
authors: Institute for Medical Technology Assessment, Department of Health Policy & Management, Erasmus University Rotterdam, Burgemeester Oudlaan 50, P.O. Box 1738, 3000 DR Rotterdam, The Netherlands; Center for Lysosomal and Metabolic Diseases, Erasmus Medical Center, Dr. Molewaterplein 60, 3015 GJ, Rotterdam, The Netherlands. Electronic address: kanters@bmg.eur.nl.
authors: Tim A
authors: TA
authors: Kanters
authors: Ans T
authors: AT
authors: van der Ploeg
authors: Werner B F
authors: WB
authors: Brouwer
authors: Leona
authors: L
authors: Hakkaart
mesh_terms: D000293:Adolescent; D017028:Caregivers / Q000523:psychology; D002648:Child; D005260:Female; D006009:Glycogen Storage Disease Type II / Q000628:therapy*; D006801:Humans; D008297:Male; D005791:Patient Care; D011788:Quality of Life; D012720:Severity of Illness Index; D012944:Social Support; D013997:Time Factors
keywords: Caregiver burden; CarerQol instrument; Informal care; Pompe disease