title: A review of the social, psychological, and economic burdens experienced by people with spina bifida and their caregivers.
abstract: BACKGROUND
  Despite measures to reduce the incidence of neural tube defects (NTDs), the rate of decline has not been as dramatic as expected. At least 300,000 newborns worldwide are known to be affected by NTDs each year. This comprehensive literature review summarizes the human and economic burden of NTDs to patients and caregivers, with particular focus on spina bifida (SB).
  
  
  METHODS
  PubMed, PsycINFO, and Embase were searched for studies from January 1976 to November 2010 that included clinical terms, such as NTD, and at least one patient-reported outcome or cost term. A conceptual model was also developed.
  
  
  RESULTS
  Areas of peoples' lives affected by SB included physical and role functioning, activities of daily living, bodily pain, vitality, emotional functioning, mental health, self-esteem, self-image, social functioning, relationships, and sexual functioning. Areas of caregivers' lives affected included activities of daily living, work impact, time consumption, parental responsibilities (including responsibilities to other children), confidence, feelings and emotions, mental health, stress, social impact, psychological adjustment, relationships (with SB child, siblings, other family members), social support, coping strategies, and termination decisions. Cost burdens on patients and caregivers also include out-of-pocket costs, lost wages, or household production due to increased morbidity and mortality, transportation and other nonmedical costs.
  
  
  CONCLUSIONS
  This review highlights the need to provide care and support to individuals with SB and their caregivers. Results also emphasize the importance of effective long-term public health campaigns and/or newer strategies to prevent NTDs, such as SB.
authors.affiliation: Adelphi Values Ltd, Grimshaw Lane, Bollington, Cheshire SK10 5JB UK.
authors.forename: Diana
authors.initials: D
authors.lastname: Rofail
authors.affiliation: Adelphi Values Ltd, Grimshaw Lane, Bollington, Cheshire SK10 5JB UK.
authors.forename: Laura
authors.initials: L
authors.lastname: Maguire
authors.affiliation: Bayer Healthcare, Müllerstr. 178, 13353 Berlin, Germany.
authors.forename: Marion
authors.initials: M
authors.lastname: Kissner
authors.affiliation: Bayer Healthcare, Müllerstr. 178, 13353 Berlin, Germany.
authors.forename: Antje
authors.initials: A
authors.lastname: Colligs
authors.affiliation: Adelphi Values Ltd, Grimshaw Lane, Bollington, Cheshire SK10 5JB UK.
authors.forename: Linda
authors.initials: L
authors.lastname: Abetz-Webb
keywords: Caregivers; Developmental disorders; Economic burden; Human burden; Neural tube defects; Patients; Quality of life; Spina bifida