title: Providing family-centred care for rare diseases in maternity services: Parent satisfaction and preferences when dysmelia is identified.
abstract: OBJECTIVE
  Dysmelia is usually detected prenatally or postnatally in maternity services. The provision of family-centred care for parents at the time of initial diagnosis is crucial to facilitate decision making, access to appropriate services, and the provision of parental care-giving, but no research has investigated parent experiences or preferences in this population.
  
  
  OBJECTIVE
  The current research aimed to address this by investigating satisfaction with service, occurrence of signposting and preferences in this group.
  
  
  METHODS
  Two online surveys were conducted. In the first survey (n=417), parents reported whether they were offered signposting information and their level of satisfaction with the service they received when initially diagnosed. In the second survey (n=130), a subgroup of participants who completed the first survey reported their preferences for signposting and health service access after diagnosis.
  
  
  RESULTS
  On average, participants were less than satisfied with the service they received and only 27% were offered signposting information. Satisfaction was higher amongst parents who had been offered signposting information. 91% of parents said they would have wanted signposting information and 67% would have wanted access to a support group.
  
  
  CONCLUSIONS
  There is a need to improve the family-centeredness of care when dysmelia is identified. Offering signposting information to relevant third-sector organisations may increase parent satisfaction and address parent preferences. These findings could have implications for parents of children with other rare diseases identified in maternity services.
authors.affiliation: School of Psychology, University of Leeds, Leeds LS29JT, UK(1); Bradford Institute for Health Research, Bradford Royal Infirmary, Bradford BD9 6RJ, UK(2). Electronic address: j.johnson@leeds.ac.uk.
authors.forename: Judith
authors.initials: J
authors.lastname: Johnson
authors.affiliation: European Dysmelia Reference and Information Centre, Axelstorpsvägen 2, Båstad 269 42, Sweden(3). Electronic address: geoff.adams-spink@edric.eu.
authors.forename: Geoff
authors.initials: G
authors.lastname: Adams-Spink
authors.affiliation: European Dysmelia Reference and Information Centre, Axelstorpsvägen 2, Båstad 269 42, Sweden(3). Electronic address: tobias.arndt@edric.eu.
authors.forename: Tobias
authors.initials: T
authors.lastname: Arndt
authors.affiliation: Bradford Institute for Health Research, Bradford Royal Infirmary, Bradford BD9 6RJ, UK(2). Electronic address: del.wijeratne@bthft.nhs.uk.
authors.forename: Dileep
authors.initials: D
authors.lastname: Wijeratne
authors.affiliation: Bradford Institute for Health Research, Bradford Royal Infirmary, Bradford BD9 6RJ, UK(2). Electronic address: jane.heyhoe@bthft.nhs.uk.
authors.forename: Jane
authors.initials: J
authors.lastname: Heyhoe
authors.affiliation: Institute of Psychology, Health & Society, University of Liverpool, Liverpool L69 3GB, UK(4). Electronic address: p.j.taylor@liverpool.ac.uk.
authors.forename: Peter
authors.initials: P
authors.lastname: Taylor
mesh_terms: D000328:Adult; D003657:Decision Making; D003695:Delivery of Health Care; D005260:Female; D019538:Health Care Surveys; D006801:Humans; D007223:Infant; D017880:Limb Deformities, Congenital / Q000175:diagnosis / Q000523:psychology; D008297:Male; D008427:Maternal Health Services; D010290:Parents / Q000523:psychology*; D017060:Patient Satisfaction; D018743:Perinatal Care; D010549:Personal Satisfaction; D035583:Rare Diseases / Q000175:diagnosis / Q000523:psychology*; D012017:Referral and Consultation; D011795:Surveys and Questionnaires
keywords: Dysmelia; Family-centred care; Maternity services; Prenatal care; Rare diseases