title: A case study of haemoglobinopathy screening in the Netherlands: witnessing the past, lessons for the future.
abstract: OBJECTIVE
  In 2007 neonatal screening (NNS) was expanded to include screening for sickle cell disease (SCD) and beta-thalassaemia. Up until that year no formal recommendations for haemoglobinopathy (carrier) screening existed in the Netherlands. Although it has been subject to debate in the past, preconceptional and prenatal haemoglobinopathy carrier screening are not part of routine healthcare in the Netherlands. This study aimed to explore the decision-making process of the past: why was the introduction of a screening programme for haemoglobinopathy considered to be untimely, and did ethnicity play a role given the history in other countries surrounding the introduction of haemoglobinopathy screening?
  
  
  METHODS
  A witness seminar was organised, inviting key figures to discuss the decision-making process concerning haemoglobinopathy screening in the Netherlands, thereby adding new perspectives on past events. The transcript was content-analysed.
  
  
  RESULTS
  The subject of haemoglobinopathy screening first appeared in the 1970s. As opposed to a long history of neglect of African-American health in the United States, the heritage of the Second World War influenced the decision-making process in the Netherlands. As a consequence, registration of ethnicity surfaced as an impeding factor. However, overall, official Dutch screening policy was restrained regarding reproductive issues caused by fear of eugenics. In the 1990s haemoglobinopathy screening was found to be 'not opportune' due to low prevalence, lack of knowledge and fear of stigmatisation. Currently the registration of ethnicity remains on the political agenda, but still proves to be a sensitive subject.
  
  
  CONCLUSIONS
  Carrier screening in general never appeared high on the policy agenda. Registration of ethnicity remains sensitive caused by the current political climate. Complexities related to carrier screening are a challenge in Dutch healthcare. Whether carrier screening will be considered a valuable complementary strategy in the Netherlands, depends partly on participation of representatives of high-risk groups in policy making.
authors.affiliation: Clinical Genetics, Section Community Genetics, EMGO Institute for Health and Care Research, VU University Medical Center, Amsterdam, The Netherlands. s.jans@vumc.nl
authors.forename: Suze M P J
authors.initials: SM
authors.lastname: Jans
authors.forename: Carla G
authors.initials: CG
authors.lastname: van El
authors.forename: Eddy S
authors.initials: ES
authors.lastname: Houwaart
authors.forename: Marjan J
authors.initials: MJ
authors.lastname: Westerman
authors.forename: Rien J P A
authors.initials: RJ
authors.lastname: Janssens
authors.forename: Antoinette L M
authors.initials: AL
authors.lastname: Lagro-Janssen
authors.forename: Anne Marie C
authors.initials: AM
authors.lastname: Plass
authors.forename: Martina C
authors.initials: MC
authors.lastname: Cornel
mesh_terms: D003657:Decision Making; D005006:Ethnicity; D006291:Health Policy; D006453:Hemoglobinopathies / Q000175:diagnosis* / Q000208:ethnology / Q000235:genetics; D006579:Heterozygote; D006801:Humans; D007231:Infant, Newborn; D015997:Neonatal Screening / Q000639:trends*; D009426:Netherlands / Q000453:epidemiology; D010342:Patient Acceptance of Health Care; D011057:Politics