Quigley S J, Linnane B, Connellan S, Ward A, Ryan P
Department of Psychology, University of Limerick, Limerick, Ireland.
Graduate Entry Medical School and Centre for Interventions, Infection, Inflammation and Immunity (4i), University Hospital Limerick, Dooradoyle, Limerick, Ireland.
J Genet Couns. 2018 Jun;27(3):589-596. doi: 10.1007/s10897-017-0150-3. Epub 2017 Sep 26.
Significant gaps have been identified in parental understanding of CF newborn screening and the consequences of carrying an altered CF gene. Seven potential causes of psychosocial adversity arising from false positive newborn screening for CF have been identified. The current study aimed to increase parents understanding of CF, reduce their levels of stress, and investigate psychosocial adversity arising from false-positive screening. This national study was run over one year in the Republic of Ireland. Parents were recruited for the study following a diagnostic sweat test confirming their child carried a single altered CF gene. Parents were randomly assigned into a control and intervention group, with those in the intervention group receiving a carefully designed information pack. All parents took part in semi-structured interviews. Parents (n = 16) who received an information pack had significantly higher CF knowledge scores than parents (n = 16) in the control group. 66% of parents in the control group misunderstood the health implications of carrying an altered CF gene, no parents in the intervention group had the same misunderstanding. There was no significant difference in stress scores between the groups. Parents of infants who had more than one sweat test due to insufficient sweat quantity had higher overall stress percentiles (50%), than parents of infants who had one sweat test (30%), indicating greater parental stress. The combination of written and audio-visual information contained in the information pack successfully increased parents comprehension of CF. The study also evaluates the potential for psychosocial adversity following false positive newborn screening for CF.
已发现父母在对囊性纤维化(CF)新生儿筛查的理解以及携带CF基因变异的后果方面存在重大差距。已确定了因CF新生儿筛查假阳性而产生的心理社会逆境的七个潜在原因。本研究旨在增进父母对CF的了解,降低他们的压力水平,并调查假阳性筛查引起的心理社会逆境。这项全国性研究在爱尔兰共和国进行了一年。在诊断性汗液测试确认孩子携带单一CF基因变异后,招募父母参与研究。父母被随机分为对照组和干预组,干预组的父母会收到精心设计的信息包。所有父母都参加了半结构化访谈。收到信息包的父母(n = 16)的CF知识得分显著高于对照组的父母(n = 16)。对照组中66%的父母误解了携带CF基因变异对健康的影响,干预组中没有父母有同样的误解。两组之间的压力得分没有显著差异。因汗液量不足而进行不止一次汗液测试的婴儿的父母的总体压力百分位数(50%)高于只进行一次汗液测试的婴儿的父母(30%),表明父母压力更大。信息包中包含的书面和视听信息的结合成功提高了父母对CF的理解。该研究还评估了CF新生儿筛查假阳性后心理社会逆境的可能性。