在新生儿血斑筛查阳性后,遗传咨询师对父母接受诊断的作用的探索性质的研究。

IF 4 Q1 GENETICS & HEREDITY
Samantha A Sandelowsky, Alison McEwen, Jacqui Russell, Kirsten Boggs, Rosie Junek, Carolyn Ellaway, Arthavan Selvanathan, Michelle A Farrar, Kaustuv Bhattacharya
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引用次数: 0

摘要

新生儿血斑筛查(NBS)可以发现在婴儿期发病的严重可治疗的健康状况。新生儿的父母在出生后的日子里是很脆弱的,而传达潜在严重诊断的令人震惊和痛苦的消息的最佳方式尚未确定。需要更多的数据来确定获得遗传咨询师(GC)是否能改善国家统计局确定的遗传疾病家庭的经历。本研究旨在探讨NBS结果为脊髓性肌萎缩症(SMA)阳性并进入GC (GC队列)的父母与被诊断为先天性代谢错误(IEM)但未进入GC(非GC队列)的父母的异同。半结构化访谈探讨了接受NBS结果的回顾性经验,包括诊断意义和随后对各自遗传诊断的适应。分组比较采用归纳主题性分析。7个SMA家庭和5个IEM家庭被纳入研究。确定了四个主题:1。最低限度的考试前咨询;2. 缺乏对当地医疗团队的了解;3. 适应的有利因素;4. 对个人和家庭的影响。GC组和非GC组均报告在测试前咨询不足,并描述在诊断交付时感到创伤。由于使用医学术语,没有后续GC输入的家庭对疾病的了解有限,对生殖选择、家庭沟通和随后的级联筛查的了解也有所减少。GCs可支持NBS诊断后的信息需求和适应。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
An Explorative Qualitative Study of the Role of a Genetic Counsellor to Parents Receiving a Diagnosis After a Positive Newborn Bloodspot Screening.

Newborn Bloodspot Screening (NBS) can detect severe treatable health conditions with onset during infancy. The parents of a newborn baby are vulnerable in the days after birth, and the optimal way to deliver the shocking and distressing news of a potential serious diagnosis is yet to be defined. More data are needed to determine whether access to a genetic counsellor (GC) improves families' experiences with genetic conditions identified by NBS. This study aimed to explore the similarities and differences for parents who received a positive NBS result for Spinal Muscular Atrophy (SMA) and received access to a GC (GC cohort), to a cohort of parents who received a diagnosis for inborn errors of metabolism (IEM) and did not have access to a GC (non-GC cohort). Semi-structured interviews explored the retrospective experiences of receiving the NBS result, including diagnosis implications and subsequent adaptation to respective genetic diagnoses. Inductive thematic analysis was used from group comparison. 7 SMA families and 5 IEM families were included in the study. Four themes were identified: 1. minimal pre-test counselling; 2. perceived lack of local healthcare team knowledge; 3. enabling factors for adaptation; 4. implications for both individuals and their families. Both the GC and non-GC cohorts reported insufficient counselling in the pre-test period and described feeling traumatised at the time of the diagnosis delivery. Families without subsequent GC input described limited understanding of the disease due to the use of medicalized terms, as well as a decreased understanding of reproductive options, familial communication and subsequent cascade screening. GCs can support information needs and adaptation following a NBS diagnosis.

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来源期刊
International Journal of Neonatal Screening
International Journal of Neonatal Screening Medicine-Pediatrics, Perinatology and Child Health
CiteScore
6.70
自引率
20.00%
发文量
56
审稿时长
11 weeks
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