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一项关于遗传咨询师对新生儿血斑筛查呈阳性后接受诊断的父母所起作用的探索性定性研究。

An Explorative Qualitative Study of the Role of a Genetic Counsellor to Parents Receiving a Diagnosis After a Positive Newborn Bloodspot Screening.

作者信息

Sandelowsky Samantha A, McEwen Alison, Russell Jacqui, Boggs Kirsten, Junek Rosie, Ellaway Carolyn, Selvanathan Arthavan, Farrar Michelle A, Bhattacharya Kaustuv

机构信息

Graduate School of Health, University of Technology Sydney, Ultimo, NSW 2007, Australia.

Department of Clinical Genetics, Sydney Children's Hospital, Randwick, NSW 2031, Australia.

出版信息

Int J Neonatal Screen. 2025 Apr 28;11(2):32. doi: 10.3390/ijns11020032.

Abstract

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.

摘要

新生儿血斑筛查(NBS)能够检测出在婴儿期发病的严重可治疗健康状况。新生儿的父母在孩子出生后的几天里较为脆弱,而传达潜在严重诊断这一令人震惊和痛苦消息的最佳方式尚未确定。需要更多数据来确定接触遗传咨询师(GC)是否能改善家庭对NBS所识别的遗传疾病的体验。本研究旨在探讨因脊髓性肌萎缩症(SMA)NBS结果呈阳性且接触了遗传咨询师的父母(GC队列)与被诊断为先天性代谢缺陷(IEM)但未接触遗传咨询师的父母队列(非GC队列)之间的异同。半结构化访谈探讨了接受NBS结果的回顾性经历,包括诊断的影响以及随后对各自遗传诊断的适应情况。采用归纳主题分析法进行组间比较。该研究纳入了7个SMA家庭和5个IEM家庭。确定了四个主题:1. 检测前咨询极少;2. 感觉当地医疗团队知识不足;3. 适应的促成因素;4. 对个人及其家庭的影响。GC队列和非GC队列均报告在检测前期咨询不足,并表示在诊断传达时受到创伤。没有后续GC帮助的家庭表示,由于使用医学术语,对疾病的理解有限,对生殖选择、家庭沟通及后续级联筛查的理解也有所下降。遗传咨询师可以在NBS诊断后支持信息需求及适应过程。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1a29/12101340/9d8a499fa0c4/IJNS-11-00032-g001.jpg

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