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一项定性研究:母亲们在新生儿筛查后其孩子迟发性庞贝病诊断的经历

A Qualitative Study: Mothers' Experiences of Their Child's Late-Onset Pompe Disease Diagnosis Following Newborn Screening.

作者信息

Crossen Kaylee, Berry Lisa, Myers Melanie F, Leslie Nancy, Goueli Cecilia

机构信息

Division of Human Genetics, Cincinnati Children's Hospital Medical Center, 3333 Burnet Ave, Cincinnati, OH 45229, USA.

College of Medicine, University of Cincinnati, 3230 Eden Ave, Cincinnati, OH 45267, USA.

出版信息

Int J Neonatal Screen. 2022 Jul 19;8(3):43. doi: 10.3390/ijns8030043.

Abstract

Pompe disease was added to the United States recommended uniform screening panel in 2015 to avoid diagnostic delay and implement prompt treatment, specifically for those with infantile-onset Pompe disease (IOPD). However, most newborns with abnormal newborn screening (NBS) for Pompe disease have late-onset Pompe disease (LOPD). An early diagnosis of LOPD raises the question of when symptoms will arise which is challenging for parents, patients, and providers managing an LOPD diagnosis. This study aimed to characterize mothers' experiences of their child's LOPD diagnosis and medical monitoring. A qualitative descriptive approach was chosen to gain an in-depth understanding of parental experiences. Eight mothers were interviewed about their experiences with positive NBS and diagnosis, experiences with living with the diagnosis, and experiences with medical monitoring. Interview transcripts were analyzed through conventional content analysis. Negative emotions like fear were more frequent with communication of NBS results. Participants expressed uncertainty surrounding age of symptom onset and the future. The medical monitoring experience increased worry but participants expressed that being vigilant with management reassured them. Parental emotions shifted to thankfulness and reassurance with time and education. These findings can provide guidance to providers about the psychosocial implications of receiving positive NBS results and an LOPD diagnosis.

摘要

2015年,庞贝病被纳入美国推荐的统一筛查项目,以避免诊断延误并及时进行治疗,特别是针对婴儿型庞贝病(IOPD)患者。然而,大多数庞贝病新生儿筛查(NBS)异常的新生儿患的是晚发型庞贝病(LOPD)。LOPD的早期诊断引发了症状何时会出现的问题,这对家长、患者以及负责管理LOPD诊断的医护人员来说都具有挑战性。本研究旨在描述母亲们对孩子LOPD诊断及医学监测的经历。我们采用定性描述方法以深入了解家长的经历。对八位母亲进行了访谈,内容包括她们对NBS结果呈阳性及诊断的经历、带着诊断生活的经历以及医学监测的经历。通过常规内容分析法对访谈记录进行了分析。在告知NBS结果时,恐惧等负面情绪更为常见。参与者对症状出现的年龄和未来表示不确定。医学监测经历增加了担忧,但参与者表示对管理保持警惕让他们安心。随着时间推移和接受教育,家长的情绪转变为感激和安心。这些发现可为医护人员提供有关收到NBS结果呈阳性及LOPD诊断的社会心理影响的指导。

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Newborn screening for Pompe disease: impact on families.新生儿庞贝病筛查:对家庭的影响。
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本文引用的文献

3
Newborn Screening for Pompe Disease.庞贝病新生儿筛查
Int J Neonatal Screen. 2020 Apr 5;6(2):31. doi: 10.3390/ijns6020031. eCollection 2020 Jun.
7
Newborn screening for Pompe disease: impact on families.新生儿庞贝病筛查:对家庭的影响。
J Inherit Metab Dis. 2018 Nov;41(6):1189-1203. doi: 10.1007/s10545-018-0159-2. Epub 2018 Mar 28.
10
Highlighting intrafamilial clinical heterogeneity in late-onset Pompe disease.强调晚发型庞贝病的家族内临床异质性。
Mol Genet Metab Rep. 2013 Dec 28;1:2-4. doi: 10.1016/j.ymgmr.2013.10.002. eCollection 2014.

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