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新生儿筛查确诊的遗传性代谢疾病患儿及其父母的心理社会和经济负担

Parental and child's psychosocial and financial burden living with an inherited metabolic disease identified by newborn screening.

作者信息

Schnabel-Besson Elena, Garbade Sven F, Gleich Florian, Grünert Sarah C, Krämer Johannes, Thimm Eva, Hennermann Julia B, Freisinger Peter, Burgard Peter, Gramer Gwendolyn, Morath Marina A, Tuncel A Tunç, Keßler Svenja, Hoffmann Georg F, Kölker Stefan, Mütze Ulrike

机构信息

Department of Pediatrics I, Division of Pediatric Neurology and Metabolic Medicine, Heidelberg University, Medical Faculty Heidelberg, Heidelberg, Germany.

Department of General Pediatrics, Adolescent Medicine and Neonatology, Medical Center - University of Freiburg, Faculty of Medicine, Freiburg, Germany.

出版信息

J Inherit Metab Dis. 2025 Jan;48(1):e12784. doi: 10.1002/jimd.12784. Epub 2024 Aug 27.

Abstract

Newborn screening (NBS) is one of the most effective measures of secondary prevention. While the benefit of NBS on the clinical long-term outcomes of children with inherited metabolic diseases (IMD) has been demonstrated, the potential burden of families living with an early diagnosed and treated child with an IMD has not been thoroughly investigated. The aim of this longitudinal questionnaire-based study on 369 families living with a child with an IMD was to investigate the psychosocial and financial burden following a true-positive NBS. The reported psychosocial burden differed between children and their parents, and was associated with the child's age, diagnosis, and treatment. At younger ages, parent-reported burden was higher for the parents than for the individual child, while it increased for children and decreased for parents as the child grew older. Furthermore, psychosocial burden increased if the child required a strict dietary treatment and was at risk of metabolic decompensation. Regardless of diagnosis and treatment, the developmental delay of their child independently increased the parental psychosocial burden. Financial burden was reported by 24% of all families, and was higher in low-income families and in families whose children required dietary treatment. In conclusion, a substantial psychosocial and financial burden was revealed for children and their families after true-positive NBS. Since this burden is likely to have a negative impact on the long-term individual health benefits of NBS, this study underlines the importance of regularly assessing the psychosocial and financial needs of these families.

摘要

新生儿筛查(NBS)是二级预防最有效的措施之一。虽然新生儿筛查对患有遗传性代谢疾病(IMD)儿童的临床长期预后的益处已得到证实,但对于早期诊断和治疗患有IMD儿童的家庭的潜在负担尚未进行全面调查。这项基于问卷的纵向研究针对369个患有IMD儿童的家庭,旨在调查NBS结果为真阳性后的心理社会和经济负担。报告显示,儿童及其父母的心理社会负担存在差异,且与儿童的年龄、诊断和治疗有关。在儿童较小的时候,父母报告的负担对父母来说高于对儿童个体,而随着儿童年龄增长,儿童的负担增加,父母的负担减轻。此外,如果儿童需要严格的饮食治疗且有代谢失代偿风险,心理社会负担会增加。无论诊断和治疗情况如何,孩子的发育迟缓都会独立增加父母的心理社会负担。24%的家庭报告有经济负担,低收入家庭以及孩子需要饮食治疗的家庭经济负担更高。总之,NBS结果为真阳性后,儿童及其家庭显示出相当大的心理社会和经济负担。由于这种负担可能会对NBS给个人带来的长期健康益处产生负面影响,本研究强调了定期评估这些家庭心理社会和经济需求的重要性。

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