通过新生儿筛查发现的遗传性代谢疾病给父母和孩子带来的社会心理和经济负担。

IF 4.2 2区 医学 Q1 ENDOCRINOLOGY & METABOLISM
Elena Schnabel-Besson, Sven F Garbade, Florian Gleich, Sarah C Grünert, Johannes Krämer, Eva Thimm, Julia B Hennermann, Peter Freisinger, Peter Burgard, Gwendolyn Gramer, Marina A Morath, A Tunç Tuncel, Svenja Keßler, Georg F Hoffmann, Stefan Kölker, Ulrike Mütze
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引用次数: 0

摘要

新生儿筛查(NBS)是最有效的二级预防措施之一。虽然新生儿筛查对遗传代谢病(IMD)患儿的临床长期预后的益处已经得到证实,但对于与早期诊断和治疗的遗传代谢病患儿生活在一起的家庭的潜在负担还没有进行深入调查。这项针对 369 个 IMD 患儿家庭的纵向问卷调查研究旨在调查 NBS 真阳性后的社会心理和经济负担。儿童及其父母所报告的社会心理负担各不相同,并且与儿童的年龄、诊断和治疗有关。在较小的年龄段,父母报告的负担高于儿童个人的负担,而随着儿童年龄的增长,儿童的负担增加,父母的负担减少。此外,如果患儿需要严格的饮食治疗并面临代谢失调的风险,其社会心理负担也会增加。无论诊断和治疗方法如何,孩子的发育迟缓都会增加父母的社会心理负担。24%的家庭报告了经济负担,低收入家庭和孩子需要饮食治疗的家庭的经济负担更重。总之,真阳性 NBS 给儿童及其家庭带来了巨大的社会心理和经济负担。由于这种负担可能会对 NBS 为个人健康带来的长期益处产生负面影响,因此本研究强调了定期评估这些家庭的社会心理和经济需求的重要性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Parental and child's psychosocial and financial burden living with an inherited metabolic disease identified by newborn screening.

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.

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来源期刊
Journal of Inherited Metabolic Disease
Journal of Inherited Metabolic Disease 医学-内分泌学与代谢
CiteScore
9.50
自引率
7.10%
发文量
117
审稿时长
4-8 weeks
期刊介绍: The Journal of Inherited Metabolic Disease (JIMD) is the official journal of the Society for the Study of Inborn Errors of Metabolism (SSIEM). By enhancing communication between workers in the field throughout the world, the JIMD aims to improve the management and understanding of inherited metabolic disorders. It publishes results of original research and new or important observations pertaining to any aspect of inherited metabolic disease in humans and higher animals. This includes clinical (medical, dental and veterinary), biochemical, genetic (including cytogenetic, molecular and population genetic), experimental (including cell biological), methodological, theoretical, epidemiological, ethical and counselling aspects. The JIMD also reviews important new developments or controversial issues relating to metabolic disorders and publishes reviews and short reports arising from the Society''s annual symposia. A distinction is made between peer-reviewed scientific material that is selected because of its significance for other professionals in the field and non-peer- reviewed material that aims to be important, controversial, interesting or entertaining (“Extras”).
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