Parental Depression and Anxiety Associated with Newborn Bloodspot Screening for Rare and Variable-Onset Disorders.
Parental Depression and Anxiety Associated with Newborn Bloodspot Screening for Rare and Variable-Onset Disorders.
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DOI：
10.3390/ijns8040059
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发表时间：
2022-11-10
影响因子：
3.5
通讯作者：
中科院分区：
文献类型：
--
作者：
筛查新生儿大量疾病的能力，包括许多具有可变表型的疾病，引发了关于扩大新生儿血斑筛查（NBS）对父母的心理社会影响的争论。本研究比较了具有一系列NBS/诊断经验的儿童父母的心理结果，特别关注溶酶体贮积症（LSDs）和X连锁肾上腺脑白质营养不良（X-ALD）作为具有复杂表现的代表性疾病。2019年完成了一项包括六个领域的在线横断面调查，调查对象是2013年至2018年期间至少有一个孩子出生的父母志愿者样本。在分析阶段，父母根据孩子的罕见疾病和诊断方法分为四组。压力和抑郁的估计使用二分措施的抑郁分量表的医院焦虑和抑郁量表和父母压力量表。Logistic回归模型估计父母组和压力/抑郁之间的关系，控制人口统计学变量（美国地区，收入，教育，主要生活事件，与孩子的关系，孩子的数量，父母年龄和种族/民族）。174名父母被纳入本分析。临床诊断为LSD或X-ALD的儿童的父母与通过NBS确定的患有相同疾病的儿童的父母相比，可能具有更高的抑郁症几率（OR：6.06，95%CI：1.64-24.96），控制协变量。虽然在父母压力方面观察到类似的模式（OR：2.85，95%CI：0.82-10.37），但这并没有达到统计学显著性。从伦理上扩展NBS和基因组测序需要了解早期发现复杂疾病对家庭的影响。这些初步发现令人放心，并可能随着国家统计局的扩大而产生影响。由于我们的样本量很小，很难将这些发现推广到所有家庭。这些初步趋势需要在更大和更多样化的人群中进行进一步研究。
The ability to screen newborns for a larger number of disorders, including many with variable phenotypes, is prompting debate regarding the psychosocial impact of expanded newborn bloodspot screening (NBS) on parents. This study compares psychological outcomes of parents of children with a range of NBS/diagnostic experiences, with a particular focus on lysosomal storage disorders (LSDs) and X-linked adrenoleukodystrophy (X-ALD) as representative disorders with complex presentations. An online cross-sectional survey with six domains was completed in 2019 by a volunteer sample of parents with at least one child born between 2013 and 2018. Parents were classified in the analysis stage into four groups based on their child’s rare disorder and means of diagnosis. Stress and depression were estimated using dichotomous measures of the depression subscale of the Hospital Anxiety and Depression Scale and the Parental Stress Scale. Logistic regression models were estimated for the relationship between the parent group and stress/depression, controlling for demographic variables (region of the US, income, education, major life events, relationship to the child, number of children, parent age, and race/ethnicity). One hundred seventy-four parents were included in this analysis. Parents of children with an LSD or X-ALD diagnosis clinically may have higher odds of depression (OR: 6.06, 95% CI: 1.64–24.96) compared to parents of children with the same disorders identified through NBS, controlling for covariates. Although a similar pattern was observed for parental stress (OR: 2.85, 95% CI: 0.82–10.37), this did not reach statistical significance. Ethically expanding NBS and genome sequencing require an understanding of the impacts of early detection for complex disorders on families. These initial findings are reassuring, and may have implications as NBS expands. Given our small sample size, it is difficult to generalize these findings to all families. These preliminary trends warrant further investigation in larger and more diverse populations.
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影响因子：
2.2
作者：
Shapiro, Danielle N.;Stewart, Abigail J.
通讯作者：
Stewart, Abigail J.
影响因子：
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作者：
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通讯作者：
Fanos J
影响因子：
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作者：
Bailey DB Jr;Porter KA;Andrews SM;Raspa M;Gwaltney AY;Peay HL
通讯作者：
Peay HL
影响因子：
4.7
作者：
Norton, Sam;Cosco, Theodore;Sacker, Amanda
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Sacker, Amanda