Department of Epidemiology, Boston University School of Public Health, Boston, Massachusetts, USA.
Birth Defects Res. 2020 Aug;112(14):1074-1084. doi: 10.1002/bdr2.1740. Epub 2020 Jun 23.
Amniotic band syndrome (ABS) includes limb deficiencies accompanied by fibrous strands originating from the amniotic lining. Terminal transverse limb deficiencies (TTLD) appear to be similar but lack fibrous strands. Both are hypothesized to result from vascular disruption. For ABS, limb deficiencies are considered secondary to amnion rupture. We explored an alternative possibility-that TTLD is the primary defect and ABS is secondary.
Using data from the National Birth Defects Prevention Study, we expanded on a previous study. We examined smoking, alcohol, and medications categorized by indicated vasoactivity as markers of vascular disruption. Logistic regression models with Firth's penalized likelihood were used to estimate adjusted odds ratios (aORs) and 95% confidence intervals (CIs).
Use of bronchodilators and aspirin appeared to increase the risk of ABS, while decongestants and nonaspirin NSAIDs increased the risk of TTLD. The risk of ABS was markedly increased in cases reporting combinations of vasoactive exposures, particularly alcohol and aspirin (aOR 3.7, 95% CI 1.6, 7.8), and alcohol and bronchodilators (aOR 3.4, 95% CI 1.4, 7.5). Increased risk of TTLD due to combinations of vasoactive exposures was only observed for smoking and decongestants (aOR 2.3, 95% CI 1.4, 3.6).
Exposures associated with increased risk of ABS had no apparent association with TTLD, supporting previous evidence that these may be distinct phenotypes. ABS appears to be associated with combined exposures with vasodilation properties, such as alcohol and bronchodilators, while increased risk of TTLD may be associated with smoking and decongestants, both vasoconstrictive exposures.
羊膜带综合征(ABS)包括由羊膜衬里起源的纤维束伴发的肢体缺陷。末端横断肢体缺陷(TTLD)似乎相似,但缺乏纤维束。两者都被假设是由血管破坏引起的。对于 ABS,肢体缺陷被认为是羊膜破裂的继发结果。我们探讨了一种替代可能性,即 TTLD 是主要缺陷,而 ABS 是继发缺陷。
我们利用国家出生缺陷预防研究的数据,扩展了之前的研究。我们检查了按血管活性分类的吸烟、饮酒和药物,将其作为血管破坏的标志物。使用带有 Firth 惩罚似然的逻辑回归模型来估计调整后的优势比(aOR)和 95%置信区间(CI)。
使用支气管扩张剂和阿司匹林似乎增加了 ABS 的风险,而减充血剂和非阿司匹林非甾体抗炎药(NSAIDs)增加了 TTLD 的风险。报告有血管活性暴露组合的病例中,ABS 的风险明显增加,尤其是酒精和阿司匹林(aOR 3.7,95%CI 1.6,7.8),以及酒精和支气管扩张剂(aOR 3.4,95%CI 1.4,7.5)。仅观察到吸烟和减充血剂的血管活性暴露组合会增加 TTLD 的风险(aOR 2.3,95%CI 1.4,3.6)。
与 ABS 风险增加相关的暴露与 TTLD 没有明显关联,这支持了之前的证据,表明这些可能是不同的表型。ABS 似乎与具有血管扩张特性的联合暴露有关,如酒精和支气管扩张剂,而 TTLD 风险增加可能与吸烟和减充血剂有关,这两种暴露均为血管收缩剂。