我们发现ART儿童Beckwith-Wiedemann综合征的校正比值比(AOR)为2.84[95%CI:1.34-6.01],而Prader-Willi综合征的风险,在ART后受孕的儿童中,Silver-Russell综合征或Angelman综合征没有增加。
早期研究,他们中的大多数都很小,提出了ART和印记障碍之间的关联。
这是一项基于二项注册的队列研究。确定了丹麦(n=45393,1994年至2014年出生)和芬兰(n=29244,1990年至2014年出生)的所有接受ART的儿童。这两个国家在同一时期出生的全部背景人口被纳入对照。计算了ART儿童与自然受孕(NC)儿童相比的印记障碍的几率。ART儿童的中位随访时间为8年零9个月,NC儿童的中位随访时间为11年零9个月。
来自丹麦和芬兰的国家卫生登记处,我们确定了所有诊断为Prader-Willi综合征的儿童(n=143),Silver-Russell综合征(n=69),Beckwith-Wiedemann综合征(n=105)和Angelman综合征(n=72)分别出生于1994/1990年和2014年。
我们确定了总共388名被诊断患有印记障碍的儿童;其中16名是在ART后受孕的。ART儿童与NC儿童相比,四种印迹障碍的总体AOR为1.35[95%CI:0.80-2.29],但是由于八名ART儿童被诊断出患有Beckwith-Wiedemann综合征,这种特异性印迹障碍的AOR为2.84[95%CI:1.34-6.01]。ART后受孕的儿童Beckwith-Wiedemann综合征的绝对风险仍然很低:10万新生儿中的10.7。Prader-Willi综合征的风险,在ART后受孕的儿童中,Silver-Russell综合征和Angelman综合征没有增加。
印记障碍是罕见的事件,我们的结果是基于少数患有印记障碍的ART儿童。病因很复杂,只有部分澄清,和临床诊断受到广泛的表型谱的挑战。
在现有的研究中,关于ART后受孕儿童印记障碍风险的结果不明确.这项研究补充说,ART儿童的印记障碍风险很小,可能仅限于Beckwith-Wiedemann综合征。
这项工作得到了北欧试验联盟的支持:这是一个由北欧部长理事会和NordForsk共同资助的试点项目(资助号:71450),北欧妇产科联合会(赠款编号:NF13041,NF15058,NF16026和NF17043)和Interreg厄勒海峡-卡特加特-斯卡格拉克欧洲区域发展基金(ReproUnion项目)。作者与这项工作没有利益冲突。
不适用。
Is the risk of imprinting disorders increased in children conceived after ART?
We found an adjusted odds ratio (AOR) of 2.84 [95% CI: 1.34-6.01] for Beckwith-Wiedemann syndrome in ART children, while the risk of Prader-Willi syndrome, Silver-Russell syndrome or Angelman syndrome was not increased in children conceived after ART.
Earlier studies, most of them small, have suggested an association between ART and imprinting disorders.
This was a binational register-based cohort
study. All children conceived by ART in Denmark (n = 45 393, born between 1994 and 2014) and in Finland (n = 29 244, born between 1990 and 2014) were identified. The full background populations born during the same time periods in the two countries were included as controls. Odds ratios of imprinting disorders in ART children compared with naturally conceived (NC) children were calculated. The median follow-up time was 8 years and 9 months for ART children and 11 years and 9 months for NC children.
From the national health registries in Denmark and Finland, we identified all children diagnosed with Prader-Willi syndrome (n = 143), Silver-Russell syndrome (n = 69), Beckwith-Wiedemann syndrome (n = 105) and Angelman syndrome (n = 72) born between 1994/1990 and 2014, respectively.
We identified a total of 388 children diagnosed with imprinting disorders; 16 of these were conceived after ART. The overall AOR for the four imprinting disorders in ART children compared with NC children was 1.35 [95% CI: 0.80-2.29], but since eight ART children were diagnosed with Beckwith-Wiedemann syndrome, the AOR for this specific imprinting disorder was 2.84 [95% CI: 1.34-6.01]. The absolute risk of Beckwith-Wiedemann syndrome in children conceived after ART was still low: 10.7 out of 100 000 newborns. The risks of Prader-Willi syndrome, Silver-Russell syndrome and Angelman syndrome were not increased in children conceived after ART.
Imprinting disorders are rare events and our results are based on few ART children with imprinting disorders. The aetiology is complex and only partly clarified, and the clinical diagnoses are challenged by a broad phenotypic spectrum.
In the existing studies, results on the risk of imprinting disorders in children conceived after ART are ambiguous. This
study adds that the risk of imprinting disorders in ART children is very small and perhaps restricted to Beckwith-Wiedemann syndrome.
This work was supported by the Nordic
Trial Alliance: a pilot project jointly funded by the Nordic Council of Ministers and NordForsk (grant number: 71450), the Nordic Federation of Obstetrics and Gynecology (grant numbers: NF13041, NF15058, NF16026 and NF17043) and the Interreg Öresund-Kattegat-Skagerak European Regional Development Fund (ReproUnion project). The authors have no conflicts of interest related to this work.
N/A.