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足月单胎婴儿在胚胎植入前遗传学诊断(PGD)后的学前年龄的心理社会发展和家庭功能:一项前瞻性病例对照研究和多信息来源方法。

Psychosocial development of full term singletons, born after preimplantation genetic diagnosis (PGD) at preschool age and family functioning: a prospective case-controlled study and multi-informant approach.

机构信息

Department of Developmental and Lifespan Psychology, Vrije Universiteit Brussel (VUB), Pleinlaan 2, 1050 Brussels, Belgium Centre for Medical Genetics, Reproduction and Genetics, Reproduction - Genetics and Regenerative Medicine, Vrije Universiteit Brussel (VUB), UZ Brussel, Laarbeeklaan 101, 1090 Brussel, Belgium

Artesis, Plantijn Hogeschool, Lange Nieuwstraat 101, 2000 Antwerpen, Belgium.

出版信息

Hum Reprod. 2015 May;30(5):1122-36. doi: 10.1093/humrep/dev036. Epub 2015 Mar 6.

Abstract

STUDY QUESTION

Do full term singletons born after preimplantation genetic diagnosis (PGD) differ in their psychosocial functioning from children born after intracytoplasmic sperm injection (ICSI) and spontaneous conceived controls (SC)?

SUMMARY ANSWER

The psychosocial maturation process of 5-6-year-old PGD children is comparable between the three conception groups (PGD, ICSI and SC).

WHAT IS ALREADY KNOWN

In general, a lot of research has been published regarding follow-up of children born after artificial reproductive technologies (ART), which mainly is reassuring. But the ART population itself is marked by broad diversity [IVF, ICSI, gamete donation, preimplantation genetic screening (PGS) or PGD] which complicates comparisons. Some literature concerning the socio-emotional development of PGD/PGS children is available and it suggests a normal maturation process. However, the complex reality of PGD families (e.g. safety of the technique and psychological burden of genetic histories) asks for an exclusive PGD sample with matched control groups and a multi-informant approach.

STUDY DESIGN, SIZE, DURATION: Between April 2011 and May 2013, the psychosocial wellbeing of preschoolers and their families born after PGD was assessed in a prospective case-controlled, matched follow-up study, with a multi-informant approach.

PARTICIPANTS/MATERIALS, SETTING, METHODS: A group of 47 PGD, 50 ICSI and 55 SC 5-6-year-old children participated in a follow-up study performed at the Centre for Medical Genetics of the Universitair Ziekenhuis Brussel (UZ Brussel). Assessments took place in the hospital and in kindergartens. Children performed the Bene-Anthony family relations test (FRT), yielding their perceptions upon family relationships. Parents and teachers completed the child behaviour checklist (CBCL) and Caregiver Teacher Report Form (C-/TRF), respectively. Parental and family functioning were measured by the NEO-FFi, the parenting stress index (PSI), the Greenberger Work-Parenting Investment Questionnaire and the Marlowe-Crowne Social Desirability Scale (MCSDS). Statistical analysis was performed by using analysis of covariance (ANCOVA).

MAIN RESULTS AND THE ROLE OF CHANCE

No differences were detected between the psychosocial development of PGD children and the control groups. Parents did not differ in reporting problem behaviour and they were stricter than teachers. Concerning family functioning the ART parents scored comparable with each other. PGD and ICSI mothers were emotionally more stable [NEO-FFi Neuroticism/emotionality: P = 0.013, η(2) = 0.066; 95% confidence interval (CI) 95% (0.003;0.148)]. They experienced less parental stress in general [PSI, Total stress: P = 0.001, η(2) = 0.102, 95% CI (0.02;0.192)] and on different sublevels opposed to their SC counterparts. Yet ART mothers presented higher ratings on the NEO-FFi Conscientiousness [P = 0.011, η(2) = 0.064; 95% CI (0.003;0.144)] indicating a higher feeling of competence and goal directedness. Mediation analysis confirmed: PGD and ICSI mothers who experienced less family stress were emotionally more stable. A power analysis indicated that a sample with 152 children is sufficient to detect a medium size effect with 80% power using ANCOVA.

LIMITATIONS, REASONS FOR CAUTION: The current sample comprised only Dutch speaking Caucasians, hence conclusions should be drawn cautiously. Future research should include larger groups, prematures, multiples and children from different cultural backgrounds.

WIDER IMPLICATIONS OF THE FINDINGS

This current research is the first to compare PGD preschoolers with matched controls. Concerns about the behavioural effects on the offspring should not inhibit the use of PGD. Furthermore, our findings suggest that on the long run ART procedures might enhance personal resources of women to cope with family stress. These findings are reassuring for women who might feel insecure and anxious during their ART trajectory.

STUDY FUNDING/COMPETING INTERESTS: This research project gained funding from the OZR (a grant by the Research group of the Vrije Universiteit Brussel), the FWO (Fonds Wetenschappelijk Onderzoek) and the Wetenschappelijk Fonds Willy Gepts. The UZ Brussel and the Centre of Medical Genetics received funding from pharmaceutical firms for data collection. UZ Brussel and the Centre for Medical Genetics have received many educational grants for organizing the data collection, from IBSA, Ferring, Organon, Shering-Plough, Merck and Merck Belgium. M.B. has received consultancy and speaker's fees from Organon, Serono Symposia and Merck. The other authors have no competing interests.

TRIAL REGISTRATION NUMBER

not applicable.

摘要

研究问题

与体外受精(IVF)、卵胞浆内单精子注射(ICSI)和自然受孕对照组(SC)相比,通过胚胎植入前遗传学诊断(PGD)出生的足月单胎婴儿在心理社会功能方面是否存在差异?

总结答案

三组受孕方式(PGD、ICSI 和 SC)的 5-6 岁 PGD 儿童的心理社会成熟过程相当。

已知内容

一般来说,已经发表了大量关于人工生殖技术(ART)后儿童随访的研究,这些研究主要是令人安心的。但 ART 人群本身具有广泛的多样性[IVF、ICSI、配子捐赠、胚胎植入前遗传学筛查(PGS)或 PGD],这使得比较变得复杂。一些关于 PGD/PGS 儿童社会情感发展的文献表明,其具有正常的成熟过程。然而,PGD 家庭的复杂现实(例如技术的安全性和遗传史的心理负担)需要一个单独的 PGD 样本,并与匹配的对照组和多信息源方法相结合。

研究设计、规模、持续时间:2011 年 4 月至 2013 年 5 月,采用前瞻性病例对照、匹配随访研究和多信息源方法,评估了 PGD 后学龄前儿童及其家庭的心理社会健康状况。

参与者/材料、设置、方法:47 名 PGD、50 名 ICSI 和 55 名 SC 5-6 岁儿童参加了这项研究。评估在布鲁塞尔大学医院和幼儿园进行。儿童进行了本内-安东尼家庭关系测试(FRT),以评估他们对家庭关系的看法。父母和教师分别填写了儿童行为检查表(CBCL)和 caregiver-teacher 报告表(C-/TRF)。采用 NEO-FFI、育儿压力指数(PSI)、格林伯格工作-父母投资问卷和马罗-克劳恩社会期望量表(MCSDS)测量父母和家庭功能。统计分析采用协方差分析(ANCOVA)。

主要结果和机会的作用

PGD 儿童与对照组的心理社会发展无差异。父母在报告问题行为方面没有差异,而且他们比教师更严格。在家庭功能方面,ART 父母之间的评分相当。PGD 和 ICSI 母亲在情绪上更稳定[NEO-FFI 神经质/情绪性:P=0.013,η(2)=0.066;95%置信区间(95%CI)95%(0.003;0.148)]。与 SC 对照组相比,她们在一般情况下经历的育儿压力较小[PSI,总压力:P=0.001,η(2)=0.102,95%CI(0.02;0.192)]。然而,ART 母亲在 NEO-FFI 尽责性方面的评分较高[P=0.011,η(2)=0.064;95%CI(0.003;0.144)],这表明她们的感觉更有能力和目标导向。中介分析证实:经历较少家庭压力的 PGD 和 ICSI 母亲在情绪上更稳定。一项功效分析表明,使用 ANCOVA 以 80%的功效检测中等大小效应,需要 152 名儿童的样本。

局限性、谨慎的原因:当前的样本仅包括讲荷兰语的白种人,因此结论应谨慎得出。未来的研究应包括更大的群体、早产儿、多胎和来自不同文化背景的儿童。

研究结果的更广泛影响

这是第一项将 PGD 学龄前儿童与匹配对照组进行比较的研究。对后代行为影响的担忧不应抑制 PGD 的使用。此外,我们的研究结果表明,从长远来看,ART 程序可能会增强女性应对家庭压力的个人资源。这些发现对那些在 ART 过程中感到不安和焦虑的女性来说是令人安心的。

研究资金/利益冲突:本研究项目获得了 OZR(布鲁塞尔自由大学研究组的一项赠款)、FWO(科学研究基金)和 Willy Gepts 科学基金的资助。UZ 布鲁塞尔和医学遗传学中心从制药公司获得了数据收集的资金。UZ 布鲁塞尔和医学遗传学中心收到了许多组织数据收集的教育赠款,来自 IBSA、Ferring、Organon、Shering-Plough、Merck 和 Merck Belgium。M.B. 曾担任过 Organon、Serono Symposia 和 Merck 的顾问和演讲嘉宾。其他作者没有利益冲突。

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