目的:学龄前植入前遗传学诊断(PGD)儿童的认知和精神运动发育是否与ICSI和自发性受孕(SC)后出生的儿童不同?
结论:PGD学龄前儿童的认知发育与ICSI和SC后出生的儿童相当,但ICSI和SC组的运动发育不同。
方法:与49名ICSI和48名SC儿童相比,对PGD后出生的5至6岁单身人士(n=47)的认知能力和运动技能进行了评估。病例控制,2011年4月至2013年5月的匹配随访研究。
方法:PGD单身人士,使用韦克斯勒学龄前和初级智力量表(WPSSI-III-NL)和运动ABC(MABC)检查了ICSI和SC学龄前儿童。WPSSI-III-NL显示了全智商的分数,言语智商和表现智商。MABC产生总分,包括平衡测量的分数,灵巧和球技。由于胚胎活检是PGD和ICSI程序之间唯一的技术区别,ICSI儿童作为对照。这些孩子是在接受纵向随访的ZiekenhuisBrussel大学(UZBrussel)辅助生殖技术(ART)后受孕的荷兰语儿童群体的一部分。SC儿童充当第二对照组,与肥沃的PGD样本相似,与ICSI组相反。SC小组是通过各种媒体的公告招募的。孩子们的年龄是匹配的,性别,母亲的出生顺序和受教育程度。对ART组进行的评估尽可能是盲目的。使用协方差分析(ANCOVA)和偏eta平方(η(2))分析数据,用作效应大小的度量。
结果:PGD患者的总体认知发展与对照组无差异[P=0.647,η(2)=0.006;95%置信区间(CI)(0,0.043)]。言语和表现智力的部分智商得分显示出类似的结果。基于总分和分量表的运动发育分析确实表明三个受孕组之间存在显着差异[P=0.033,η(2)=0.050,95%CI(0,0.124)]。事后分析表明,ICSI和SC儿童的表现之间存在显着差异。平衡容量[P=0.004,η(2)=0.079,95%CI(0.025,0.163)]及其事后分析产生了等效结果。PGD单缸电机容量,然而,与其他两个概念组没有区别。
结论:鉴于我们仅评估PGD后出生的白人单身人士,从我们的结果中得出更一般的推论时,需要谨慎行事。小样本大小可能是一个限制。先验功率分析,然而,显示,每组至少需要52名儿童使用ANCOVA检测到中等效应和80%功率。最初,我们的样本符合此阈值,但我们必须排除6个案例,以消除异常值和由于缺少数据。
结论:对胚胎活检后出生的儿童进行长期随访,在这种情况下对于PGD,需要确认这些儿童的发育仍与ICSI和SC儿童相当。我们的发现确实支持了PGD技术的安全性,并将使患有遗传性遗传疾病的患者放心,他们未来的后代受孕于PGD。
背景:这项研究的资金来自OZR(布鲁塞尔自由大学研究小组),FWO(FondsWetenschappelijkOnderzoek)和WetenschappelijkFondsWillyGepts。UZBrussel和医学遗传学中心从制药公司获得了用于数据收集的资金。UZBrussel和医学遗传学中心收到了许多教育补助金,用于组织数据收集,来自IBSA,套圈,Organon,Shering-Plough,默克公司和比利时默克公司。M.B.已从Organon收到咨询和演讲者的费用,塞罗诺研讨会和默克。
OBJECTIVE: Do preschool preimplantation genetic diagnosis (
PGD) children differ in their cognitive and psychomotor development from children born after ICSI and spontaneous conception (SC)?
CONCLUSIONS: The cognitive development of
PGD pre-schoolers was comparable to children born after ICSI and SC but motor development differed between ICSI and SC groups.
METHODS: The cognitive abilities and motor skills of 5- to 6-year-old singletons born after PGD (n = 47) were assessed in comparison with 49 ICSI and 48 SC children in a prospective,
case-controlled, matched follow-up study between April 2011 and May 2013.
METHODS: PGD singletons, ICSI and SC children of preschool age were examined with the Wechsler Preschool and Primary Scale of Intelligence (WPSSI-III-NL) and the Movement ABC (M ABC). The WPSSI-III-NL revealed scores for Full IQ, Verbal IQ and Performance IQ. The M ABC yields a total score and comprising scores for measurements of balance, dexterity and ball skills. Since embryo biopsy is the only technical difference between the PGD and ICSI procedures, ICSI children were included as controls. These children were part of a Dutch-speaking cohort of children conceived after assisted reproduction technology (ART) at the Universitair Ziekenhuis Brussel (UZ Brussel) who received longitudinal follow-up. The SC children acted as a second control group similar to the fertile PGD sample and in contrast to the ICSI group. The SC group was recruited through announcements in a variety of media. The children were matched for age, gender, birth order and educational level of the mother. The assessments carried out for the ART groups were blinded whenever possible. The data were analysed using analysis of covariance (ANCOVA) and partial eta squared (η(2)), which was used as a measurement of effect size.
RESULTS: The overall cognitive development of PGD singletons did not differ from controls [P = 0.647, η(2) = 0.006; 95% confidence interval (CI) (0, 0.043)]. The partial IQ scores for Verbal and Performance intelligence revealed similar results. Analysis of motor development based on the total score as well as subscales did indicate a significant difference between the three conception groups [P = 0.033, η(2) = 0.050, 95% CI (0, 0.124)]. Post hoc analysis indicated that the significant difference was situated between performances of ICSI and SC children. Balance capacities [P = 0.004, η(2) = 0.079, 95% CI (0.025, 0.163)] and its post hoc analysis yielded equivalent results. Motor capacities of PGD singletons, however, did not differ from any of the two other conception groups.
CONCLUSIONS: Given that we only assessed Caucasian singletons born after PGD, caution is required when drawing more general inferences from our results. The small sample size may be a limitation. A priori power analysis, however, revealed that at least 52 children per group were needed to detect a medium effect and 80% power using ANCOVA. Originally our sample met this threshold but we had to exclude six cases in order to remove outliers and due to missing data.
CONCLUSIONS: Long-term follow-up of children born after embryo biopsy, in this
case for
PGD, is needed to confirm that the development of these children remains comparable to ICSI and SC children. Our findings do support the safety of the PGD technique and will reassure patients with hereditary genetic diseases regarding the health of their future offspring conceived with
PGD.
BACKGROUND: Funding for this study was obtained from the OZR (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.