The Journal of Pediatrics ( IF 5.1 ) Pub Date : 2021-09-07 , DOI: 10.1016/j.jpeds.2021.09.002 Patricia J C Knijnenburg 1 , Marjolijn S Spruijt 1 , Lisette Jansen 2 , Monique Rijken 1 , Ratna N G B Tan 1 , Femke Slaghekke 3 , Johanna M Middeldorp 3 , Enrico Lopriore 1 , Jeanine M M van Klink 2
Objective
To investigate the neurodevelopmental outcome at age 2 and 5 years in survivors of twin–twin transfusion syndrome (TTTS) treated with fetoscopic laser surgery and born premature and/or small for gestational age.
Study design
At 2 and 5 years of age, standardized neurologic, motor, and cognitive assessments were performed by a neonatologist, a pediatric physical therapist, and a psychologist. Behavior was assessed using a validated questionnaire completed by parents.
Results
Neurodevelopmental assessment at both time points was available for 73 survivors of TTTS. Mild to moderate neurodevelopmental impairment (NDI) was detected in 34% of survivors (25 of 73) at 5 years, compared with 25% (18 of 73) at 2 years (P = .178). Severe NDI was observed in 12% (9 of 73) at 5 years and in 3% (2 of 73) at 2 years (P = .035). Mean cognitive score was lower at the 5-year follow-up (90.7 ± 12.3 vs 95.6 ± 13.1 at 2 years; P = .001), and more children were diagnosed with mild cognitive impairment at 5 years (29% vs 11% at 2 years; P = .007). When comparing individual outcomes at both time points, 35% (25 of 71) moved from a normal outcome or mild to moderate impairment at 2 years toward more severe impairment at 5 years.
Conclusions
A high rate of mild to moderate cognitive impairment and severe NDI at age 5 years was not identified at age 2 years. Our data highlight the importance of longitudinal follow-up of survivors of TTTS beyond age 2 years and emphasize the precautions that should be taken when diagnosing an absence of impairment before school age.
中文翻译:
双胞胎输血综合征早产幸存者的神经发育轨迹:从出生到 5 岁
客观的
调查接受胎儿镜激光手术治疗的早产和/或小于胎龄的双胞胎输血综合征 (TTTS) 幸存者 2 岁和 5 岁时的神经发育结果。
学习规划
在 2 岁和 5 岁时,由新生儿科医生、儿科物理治疗师和心理学家进行标准化的神经、运动和认知评估。使用由父母填写的经过验证的问卷评估行为。
结果
73 名 TTTS 幸存者可在两个时间点进行神经发育评估。5 年时,34% 的幸存者(73 人中的 25 人)检测到轻度至中度神经发育障碍 (NDI),而 2 年时这一比例为 25%(73 人中的 18 人)(P = .178)。5 年时 12%(73 人中的 9 人)和 2 年时 3%(73 人中的 2 人)观察到严重 NDI(P = .035)。5 年随访时的平均认知评分较低(90.7 ± 12.3 vs 2 年的 95.6 ± 13.1;P = .001),更多儿童在 5 年被诊断为轻度认知障碍(29% vs 11% 2 年;P = .007)。在比较两个时间点的个体结果时,35%(71 人中的 25 人)在 2 年时从正常结果或轻度至中度损伤转向 5 年时更严重的损伤。
结论
2 岁时未发现 5 岁时轻度至中度认知障碍和严重 NDI 的高发生率。我们的数据强调了对 2 岁以上 TTTS 幸存者进行纵向随访的重要性,并强调了在学龄前诊断无损伤时应采取的预防措施。