Neurodevelopmental outcome in infants with esophageal atresia: risk factors in the first year of life

Abstract

Summary Data on neurodevelopmental outcomes of infants born with esophageal atresia (EA) are still scarce and controversial. The aims of our study were to evaluate motor and cognitive development during the first year of life, in patients operated on of EA and to investigate potential risk factors for motor and cognitive development both at 6 and 12 months. This is an observational prospective longitudinal study in a selected cohort of type C and D EA infants enrolled in our follow-up program from 2009 to 2017. In order to exclude possible confounding factors, the following exclusion criteria were applied: (i) gestational age ≤ 32 weeks and/or birth weight ≤ 1500 g; (ii) genetic syndrome or chromosomal anomaly known to be associated with neurodevelopmental delay; (iii) neurologic disease; (iv) esophageal gap ≥three vertebral bodies. Patients were evaluated at 6 and 12 months of life (corrected age for infants with a gestational age of 32–37 weeks) with the Bayley Scales of Infant and Toddler Development—3rd Edition. In our selected cohort of EA infants, 82 were evaluated at 6 months and 59 were reevaluated at 12 months. Both Motor and Cognitive average scores were within the norm at both time points. However, we report increased number of infants with motor delay with time: 14% at 6 months and 24% at 12 months. Multiple regression analysis for Motor scores at 6 [F(4,74) = 4.363, P = 0.003] and 12 months [F(6,50) = 2.634, P = 0.027] identified (i) low birth weight, (ii) longer hospital stay and (iii) weight < fifth percentile at 1 year as risk factors. Interestingly, average Cognitive scores also increased with time from 85.2% at 6 months and 96.6% at 12 months. Multiple regression models explaining variance of Cognitive scores at 6 [F(4, 73) = 2.458, P = 0.053] and 12 months [F(6, 49) = 1.232, P = 0.306] were nonsignificant. Our selected cohort of EA patients shows, on the average, Motor and Cognitive scores within the norm both at 6 and 12 months. Nevertheless, the percentage of infants with Motor scores below the average increases regardless gestational age. None of clinical and sociodemographic variables taken into consideration was able to predict cognitive development both at 6 and 12 months whereas risk factors for Motor development change during the first year of life. Healthcare providers should pay particular attention to patients with low birth weight, longer hospital stays and weight under fifth percentile at 1 year. Future studies should include long-term outcomes to reveal possible catch up in motor development and/or possible findings in Cognitive scores.

Long-term neurodevelopment in children born with esophageal atresia: a systematic review

“…At 6 months of age, normal neurodevelopment was found in two longitudinal cohort studies: Gischler et al (BSID-I, ages 6, 12, 18, and 24 months, n = 13) 22 and Francesca et al (Bayley-3, ages 6 and 12 months, n = 82). 34 At 12 months of age, both these studies again found normal motor and cognitive functioning. 22 , 34 Francesca et al found a delay with time.…”

“… 34 At 12 months of age, both these studies again found normal motor and cognitive functioning. 22 , 34 Francesca et al found a delay with time. They found a significantly lower median motor score at 12 months, compared with the score at 6 months old ( P = 0.033), but higher cognitive function at 12 months compared with the score at 6 months ( P = 0.000).…”

“… 22 With multivariate regression, Francesca et al found birth weight to be positively associated with motor function at 6 months of age, whereas length of stay and weight at 12 months beneath the 5th percentile were both negatively associated with motor function at 12 months. 34 …”

The Behavioral Problems in 2.5–5 Years Old Children Linked with Former Neonatal/Infantile Surgical Parameters

“…Thus, it seems that a cohort of our patients, who had undergone median/major neonatal/infantile surgery up till 3 months of age for diverse abdominal, urogenital and other congenital non-cardiac abnormalities, has similar long-term behavioral outcome at mean 4.3 years of age as an omni-cultural general population. It is worth mentioning, though, that our studied patient cohort did not include children who had suffered such severe pathologies as esophageal atresia or diaphragmatic hernia, which had been associated with worse outcome in certain neurodevelopmental domains in previous studies [23,24].…”

“…This sample, though, must adequately represent the specific population intended to be studied [40]. We did not include children who had surgeries for esophageal atresia, congenital diaphragmatic hernia, or ear, throat, nose abnormalities, conditions that had been associated with worse long-term outcome previously [23,24,41]. Thus, our studied patients had surgeries for anomalies or diseases that do not directly interfere with respiratory, cardiovascular or central nervous system, and abdominal or urological malformations were predominant.…”