A prospective comparative evaluation of persistent respiratory morbidity in esophageal atresia and congenital diaphragmatic hernia survivors

Extracorporeal membrane oxygenation (ECMO) provides life support in acute reversible cardiorespiratory failure. Assessment of long-term morbidity is essential to confirm survival advantage. This study aimed to assess exercise capacity in the first 12 yrs of life after neonatal ECMO, and to evaluate the effect of primary diagnosis, lung function or perinatal characteristics on exercise capacity.120 children who, as neonates, underwent ECMO performed 191 reliable exercise tests according to the Bruce treadmill protocol at ages 5, 8 and/or 12 yrs between 2001 and 2010. Primary diagnoses were meconium aspiration syndrome (n566), congenital diaphragmatic hernia (n518) and other diagnoses (n536).At ages 5, 8 and 12 yrs, ANOVA resulted in mean¡SE standard deviation score endurance time on the treadmill of -0.5¡0.1, -1.1¡0.1, and -1.5¡0.2, respectively, all significantly less than zero (p,0.001). Exercise capacity declined significantly over time irrespective of the primary diagnosis.Neonates treated with ECMO are at risk of decreased exercise capacity at school age. We therefore propose prolonged follow-up. Proactive advice on sports participation or referral to a physical therapist is recommended, especially when either the parents or the children themselves report impaired exercise capacity.

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“…Recently, GISCHLER et al [24], from our group, evaluated maximal exercise performance in 16 5-yr-old children born …”

Section: Discussionmentioning

confidence: 99%

“…The mean SD-score for endurance time was -0.84 (also significantly different from zero, p50.012) [24]. In a study by PEETSOLD et al [25], 36 out of 53 survivors of CDH who had not received ECMO treatment (mean¡SD age 11.9¡3.5 yrs) reliably performed the Bruce treadmill test.…”

Section: Paediatric Lung Diseasementioning

confidence: 94%

Deterioration of exercise capacity after neonatal extracorporeal membrane oxygenation

Zijp¹,

Gischler²,

Hop

et al. 2011

European Respiratory Journal

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“…As the traditional, mono-specialistic approach to children with congenital anomalies was felt to be inadequate, a multidisciplinary team and a multidisciplinary follow-up programme for these children and their parents were instituted in 1999. 7 …”

Section: Methods Settingmentioning

confidence: 99%

Early developmental assessment of children with major non‐cardiac congenital anomalies predicts development at the age of 5 years

Mazer

Gischler

Zijp

et al. 2010

Develop Med Child Neuro

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MDI Mental Developmental Index PDIPsychomotor Developmental Index SES Socioeconomic status TIS Total impairment score AIM The aim of this study was to evaluate cognitive and motor development in children with major congenital anomalies and the predictability of development at age 5 years.METHOD A prospective, longitudinal follow-up study was undertaken. The Dutch version of the Bayley Scales of Infant Development -Mental Developmental Index (MDI) and Psychomotor Developmental Index (PDI) -were administered at the ages of 6, 12, and 24 months. The Revised Amsterdam Children's Intelligence Test -IQ and the Movement Assessment Battery for ChildrenTotal impairment score (TIS) were used at age 5 years. A total of 117 children participated in the study. After excluding 12 children who had a major chromosomal or syndromal abnormality, the analysis was limited to 105 children (50 females, 55 males). Seven groups of congenital anomalies were distinguished: (1) small intestinal anomalies; (2) abdominal wall defects, comprising gastroschisis and omphalocele; (3) oesophageal atresia; (4) congenital diaphragmatic hernia; (5) Hirschsprung disease; (6) anorectal malformations; and (7) miscellaneous diagnoses. Logistic regression analyses served to determine the ability of MDI and PDI to predict IQ and TIS at age 5 years.RESULTS At age five, 83.7% of 104 children had an IQ of 85 or above and 16.3% an IQ of less than 85. TIS was normal in 71.3% of 87 children, while 17.2% demonstrated a borderline score and 11.5% a definite motor problem. MDI and PDI scores showed equal sensitivity to predict IQ (p=0.004 at 6 and 12mo, p=0.001 at 24mo) and TIS (p<0.001 at 6 and 12mo, p=0.002 at 24mo). MDI and PDI were positively correlated with IQ and TIS; TIS was positively correlated with IQ.INTERPRETATION IQ scores at 5 years of age corresponded to Dutch population scores, but TIS scores differed significantly. Early development of children with major congenital anomalies is predictive of development at 5 years, which can guide individualized follow-up for this vulnerable group of children.About 2 to 3% of neonates in the Netherlands are born with major anatomical congenital anomalies, which are almost always detected shortly after birth. Most congenital anomalies are life-threatening and require early surgical intervention. 1 Examples are intestinal atresias, abdominal wall defects, such as gastroschisis and omphalocele, congenital diaphragmatic hernia, anorectal malformations, and Hirschsprung disease. Presentation may be isolated or as part of a spectrum of anomalies. Advances in surgery and perioperative care have reduced mortality rates (except for congenital diaphragmatic hernia) to approximately 10%. 2 Physical morbidity has become more prevalent, however, and may extend into adulthood. [3][4][5] None of these congenital anomalies has a cerebral component that might be primarily responsible for psychomotor delay. However, moderate to severe psychomotor delay and neurological impairment have been observed in some children with congenita...

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“…52,53)) fits the pattern of less conspicuous lung hypoplasia described in our study. In a parallel investigation of the long-term respiratory morbidity of EA-TEF and congenital diaphragmatic hernia survivors, it was demonstrated that 25% of cases in both groups had forced expiratory volume decreased by −2 z scores and that both had similar degrees of compromise of maximal exercise performance (54). Pulmonary hypoplasia persisting after the neonatal period will be a likely explanation.…”

Section: Lung Hypoplasia In Esophageal Atresiamentioning

confidence: 98%

Lung hypoplasia in rats with esophageal atresia and tracheo–esophageal fistula

et al. 2012

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IntroductIon: survivors of esophageal atresia and tracheo-esophageal fistula (ea-TeF) often suffer chronic respiratory tract disease. ea-TeF results from abnormal emergence of the trachea from the foregut. This study in a rat model tests the hypothesis that primary lung maldevelopment might be a downstream consequence of this defect. results: The lung was hypoplastic in rats with ea-TeF although the histological pattern was normal. Maturation and arteriolar wall thickness were unchanged, but mesenchymal control of airway branching was weakened. This branching was deficient from embryonal day (e13) on in adriamycin-treated explants. dIscussIon: In conclusion, the lungs were hypoplastic in rats with experimental ea-TeF due to defective embryonal airway branching. however, arteriolar wall and respiratory epithelial patterns remained normal. These findings suggest that similarly defective lung development might contribute to chronic respiratory disease in ea-TeF patients. Methods: Pregnant rats received either 1.75 mg/kg i.p. adriamycin or vehicle on e7, e8, and e9. Lungs were recovered at e15, e18, and e2. Lung weight/body weight ratio, total DNa and protein, radial alveolar count, arteriolar wall thickness, lung maturity, and mesenchymal control of airway branching were assessed. e13 lungs were cultured for 72 h and explant airway branching was measured daily. For comparisons, nonparametric tests (*P < 0.05) were used.

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A prospective comparative evaluation of persistent respiratory morbidity in esophageal atresia and congenital diaphragmatic hernia survivors

Cited by 87 publications

References 39 publications

Deterioration of exercise capacity after neonatal extracorporeal membrane oxygenation

Deterioration of exercise capacity after neonatal extracorporeal membrane oxygenation

Early developmental assessment of children with major non‐cardiac congenital anomalies predicts development at the age of 5 years

Lung hypoplasia in rats with esophageal atresia and tracheo–esophageal fistula

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