Outcome of Oesophageal Atresia Beyond Childhood

Rintala, Risto; Sistonen, Saara J.; Pakarinen, Mikko P.

doi:10.1097/mpg.0b013e318212808e

Cited by 15 publications

(15 citation statements)

References 7 publications

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“…EA is no longer considered to be solely a disease of childhood. With growing numbers of children reaching adulthood and middle age, the long‐term medical morbidity associated with EA is becoming increasingly apparent . Lifelong follow up is therefore now becoming a priority in the care of EA patients…”

Section: Psychological Functioning In Adulthoodmentioning

confidence: 99%

Psychological impact of esophageal atresia: review of the research and clinical evidence

Caplan

2013

Dis Esophagus

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Esophageal atresia (EA) occurs in one out of 2500 to 4500 live births. As the vast majority of infants are now surviving neonatal corrective surgery, the focus has shifted from mortality to morbidity associated with EA. However, little is known about its psychological morbidity. This paper synthesizes research and clinical evidence to highlight the psychological sequelae of EA, including its impact on parents' psychological functioning and its effects on child development from infancy to adulthood. Whether it is discovered at birth or prenatally, EA is a psychologically traumatic event, and parents are at risk for developing traumatic stress reactions following diagnosis. Neonatal surgery and intensive care, risk of complications, associated anomalies, and genetic etiologies multiply risk for parents' acute and post-traumatic stress disorders (PTSD). Parental PTSD has a negative impact on infant and child development through its effects on parenting skills and parent-child interactions. EA children are also at risk for PTSD because of invasive and stressful procedures they undergo during the neonatal period. Consequences of EA can have an important long-term impact on children's psychological and social development. The scant studies pertaining to cognitive functioning suggest that EA does not affect mental development during infancy, but may be associated with deficits as children reach school age. Long-term sequelae are unclear because psychological functioning in adults has not yet been adequately examined. Research and clinical evidence of psychological morbidity associated with EA has implications for clinical practice. Psychological support for parents must begin during the neonatal period and should continue as an integral component of long-term follow up for both children and parents. Support is best provided within the context of a multidisciplinary treatment team that follows patients from birth through childhood and adolescence. Psychological follow up should continue into adulthood, as patients grow up and transition from pediatric to adult health-care settings.

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Section: Psychological Functioning In Adulthoodmentioning

confidence: 99%

Psychological impact of esophageal atresia: review of the research and clinical evidence

Caplan

2013

Dis Esophagus

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“…The prognosis of esophageal atresia (EA) has improved significantly over the past three decades. Because survival rate now exceeds 90% and an increasing number of patients reach adulthood, scientific and clinical attention has shifted to long‐term morbidity and quality of life . At present, data on neurodevelopmental outcome of EA survivors are scarce, controversial, and based on small samples .…”

Section: Introductionmentioning

confidence: 99%

“…Because survival rate now exceeds 90% and an increasing number of patients reach adulthood, scientific and clinical attention has shifted to long-term morbidity and quality of life. 1,2 At present, data on neurodevelopmental outcome of EA survivors are scarce, controversial, and based on small samples. [2][3][4] Therefore, whether or not EA per se is a risk factor for neurodevelopmental outcome is still unclear as is the role played by clinical and sociodemographic variables.…”

Section: Introductionmentioning

confidence: 99%

Short-term neurodevelopmental outcome of babies operated on for low-risk esophageal atresia: a pilot study

et al. 2013

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Data on the neurodevelopmental outcome of esophageal atresia (EA) survivors are scarce, controversial, and based on small samples. This is an observational prospective longitudinal study on a selected cohort of low-risk EA survivors. We considered a low-risk EA survivor a patient with the following characteristics: gestational age >32 weeks, no long gap, no genetic or chromosomic anomaly associated with neurodevelopmental delay, and no further major surgical congenital anomalies. Infants were evaluated with scales derived from the Bayley Scales of Infant and Toddler Development - 3rd Edition at 6 and 12 months, with a score of 100 considered normal for each scale. Analysis of variance was used to assess differences of cognitive and motor development. Linear regression was used to assess the impact of the following clinical and sociodemographic variables: gender, birthweight, gestational age, length of hospital stay, number of surgeries and number of esophageal dilatations during first hospitalization, days of mechanical ventilation, weight at follow up, number of surgeries and esophageal dilatations at follow up, parental age, educational level, and socioeconomic status. Thirty children form the object of the study. The mean (standard deviation [SD]) cognitive scale's score was 93.7 (7.5) and 98.2 (9.6) at 6 and 12 months, respectively (P < 0.05). The mean (SD) motor scale's score was 97.6 (9.3) and 98.0 (12.1) at 6 and 12 months, respectively (P = n.s.). Children with a body weight <5° percentile at 12 months showed a mean (SD) cognitive score significantly lower when compared with those with a body weight >5° percentile: 88.8 (6.3) and 100.5 (8.9), respectively. At 12 months, children with unemployed mothers had a mean (SD) motor score significantly lower when compared with those in the other socioeconomic classes: 87.7 (9.8) and 100.6 (12.4), respectively. In conclusion, parents of babies operated on for low-risk EA can be reassured about neurodevelopmental outcome at least up to 1 year of age. When offering a multidisciplinary follow-up program, underweight patients should deserve particular attention to promote their quality of life and support their global development.

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“…The embryology of OA is incompletely understood [4], and over half these infants have associated anomalies [3,5]. The malformation itself and surgical trauma result in impaired oesophageal motility, a dysmotility predisposing to gastro-oesophageal reflux (GOR) and its complications [6,7]. A strong connection exists between severity of GOR and persistence of respiratory symptoms among OA survivors [4], but respiratory problems become less frequent with time.…”

mentioning

confidence: 99%

Repaired oesophageal atresia: respiratory morbidity and pulmonary function in adults

Sistonen¹,

Malmberg²,

Malmström³

et al. 2010

European Respiratory Journal

117

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Although after oesophageal atresia (OA) repair in infancy, respiratory problems are common, their natural history remains unclear. We assessed morbidity, pulmonary function (PF), and bronchial hyperresponsiveness (BHR) in adults with repaired OA respiratory.588 patients who underwent surgery for OA during 1947-1985 were identified and those 262 who were alive and had their native oesophagus were included. Respiratory symptoms and respiratory symptom-related quality of life (RSRQoL) were assessed by questionnaire and interview, and the patients underwent spirometry, a histamine challenge test, and an exhaled nitric oxide test. For the questionnaires, we added 287 carefully matched general populationderived controls.Among the 101 (58 male) patients, median age 36 yrs (range 22-56 yrs), respiratory morbidity was significantly increased compared to controls. Patients had more respiratory symptoms and infections, as well as asthma and allergies, and more often impaired RSRQoL (p,0.001 for all). PF tests revealed restrictive ventilatory defect in 21 (21%) patients, obstructive ventilatroy defect in 21 (21%) patients, and both in 36 (36%) patients. A total of 41 (41%) had BHR, and in 15 (15%), it was consistent with asthma. The most significant risk factors for restrictive ventilatory defect were thoracotomy-induced rib fusions (OR 3.4, 95% CI 1.3-8.7; p50.01) and oesophageal epithelial metaplasia (OR 3.0, 95% CI 1.0-8.9; p50.05).After repair of OA, respiratory-related morbidity, restrictive ventilatory defect and BHR extended into adulthood. Nearly half the patients had BHR and over half had a restrictive ventilatory defect. Thoracotomy-induced rib fusions and gastro-oesophageal reflux-associated oesophageal epithelial metaplasia were the strongest risk factors for restrictive ventilatory defect.

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Outcome of Oesophageal Atresia Beyond Childhood

Cited by 15 publications

References 7 publications

Psychological impact of esophageal atresia: review of the research and clinical evidence

Psychological impact of esophageal atresia: review of the research and clinical evidence

Short-term neurodevelopmental outcome of babies operated on for low-risk esophageal atresia: a pilot study

Repaired oesophageal atresia: respiratory morbidity and pulmonary function in adults

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