Longitudinal Follow-up of Bronchial Inflammation, Respiratory Symptoms, and Pulmonary Function in Adolescents after Repair of Esophageal Atresia with Tracheoesophageal Fistula

Malmström, Kristiina; Lohi, Jouko; Lindahl, Harry; Pelkonen, Antti; Kajosaari, Merja; Sarna, Seppo; Malmberg, L. Pekka; Mäkelä, Mika J.

doi:10.1016/j.jpeds.2008.03.034

Cited by 103 publications

(150 citation statements)

References 28 publications

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“…41 Fraction of exhaled NO is known to be associated with eosinophilic airway inflammation. 31,42 Almost all patients in the present study had normal FE NO levels, in line with findings by Malmström et al 34 The FE NO level was increased in only one CDH patient, who showed an atopic constitution. Low or normal FE NO levels have also been described in children with bronchopulmonary dysplasia, 25 which might explain the lower median FE NO in the CDH patients.…”

Section: Discussionsupporting

confidence: 92%

“…32 Less frequent are RTIs, wheezing, and cough; these findings mainly are derived from studies using a cross-sectional design. [33][34][35][36] showed that 41% of adolescents after repair of EA still had respiratory symptoms, and 52% had ever had pneumonia or wheezing. Pneumonia in the first years of life may give rise to mild lung function abnormalities later in life.…”

Section: Discussionmentioning

confidence: 99%

“…Nevertheless, the impact of gastrointestinal problems on RTI in EA patients remains inconclusive so far. 34,37,38 Pulmonary function testing proved problematic. For no more than half of the patients (n = 20), we could interpret FEV 1 before bronchodilation.…”

Section: Discussionmentioning

confidence: 99%

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

Gischler

Zijp

Mazer

et al. 2009

Journal of Pediatric Surgery

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PurposeThe aim of the study was to compare long-term respiratory morbidity in children after repair of esophageal atresia (EA) or congenital diaphragmatic hernia (CDH). Patients and methodsChildren were seen at 6, 12, and 24 months and 5 years within a prospective longitudinal follow-up program in a tertiary children's hospital. Respiratory morbidity and physical condition were evaluated at all moments. At age 5 years, pulmonary function and maximal exercise performance were tested. ResultsIn 3 of 23 atresia patients and 10 of 20 hernia patients, bronchopulmonary dysplasia was developed. Seventeen atresia and 11 hernia patients had recurrent respiratory tract infections mainly in the first years of life. At age 5, 25% of EA and CDH patients measured showed reduced forced expiratory volume in 1 second (z-score < −2). Both atresia and hernia patients showed impaired growth, with catch-up growth at 5 years in patients with EA but not in those with hernia. Maximal exercise performance was significantly below normal for both groups. ConclusionsEsophageal atresia and CDH are associated with equal risk of long-term respiratory morbidity, growth impairment, and disturbed maximal exercise performance. Prospective follow-up of EA patients aimed at identifying respiratory problems other than tracheomalacia should be an integral part of interdisciplinary follow-up programs.

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Section: Discussionsupporting

confidence: 92%

Section: Discussionmentioning

confidence: 99%

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

Gischler

Zijp

Mazer

et al. 2009

Journal of Pediatric Surgery

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“…After repair of OA, approximately one-third of these patients suffer respiratory symptoms in adolescence and adulthood [8][9][10][11][12]. Prevalence of wheeze ranges 26-43% [10,13,14], and that of doctor-diagnosed asthma (DDA) 12-29% [9,10,12,14,15].…”

mentioning

confidence: 99%

Repaired oesophageal atresia: respiratory morbidity and pulmonary function in adults

Sistonen¹,

Malmberg²,

Malmström³

et al. 2010

Eur Respir J

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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“…Recurrent milk inhalation was considered to be a likely explanation (3). Repeated pneumonia occurs in up to 30% of survivors (35), constrictive bronchitis in 40-70% (36,37), obstructive or restrictive respiratory disease in 18-35% (36)(37)(38), and bronchial hyper-reactivity in more than 20% of them (36,37). The possible influence of gastro-esophageal reflux on these symptoms was repeatedly evoked, but other causes were also mentioned (4,36).…”

Section: Lung Hypoplasia In Esophageal Atresiamentioning

confidence: 99%

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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Longitudinal Follow-up of Bronchial Inflammation, Respiratory Symptoms, and Pulmonary Function in Adolescents after Repair of Esophageal Atresia with Tracheoesophageal Fistula

Cited by 103 publications

References 28 publications

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

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

Repaired oesophageal atresia: respiratory morbidity and pulmonary function in adults

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

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