Esophageal atresia with tracheoesophageal fistula: Suggested mechanism in faulty organogenesis

Crisera, Christopher A.; Connelly, Patrick R.; Marmureanu, Alexander; Colen, Kari L.; Rose, Michael I.; Li, Min; Longaker, Michael T.; Gittes, George K.

doi:10.1016/s0022-3468(99)90258-0

Cited by 59 publications

(41 citation statements)

References 8 publications

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“…In our study, histological assessments of the EA and TEF and control specimens essentially confirmed previously reported observations in an adriamycin rat model [2,3,15]. In addition, Cheng et al [14] found neuropeptide abnormality in the lower esophagus morphological abnormality of the distal esophagus.…”

Section: Discussionsupporting

confidence: 91%

“…The adriamycin rat model produces EA and TEF similar to the most common human anatomic variant allows better understanding pathophysiology of EA and associated anomalies [1,2,15]. In this experimental study we have found several histological and morphological changes in distal esophagus, tracheobronchial tree, and lung parenchyma.…”

Section: Discussionmentioning

confidence: 62%

“…EA and TEF are relatively common congenital anomalies occurring in 1 per 3,000 births [2,3]. Respiratory complications in children with EA are still a common source of morbidity [4].…”

Section: Introductionmentioning

confidence: 99%

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Esophageal, Tracheal and Pulmonary Parenchymal Alterations in Experimental Esophageal Atresia and Tracheoesophageal Fistula

Otcu

Kaya²,

Öztürk³

et al. 2002

Eur Surg Res

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Pulmonary complications are among the most important causes of morbidity and mortality in neonates with esophageal atresia and tracheofistula. We aimed to investigate the possible causes of respiratory complications encountered in esophageal atresia (EA) and tracheoesophageal fistula (TEF) in an experimental model. Sprague-Dawley fetal rats treated with adriamycin were used for the experiment. Time mated pregnant rats were given 1.75 mg/kg of adriamicyn intraperitoneally on days 6–9 of gestation. The fetuses were sacrified on day 21, weighed, and dissected under the surgical microscope. The animals were divided into four groups: (1) control group; (2) saline-injected group; (3) adriamycin-induced EA group, and (4) adriamycin administered but without development of EA. The lungs, esophagus, and trachea were excised and underwent histological examination. The mucosa of distal esophagus was thickened (p < 0.05); the submucosa was thinner (p < 0.05); and the muscular layer was thickened (p < 0.05) in fetuses with EA and TEF. In adriamycin-treated rats, in which EA and TEF developed, tracheal cartilage was loosened and formed into a D or C shape. The cartilage was fragmented into several segments on transverse sections in most fetuses. Alveolar septa were thin in lungs of fetus with EA and TEF (p < 0.05), without any fibrosis or evidence of parenchymal abnormality microscopically. Our findings suggest that respiratory complications may contribute to structural lesions in the trachea and particularly in the distal esophagus but not in the pulmonary parenchyma itself.

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Section: Discussionsupporting

confidence: 91%

Section: Discussionmentioning

confidence: 62%

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Esophageal, Tracheal and Pulmonary Parenchymal Alterations in Experimental Esophageal Atresia and Tracheoesophageal Fistula

Otcu

Kaya²,

Öztürk³

et al. 2002

Eur Surg Res

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“…25 The study was prompted by observations that such human congenital pulmonary malformations as lung hypoplasia, tracheoesophageal fistula, and esophageal atresia may be attributed to defective SHH signaling. [26][27][28][29] Taken together, these studies illustrate how molecular technology may be used to identify the timing of PNEC fate restriction and lineage relationships during morphogenesis. 17 In the immature, developing lung markers of NE differentiation are co-expressed with the epithelial markers before they are restricted to specific PNECs, suggesting a common cellular origin for airway cells.…”

mentioning

confidence: 89%

Functional facets of the pulmonary neuroendocrine system

Linnoila

2006

Laboratory Investigation

178

143

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Pulmonary neuroendocrine cells (PNECs) have been around for 60 years in the scientific literature, although phylogenetically they are ancient. Their traditionally ascribed functions include chemoreception and regulation of lung maturation and growth. There is recent evidence that neuroendocrine (NE) differentiation in the lung is regulated by genes and pathways that are conserved in the development of the nervous system from Drosophila to humans (such as achaete-scute homolog-1), or implicated in the carcinogenesis of the nervous or NE system (such as the retinoblastoma tumor suppressor gene). In addition, complex neural networks are in place to regulate chemosensory and other functions. Even solitary PNECs appear to be innervated. For the first time ever, we have mouse models for lung NE carcinomas, including the most common and virulent small cell lung carcinoma. Moreover, PNECs may be important for inflammatory responses, and pivotal for lung stem cell niches. These discoveries signify an exciting new era for PNECs and are likely to have therapeutic and diagnostic applications.

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“…None of the patients had well-developed lower esophagus. Lack of stimulus from swallowing or different organogenesis of lower esophagus as suggested previously, might be the causative factors [13]. However, none of our patients had anastomotic leak in postoperative period.…”

Section: Discussionmentioning

confidence: 49%

Esophageal Atresia with Tracheo-Esophageal Fistula Presenting Beyond 7 Days

et al. 2017

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Aim: To describe our experience of neonates with esophageal atresia with tracheo-esophageal fistula (EA with TEF) who presented after a week.Design: Retrospective study of the patients of EA with TEF who presented after a week. Study Setting: Department of Pediatric Surgery, Government Medical College Nagpur. Study Duration: Eight years.Materials and Methods: Demographic information, hematological, biochemical and radiological data were obtained from the patients' medical records. The gap between two ends of the esophagus, nature of upper pouch and lower esophagus were noted intra-operatively. Outcome in terms of mortality and surgical complications were noted. In operated group, babies who survived were compared with non-survivors with respect to various preoperative variables.Results: Of 52 patients, 27 babies expired during initial stabilisation period before surgery. The causes of mortality were severe pneumonitis and septicemia. One baby had associated cyanotic heart disease. Twenty-five patients with mean age of 8.28±1.21 days underwent surgery. Nearly two-third of them were male. All of them were born at full-term with mean birth weight of 2.47±0. 12 kg. More than 80% were previously hospitalised and nearly 70% babies were given feeds before present hospitalization. Mean Downe's score for respiratory distress was 5.8±1.49. All patients were positive for septic profile. Associated congenital anomalies were present in ten patients. Intra-operatively, two ends of esophagus were either approximating or have short gap in 24 patients. All patients had well developed, thick and muscular upper oesophageal pouch. Lower esophagus at fistula was thin but dilated in 18 patients while thin and narrowed in 7 patients. However, esophageal anastomosis was possible with ease without any tension in all except one patient. There were 15 deaths in our study (13 due to pneumonitis and 2 during follow up due to aspiration). Three survivors required anti-reflux surgery. Comparison of preoperative variables of survivors and non-survivors showed a significant difference with respect to the variables like feedings, abdominal girth, immature band cells to neutrophil ratio and nature of pharyngeal or endotracheal aspirate.Conclusions: Late presentations in EA with TEF are associated with high mortality but less anastomotic complications after surgery. Preoperative factors like feedings, abdominal distension, immature band cells to neutrophil ratio and bilious pharyngeal or endotracheal aspirate are associated with high mortality.

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Esophageal atresia with tracheoesophageal fistula: Suggested mechanism in faulty organogenesis

Cited by 59 publications

References 8 publications

Esophageal, Tracheal and Pulmonary Parenchymal Alterations in Experimental Esophageal Atresia and Tracheoesophageal Fistula

Esophageal, Tracheal and Pulmonary Parenchymal Alterations in Experimental Esophageal Atresia and Tracheoesophageal Fistula

Functional facets of the pulmonary neuroendocrine system

Esophageal Atresia with Tracheo-Esophageal Fistula Presenting Beyond 7 Days

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