Eur J Pediatr Surg 2021; 31(03): 226-235
DOI: 10.1055/s-0040-1713663
Original Article

Outcome of Very Low and Low Birth Weight Infants with Esophageal Atresia: Results of the Turkish Esophageal Atresia Registry

1   Department of Pediatric Surgery, Izmir Katip Celebi University, Izmir, Turkey
,
2   Department of Pediatric Surgery, Hacettepe University, Ankara, Turkey
,
3   Department of Pediatric Surgery, Ankara Yildirim Beyazit Universitesi, Ankara, Turkey
,
Binali Firinci
4   Department of Pediatric Surgery, Ataturk University, Erzurum, Turkey
,
Çiğdem U. Durakbaşa
5   Department of Pediatric Surgery, Istanbul Medeniyet University Goztepe Training and Research Hospital, Istanbul, Turkey
,
Zafer Dokumcu
6   Department of Pediatric Surgery, Ege University, Izmir, Turkey
,
Gulnur Gollu
7   Department of Pediatric Surgery, Ankara University, Ankara, Turkey
,
Ibrahim Akkoyun
8   Department of Pediatric Surgery, Konya Education and Research Hospital, Konya, Turkey
,
Dilek Demirel
9   Department of Pediatric Surgery, Ondokuz Mayıs University, Samsun, Turkey
,
Ayşe Karaman
10   Department of Pediatric Surgery, Dr. Sami Ulus Maternity and Children Health and Research Application Center, University of Health Sciences, Ankara, Turkey
,
Ilhan Ciftci
11   Department of Pediatric Surgery, Selçuk University, Konya, Turkey
,
Huseyin Ilhan
12   Department of Pediatric Surgery, Eskisehir Osmangazi University, Eskisehir, Turkey
,
Ayse Parlak
13   Department of Pediatric Surgery, Uludag University, Bursa, Turkey
,
Onder Ozden
14   Department of Pediatric Surgery, Cukurova University, Adana, Turkey
,
Hatice S. Y. Cömert
15   Department of Pediatric Surgery, Karadeniz Technical University, Trabzon, Turkey
,
Akgun Oral
16   Department of Pediatric Surgery, Dr. Behcet Uz Education and Research Hospital, Izmir, Turkey
,
Gonca Tekant
17   Department of Pediatric Surgery, Istanbul University-Cerrahpasa, Istanbul, Istanbul, Turkey
,
Gursu Kiyan
18   Department of Pediatric Surgery, Marmara University, Istanbul, Turkey
,
Başak Erginel
19   Department of Pediatric Surgery, Istanbul Faculty of Medicine, Istanbul University, Istanbul, Turkey
,
Unal Guvenc
20   Department of Pediatric Surgery, Kanuni Sultan Suleyman Education and Research Hospital, Istanbul, Turkey
,
Ali Onur Erdem
21   Department of Pediatric Surgery, Adnan Menderes University, Aydin, Turkey
,
Nazile Erturk
22   Department of Pediatric Surgery, Mugla Sitki Kocman University Ringgold standard institution, Mugla, Turkey
,
Abdullah Yildiz
23   Department of Pediatric Surgery, Sisli Hamidiye Etfal Education and Research Hospital, Istanbul, Turkey
› Author Affiliations
Funding None.

Abstract

Introduction The data of the Turkish Esophageal Atresia Registry (TEAR) was evaluated to define the outcome of very low birth weight (VLBW) and low BW (LWB) infants with esophageal atresia (EA).

Materials and Methods The data registered by 24 centers between 2014 and 2018 were evaluated for demographic features, prenatal findings, associated anomalies, surgical treatment, and outcome. Patients were enrolled in three groups according to their BWs (VLBW <1,500 g), LWB = 1,500–2,500 g), and normal BW (NBW; >2,500 g).

Results Among the 389 cases, there were 37 patients (9.5%) in the VLBW group, 165 patients (42.4%) in the LBW group, and 187 patients (48.1%) in the NBW group. Prenatal diagnosis rates were similar among the three groups (29.7, 34.5, and 24.6%, respectively). The standard primary anastomosis was achieved at a significantly higher rate in NWB cases than in the other groups (p < 0.05). In patients with tracheoesophageal fistula (TEF), patients of the NBW group had significantly higher rates of full oral feedings, when compared with VLBW and LBW cases (p < 0.05). At the end of the first year, when we evaluate all patients, the number of cases with fistula recanalization and esophageal anastomotic strictures (AS) requiring esophageal dilatation was similar among the groups. The weight and height measurements at 6 months and 1 year of age of the survivors were similar in all the groups. The overall mortality rate was significantly higher in the VLBW and LBW groups, when compared with the NBW patients, even in patients with tension-free anastomosis (p < 0.05). The incidence of the associated anomalies was 90.6% in cases with mortality, which was significantly higher than in survivors (59.6%; p < 0.05). According to Spitz's classification, the survival rate was 87.1% in class I, 55.3% in class II, and 16.7% in class III. The most common causes of mortality were associated with cardiovascular diseases, pneumonia, and sepsis.

Conclusion The national data of TEAR demonstrates that the developmental and feeding parameters are better in NBW patients. Although VLBW patients have higher risk of developing fistula canalization than the LBW and NBW groups, long-term complications, such as anastomotic strictures, weight, and height values, after 1 year are similar in both groups. According to our results, associated anomalies and LBWs are still significant risk factors for mortality in cases with EA.



Publication History

Received: 15 January 2020

Accepted: 19 May 2020

Article published online:
06 July 2020

© 2020. Thieme. All rights reserved.

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  • References

  • 1 Pedersen RN, Calzolari E, Husby S, Garne E. EUROCAT Working group. Oesophageal atresia: prevalence, prenatal diagnosis and associated anomalies in 23 European regions. Arch Dis Child 2012; 97 (03) 227-232
  • 2 Oddsberg J, Lu Y, Lagergren J. Aspects of esophageal atresia in a population-based setting: incidence, mortality, and cancer risk. Pediatr Surg Int 2012; 28 (03) 249-257
  • 3 Sfeir R, Bonnard A, Khen-Dunlop N. et al. Esophageal atresia: data from a national cohort. J Pediatr Surg 2013; 48 (08) 1664-1669
  • 4 Leoncini E, Bower C, Nassar N. Oesophageal atresia and tracheo-oesophageal fistula in Western Australia: prevalence and trends. J Paediatr Child Health 2015; 51 (10) 1023-1029
  • 5 Seitz G, Warmann SW, Schaefer J, Poets CF, Fuchs J. Primary repair of esophageal atresia in extremely low birth weight infants: a single-center experience and review of the literature. Biol Neonate 2006; 90 (04) 247-251
  • 6 Petrosyan M, Estrada J, Hunter C. et al. Esophageal atresia/tracheoesophageal fistula in very low-birth-weight neonates: improved outcomes with staged repair. J Pediatr Surg 2009; 44 (12) 2278-2281
  • 7 Zani A, Wolinska J, Cobellis G, Chiu PP, Pierro A. Outcome of esophageal atresia/tracheoesophageal fistula in extremely low birth weight neonates (<1000 grams). Pediatr Surg Int 2016; 32 (01) 83-88
  • 8 Gross RE. The Surgery of Infancy and Childhood. 1st ed. Philadelphia PA: WB Saunders; 1953
  • 9 Spitz L, Kiely EM, Morecroft JA, Drake DP. Oesophageal atresia: at-risk groups for the 1990s. J Pediatr Surg 1994; 29 (06) 723-725
  • 10 Poenaru D, Laberge JM, Neilson IR, Guttman FM. A new prognostic classification for esophageal atresia. Surgery 1993; 113 (04) 426-432
  • 11 Choudhury SR, Ashcraft KW, Sharp RJ, Murphy JP, Snyder CL, Sigalet DL. Survival of patients with esophageal atresia: influence of birth weight, cardiac anomaly, and late respiratory complications. J Pediatr Surg 1999; 34 (01) 70-73
  • 12 Okamoto T, Takamizawa S, Arai H. et al. Esophageal atresia: prognostic classification revisited. Surgery 2009; 145 (06) 675-681
  • 13 Spaggiari E, Faure G, Rousseau V. et al. Performance of prenatal diagnosis in esophageal atresia. Prenat Diagn 2015; 35 (09) 888-893
  • 14 Stoll C, Alembik Y, Dott B, Roth MP. Associated anomalies in cases with esophageal atresia. Am J Med Genet A 2017; 173 (08) 2139-2157
  • 15 Singh A, Bajpai M, Bhatnagar V, Agarwala S, Srinivas M, Sharma N. Effect of number of associated anomalies on outcome in oesophageal atresia with or without tracheoesophageal fistula patient. Afr J Paediatr Surg 2013; 10 (04) 320-322
  • 16 Mee RBB, Beasley SW, Auldist AW. et al. Influence of congenital heart disease on management of oesophageal atresia. Pediatr Surg Int 1993; 7: 90-93
  • 17 Chittmittrapap S, Spitz L, Kiely EM, Brereton RJ. Oesophageal atresia and associated anomalies. Arch Dis Child 1989; 64 (03) 364-368
  • 18 Mili F, Edmonds LD, Khoury MJ, McClearn AB. Prevalence of birth defects among low-birth-weight infants. A population study. Am J Dis Child 1991; 145 (11) 1313-1318
  • 19 Beasley SW, Allen M, Myers N. The effects of Down syndrome and other chromosomal abnormalities on survival and management in oesophageal atresia. Pediatr Surg Int 1997; 12 (08) 550-551
  • 20 Way C, Wayne C, Grandpierre V, Harrison BJ, Travis N, Nasr A. Thoracoscopy vs. thoracotomy for the repair of esophageal atresia and tracheoesophageal fistula: a systematic review and meta-analysis. Pediatr Surg Int 2019; 35 (11) 1167-1184
  • 21 Zani A, Eaton S, Hoellwarth ME. et al. International survey on the management of esophageal atresia. Eur J Pediatr Surg 2014; 24 (01) 3-8
  • 22 Vergouwe FWT, Vlot J, IJsselstijn H. , et al; DCEA Study Group. Risk factors for refractory anastomotic strictures after oesophageal atresia repair: a multicentre study. Arch Dis Child 2019; 104 (02) 152-157
  • 23 Donoso F, Kassa AM, Gustafson E, Meurling S, Lilja HE. Outcome and management in infants with esophageal atresia - A single centre observational study. J Pediatr Surg 2016; 51 (09) 1421-1425
  • 24 Shah R, Varjavandi V, Krishnan U. Predictive factors for complications in children with esophageal atresia and tracheoesophageal fistula. Dis Esophagus 2015; 28 (03) 216-223
  • 25 Allin B, Knight M, Johnson P, Burge D. BAPS-CASS. Outcomes at one-year post anastomosis from a national cohort of infants with oesophageal atresia. PLoS One 2014; 9 (08) e106149
  • 26 Serhal L, Gottrand F, Sfeir R. et al. Anastomotic stricture after surgical repair of esophageal atresia: frequency, risk factors, and efficacy of esophageal bougie dilatations. J Pediatr Surg 2010; 45 (07) 1459-1462
  • 27 Koivusalo AI, Pakarinen MP, Rintala RJ. Modern outcomes of oesophageal atresia: single centre experience over the last twenty years. J Pediatr Surg 2013; 48 (02) 297-303
  • 28 Laín A, Cerdá J, Cañizo A. et al. [Analysis of esophageal strictures secondary to surgical correction of esophageal atresia] (in Spanish). Cir Pediatr 2007; 20 (04) 203-208
  • 29 Okata Y, Maeda K, Bitoh Y. et al. Evaluation of the intraoperative risk factors for esophageal anastomotic complications after primary repair of esophageal atresia with tracheoesophageal fistula. Pediatr Surg Int 2016; 32 (09) 869-873
  • 30 Rayyan M, Embrechts M, Van Veer H. et al. Neonatal factors predictive for respiratory and gastro-intestinal morbidity after esophageal atresia repair. Pediatr Neonatol 2019; 60 (03) 261-269
  • 31 Castilloux J, Noble AJ, Faure C. Risk factors for short- and long-term morbidity in children with esophageal atresia. J Pediatr 2010; 156 (05) 755-760
  • 32 Little DC, Rescorla FJ, Grosfeld JL, West KW, Scherer LR, Engum SA. Long-term analysis of children with esophageal atresia and tracheoesophageal fistula. J Pediatr Surg 2003; 38 (06) 852-856
  • 33 Puntis JWL, Ritson DG, Holden CE, Buick RG. Growth and feeding problems after repair of oesophageal atresia. Arch Dis Child 1990; 65 (01) 84-88
  • 34 Menzies J, Hughes J, Leach S, Belessis Y, Krishnan U. Prevalence of malnutrition and feeding difficulties in children with esophageal atresia. J Pediatr Gastroenterol Nutr 2017; 64 (04) e100-e105
  • 35 Gottrand M, Michaud L, Sfeir R, Gottrand F. Motility, digestive and nutritional problems in esophageal atresia. Paediatr Respir Rev 2016; 19: 28-33
  • 36 Neyzi O, Bundak R, Gökçay G. et al. Reference values for weight, height, head circumference, and body mass index in Turkish children. J Clin Res Pediatr Endocrinol 2015; 7 (04) 280-293
  • 37 Waterston DJ, Carter RE, Aberdeen E. Oesophageal atresia: tracheo-oesophageal fistula. A study of survival in 218 infants. Lancet 1962; 1 (7234): 819-822