Ten Years of Management of Esophageal Atresia in Benin: State of the Art, Experiences and Needs for Families of Children with Stoma
DOI:
https://doi.org/10.12974/2311-8687.2023.11.01Keywords:
Esophageal atresia, Staged repair, Mortality, Family needs, Low-income countriesAbstract
Background/Purpose: Esophageal atresia (EA) is a congenital defect in the thoracic esophagus associated or not with a tracheoesophageal fistula (TEF). It is associated with high mortality in low-income countries such as Burkina Faso, Madagascar and Ghana. The purpose of this study was to present the recent management of newborns with EA/TEF in Benin and to identify the needs of families whose children have survived.
Method: Over a period of 10 years, 54 newborns with EA/TEF operated on in the 2 largest university hospitals in Benin were included. Two groups were identified. Group A (n=33) included newborns in whom a primary repair had been performed. Group B (n=21) involved newborns who underwent staged repair. Staged repair involved cervical oesophagostomy + gastrostomy (n= 7) or upper oesophageal pouch suction + gastrostomy (n=2) or cervical esophagostomy + stoma of the lower esophageal pouch (n=12). Ten parents from group B were interviewed for the needs survey. The significance level was defined as p < 0.05.
Results: There were 31 male newborns. The median for gestational age was 37 gestational weeks (range: 35 - 38). Polyhydramnios was observed in 4 cases. The mean birth weight was 2365 g. (range: 1000 g - 3500 g). The mean age at diagnosis was 3.48 days (range: 24 hours - 19 days). Surgery was performed at day 5 on average with extremes from day 2 to day 17. The surgery lasted an average of 2h12 min and the overall mortality rate was 74.07%. Mortality rate in group A was 100% and 33.33% in group B. There was a strong statistically significant difference in survival between the two groups. (p=19.10-8). Sepsis was the most common cause of death. Stress, fear and anxiety of losing the child were the difficulties frequently encountered at bedtime and every day. Four parents had to stop their activities to be available to take care of their child. All the parents had expressed the need for psychological support.
Conclusion: Pending an improvement of the technical platform, staged repair remains an essential option for the survival of patients. The creation of a framework for discussion with families authorizing social actions and quality medical support is desired.
References
Oddsberg J. Environmental factors in the etiology of esophageal atresia. J Pediatr Gastroenterol Nutr. 2011; 52: 4-5. https://doi.org/10.1097/MPG.0b013e3182111c00 DOI: https://doi.org/10.1097/MPG.0b013e3182111c00
Depaepe A, Dolk H, Lechat MF. The epidemiology of tracheo-oesophageal fistula and oesophageal atresia in Europe. EUROCAT Working Group. Arch Dis Child. 1993; 68: 743-48. https://doi.org/10.1136/adc.68.6.743 DOI: https://doi.org/10.1136/adc.68.6.743
Sparey C, Jawaheer G, Barrett AM, Robson SC. Esophageal atresia in the Northern Region Congenital Anomaly Survey, 1985-1997: prenatal diagnosis and outcome. Am J Obstet Gynecol. 2000; 182: 427-31. https://doi.org/10.1016/S0002-9378(00)70234-1 DOI: https://doi.org/10.1016/S0002-9378(00)70234-1
British Isles of Congenital Anomaly Registers. Congenital Anomaly Statistics 2012- England and Wales
[En ligne]. December 2014
[cité 17 August 2022]. Available on : http: // www.binocar.org/content/Annual%20report%202012_FINAL _nologo.pdf
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: 227-32. https://doi.org/10.1136/archdischild-2011-300597 DOI: https://doi.org/10.1136/archdischild-2011-300597
Bandre E, Niandolo KA, Wandaogo A, Bankole R, Mobiot ML. Atrésie de l'œsophage : problèmes de prise en charge en Afrique sub-saharienne. Arch Pediatr. 2010; 17(3): 300-1. https://doi.org/10.1016/j.arcped.2009.11.011 DOI: https://doi.org/10.1016/j.arcped.2009.11.011
Bode CO, Ademuyiwa AO. Reverse gastric tube esophageal substitution for staged repair of esophageal atresia and Tracheo-esophageal fistula. Afr J Paediatr Surg. 2014; 11: 366-70. https://doi.org/10.4103/0189-6725.143180 DOI: https://doi.org/10.4103/0189-6725.143180
Leven NL. Congenital atresia of the esophagus with tracheo- esophageal fistula. J Thorac Cardiovasc Surg. 1941; 10: 648-57. https://doi.org/10.1016/S0096-5588(20)32186-3 DOI: https://doi.org/10.1016/S0096-5588(20)32186-3
Ladd WE. The surgical treatment of esophageal atresia and tracheoesophageal fistulas. N Engl J Med. 1944; 230: 62537. https://doi.org/10.1056/NEJM194405252302101 DOI: https://doi.org/10.1056/NEJM194405252302101
Haight C, Towsley H. Congenital atresia of the esophagus with tracheoesophageal fistula: extrapleural ligation of fistula and end-to-end anastomosis of esophageal segments. Surg Gynecol Obstet. 1943; 76: 672-88.
Waterston DJ, Carter RE, Aberdeen E. Esophageal atresia: tracheo-oesophageal fistula. A study of survival in 218 infants. Lancet. 1962; 1: 819-22. https://doi.org/10.1016/S0140-6736(62)91837-8 DOI: https://doi.org/10.1016/S0140-6736(62)91837-8
Spitz L. Esophageal atresia. Orphanet J Rare Dis. 2007; 2: 24. https://doi.org/10.1186/1750-1172-2-24 DOI: https://doi.org/10.1186/1750-1172-2-24
Sfeir R, Bonnard A, Khen-Dunlop N, Auber F, Gelas T, Michaud L, et al. Esophageal atresia: data from a national cohort. J. Pediatr Surg. 2013: 48; 1664-1669 https://doi.org/10.1016/j.jpedsurg.2013.03.075 DOI: https://doi.org/10.1016/j.jpedsurg.2013.03.075
Pini Prato A, Carlucci M, Bagolan P, Gamba PG, Bernardi M, Leva E et al. A cross-sectional nationwide survey on esophageal atresia and tracheoesophageal fistula. J Pediatr Surg. 2015; 50: 1441-56. https://doi.org/10.1016/j.jpedsurg.2015.01.004 DOI: https://doi.org/10.1016/j.jpedsurg.2015.01.004
Sulkowski JP, Cooper JN, Lopez JJ, Jadcherla Y, Cuenot A, Mattei P, et al. Morbidity and mortality in patients with esophageal atresia. Surgery. 2014; 156: 483-91. https://doi.org/10.1016/j.surg.2014.03.016 DOI: https://doi.org/10.1016/j.surg.2014.03.016
Mouafo Tambo FF, Ngo Nonga B, Andze OG, Chiabi A, Mikande Ze J, Ngowe MN, et al. Problématique de la prise en charge de l'atrésie de l'œsophage en pays sous médicalisés. Mali Med. 2010; 25(4): 36-8. https://doi.org/10.4314/racs.v4i9.69958 DOI: https://doi.org/10.4314/racs.v4i9.69958
Ekenze SO, Ajuzieogu OV, Nwankwo PE. Effect of cardia banding and improved anaesthetic care on outcome of oesophageal atresia in a developing country. J Trop Pediatr. 2017; 0: 1-5.
Bouguermouh D, Salem A. Esophageal atresia: a critical review of management at a single center in Algeria. Dis Esophagus. 2014; 28(3): 205-10. https://doi.org/10.1111/dote.12174 DOI: https://doi.org/10.1111/dote.12174
Randolph JG, Tunell WP, Lilly JR. Gastric division in the critically ill infant with esophageal atresia and tracheoesophageal fistula. Surgery. Mars 1968; 63: 496-502.
Tönz M, Köhli S, Kaiser G. Oesophageal atresia: what has changed in the last 3 decades?. Pediatr Surg Int. 2004; 20: 768-72. https://doi.org/10.1007/s00383-004-1139-1 DOI: https://doi.org/10.1007/s00383-004-1139-1
Engum SA, Grosfeld JL, West KW, Rescorla FJ, Scherer LR. Analysis of morbidity and mortality in 227 cases of esophageal atres ia and/or tracheoesophageal fistula over two decades. Arch Surg. 1995; 130: 502-08. https://doi.org/10.1001/archsurg.1995.01430050052008 DOI: https://doi.org/10.1001/archsurg.1995.01430050052008
Louhimo I, Lindal H. Esophageal atresia: primary results of 500 consecutively treated patients. J Pediatr Surg. 1983; 18: 217-29. https://doi.org/10.1016/S0022-3468(83)80089-X DOI: https://doi.org/10.1016/S0022-3468(83)80089-X
Rowe MI, Rowe SA. The last fifty years of neonatal surgical management. Am J Surg. 2000; 180: 345-52. https://doi.org/10.1016/S0002-9610(00)00545-6 DOI: https://doi.org/10.1016/S0002-9610(00)00545-6
Sharma AK, Shekhawat NS, Agrawal LD, Chaturvedi V, Kothari SK, Goel D. Esophageal atresia and tracheo- esophageal fistula: a review of 25 years' experience. Pediatr Surg Int. 2000; 16: 478-82. https://doi.org/10.1007/s003830000393 DOI: https://doi.org/10.1007/s003830000393
Fall M, Mbaye PA, Horace HJ, Wellé IB, Lo FB, Traore MM, et al. Oesophageal atresia: Diagnosis and prognosis in Dakar, Senegal. Afr J Paediatr Surg. 2015; 12: 187-90. https://doi.org/10.4103/0189-6725.170196 DOI: https://doi.org/10.4103/0189-6725.170196
Randriamizao HMR, Rakotondrainibe A, Rahanitriniaina NMP, Rajaonera AT, Andriamanarivo ML. Prise en charge péri-opératoire de l'atrésie de l'œsophage: petits pas non négligeables à Madagascar. Pan Afr Med J. 2017; 27: 9 https://doi.org/10.11604/pamj.2017.27.9.10817 DOI: https://doi.org/10.11604/pamj.2017.27.9.10817
Osei-Nketiah S, Hesse AA, Appeadu-Mensah W, GloverAddy H, Etwire VK, Sarpong P. Management of oesophageal atresia in a developing country: Is primary repair forbidden?. Afr J Paediatr Surg. 2016; 13: 114-9. https://doi.org/10.4103/0189-6725.187801 DOI: https://doi.org/10.4103/0189-6725.187801
Gottrand F, Sfeir R, Coopman S, Deschildre A, Michaud L. Atrésie de l'œsophage: devenir des enfants opérés. Arch Pediatr. 2008; 15: 1837-42. https://doi.org/10.1016/j.arcped.2008.09.027 DOI: https://doi.org/10.1016/j.arcped.2008.09.027
Donoso F, Kassa AM, Gustafson E, Meurling SL, Engstrand H. Outcome and management in infants with esophageal atresia - A single center observational study. J Pediatr Surg 2016; 51: 1421-5. https://doi.org/10.1016/j.jpedsurg.2016.03.010 DOI: https://doi.org/10.1016/j.jpedsurg.2016.03.010
Cassina M, Ruol M, Pertile R, Midrio P, Piffer S, Vicenzi V, et al. Prevalence, Characteristics, and Survival of Children with Esophageal Atresia: A 32-Year Population-Based Study Including 1,417,724 Consecutive Newborns. Birth Defects Res A Clin Mol Teratol. 2016; 106(7): 542-8. https://doi.org/10.1002/bdra.23493 DOI: https://doi.org/10.1002/bdra.23493
Chirdan LB, Uba AF, Pam SD. Intestinal atresia: Management problems in a developing country. Pediatr Surg Int. 2004; 20: 834-7. https://doi.org/10.1007/s00383-004-1152-4 DOI: https://doi.org/10.1007/s00383-004-1152-4
Ademuyiwa AO, Sowande OA, Ijaduola TK, Adejuyigbe O. Determinants of mortality in neonatal intestinal obstruction in Ile Ife, Nigeria. Afr J Paediatr Surg. 2009; 6: 11-3. https://doi.org/10.4103/0189-6725.48568 DOI: https://doi.org/10.4103/0189-6725.48568
Pinheiro PFM, Silva ACS, Pereira RM. Current knowledge on esophageal atresia. World J Gastroenterol. 2012; 18: 3662-72. https://doi.org/10.3748/wjg.v18.i28.3662 DOI: https://doi.org/10.3748/wjg.v18.i28.3662
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: 247-51. https://doi.org/10.1159/000094037 DOI: https://doi.org/10.1159/000094037
Hamza AF. Colonic replacement in cases of esophageal atresia. Semin Pediatr Surg. 2009; 18: 40-3. https://doi.org/10.1053/j.sempedsurg.2008.10.008 DOI: https://doi.org/10.1053/j.sempedsurg.2008.10.008
Rintala RJ, Sistonen S, Pakarinen MP. Outcome of esophageal atresia beyond childhood. Semin Pediatr Surg. 2009; 18: 50-6. https://doi.org/10.1053/j.sempedsurg.2008.10.010 DOI: https://doi.org/10.1053/j.sempedsurg.2008.10.010
Orford J, Cass DT, Glasson MJ. Advances in the treatment of oesophageal atresia over three decades: the 1970s and the 1990s. Pediatr Surg Int. 2004; 20: 402-07. https://doi.org/10.1007/s00383-004-1163-1 DOI: https://doi.org/10.1007/s00383-004-1163-1
Vukadin M, Savic D, Malikovic A, Jovanovic D, Milickovic M, Bosnic S, et al. Analysis of Prognostic Factors and Mortality in Children with Esophageal Atresia. Indian J Pediatr. July 2015; 82(7): 586-90. https://doi.org/10.1007/s12098-015-1730-6 DOI: https://doi.org/10.1007/s12098-015-1730-6
Dave S, Bajpai M, Gupta DK, Agarwala S, Bhatnagar V, Mitra DK. Esophageal atresia and tracheo-esophageal fistula: a review. Indian J Pediatr. 1999; 66(5): 759-72. https://doi.org/10.1007/BF02726269 DOI: https://doi.org/10.1007/BF02726269
Association Française de l'Atrésie de l'Œsophage. Enquête sur les familles AO: synthèse des résultats et analyse
[en ligne]. Octobre 2020
[cite 15 August 2022]. Disponible sur https: //www.fimatho.fr/images/CRACMO/Synthèse_Enquête_adhé rents_20206291.pdf.
Association Française de l'Atrésie de l'Œsophage. EAT, un réseau européen autour de l'atrésie de l'œsophage
[en ligne]. Décembre 2015
[cite 15 August 2022]. Disponible sur https: //afao.asso.fr/nos-actions/sensibiliser/eat/.
Centre de Référence des Affections Chroniques et Malformatives de l'œsophage. Protocole National de Diagnostic et de Soins: Atrésie de l'œsophage
[en ligne]. Décembre 2018
[cité 14 Avril 2022]., Disponible sur https: //www.has-sante.fr/jcms/c_715169/fr/atresie-de-l-oesophage.
Filière des Maladies rares Abdomino-Thoraciques. Accompagner l'atrésie de l'œsophage : retour serein a domicile. Guide pratique à destination des parents[en ligne]. Janvier 2018 [cité 14 Juillet 2022]. Disponible sur https://www.fimatho.fr/images/LIVRET-FICHES- PRATIQUES_version_site_internet_cracmo.pdf.