Effect of cardia banding and improved anaesthetic care on outcome of oesophageal atresia in a developing country

Effect of cardia banding and improved anaesthetic care on outcome of oesophageal atresia in a... Abstract We comparatively analysed cases of oesophageal atresia (OA) managed in Enugu, south-eastern Nigeria from October 2010 to September 2015 to evaluate our short-term outcome with management following incorporation of temporary cardia banding to gastrostomy for late presenting cases and improved anaesthesia in 2013. Overall, 19 cases were analysed. The clinical parameters did not differ in the cases managed before (Group A) and after (Group B) these introductions. Four (21.1%) cases had primary repair (2 per group), six (31.6%) had delayed primary repair after treatment of pneumonitis (Group A 5; Group B 1) and nine (47.3%) had delayed primary repair after gastrostomy (Group A 4; Group B 5). Anaesthesia-related mortality dropped from 53.8 to 7.7% and survival improved from 9.1 to 62.5% following the introductions. Despite persisting barriers to care, outcome of OA in our setting may improve with better anaesthesia and incorporation of temporary cardia banding to gastrostomy. oesophageal atresia, anaesthesia, cardia banding, outcome, developing country INTRODUCTION Oesophageal atresia (OA) is a rare malformation with a prevalence of 2.43 per 10 000 births [1]. Refinements in surgical techniques, anaesthesia and neonatal intensive care have improved outcome [1–5]. Studies indicate that management of OA in most high-income countries (HICs) results in significantly higher survival compared with the outcome from most low- and middle-income countries (LMICs) [6, 7]. Factors reported to contribute to the poorer outcome of OA in the latter include late diagnosis with its attendant problems, and inadequate perioperative and anaesthetic care [6–9, 10]. Temporary gastric banding has been previously used in cases of premature babies with oesophageal atresia with tracheo-oesophageal fistula (OA-TEF) and severe respiratory distress [11, 12]. In 2013, we incorporated temporary cardia banding to gastrostomy in OA cases with complications of late presentation, before delayed primary repair. In the temporary banding, a purse-string suture (non-absorbable) is applied to the cardia of the stomach (avoiding the vagus nerve and other structures in the oesophagogastric junction) during gastrostomy procedure. The suture is removed during definitive repair of OA-TEF. In addition to this modification of procedure, one of our anaesthesiologists underwent additional training on neonatal anaesthesia. This study is a preliminary report of our experience with the management OA following improved anaesthesia care and incorporation of cardia banding. MATERIALS AND METHODS We retrospectively analysed the cases of OA managed from October 2010 to September 2015 at the University of Nigeria Teaching Hospital, and Mother of Christ Specialist Hospital, Enugu. Health research and ethics committee gave approval for the study (# NHREC/05/01/2008B-FWA00002458-1RB00002323). For the study, we collected the following data from the case notes, theatre records and discharge summaries: age at presentation and age at operation; gestational age at birth; birth weight, weight on presentation and weight at operation; sex; and findings on evaluation, type of atresia, operative procedures, outcome and follow-up. We compared the cases managed before September 2013 (Group A) with those managed from October 2013 (Group B) following improved anaesthesia care and incorporation of cardia banding during gastrostomy. We used Statistical Package for Social Sciences (SPSS 21.0 version, SPSS Inc., Chicago, Ill) for data entry and analysis. Results were expressed as percentages, or mean. Data were analysed by chi-square test with the significance level set at p < 0.05. RESULTS Overall, there were 19 patients (11 in Group A; 8 in Group B). In total, 11 (57.9%) cases were female and 8 (42.1%) were male. Their mean age at presentation was 8.3 ± 7 days (range 2–26 days). All the cases had proximal atresia and distal tracheaoesophageal fistula, and were delivered outside the two hospitals. The Patients’ characteristics are summarized in Table 1. Table 1 Summary of the patients’ characteristics in the two groups Parameter Group A Group B Median birth weight (kg) 2.6 kg (IQRa 2.4–3.0) 2.5 kg (IQR 2.4–2.8) Term gestational age 10/11 (91%) 8/8 (100%) Associated malformations 4/11 (36.4%) 3/8 (37.5%) Pneumonitis on presentation 9/11 (81.8%) 6/8 (75%) Median age at presentation 7 days (IQR 4–14) 10.5 days (IQR 2.5–15.8) Parameter Group A Group B Median birth weight (kg) 2.6 kg (IQRa 2.4–3.0) 2.5 kg (IQR 2.4–2.8) Term gestational age 10/11 (91%) 8/8 (100%) Associated malformations 4/11 (36.4%) 3/8 (37.5%) Pneumonitis on presentation 9/11 (81.8%) 6/8 (75%) Median age at presentation 7 days (IQR 4–14) 10.5 days (IQR 2.5–15.8) a IQR, interquartile range. Table 1 Summary of the patients’ characteristics in the two groups Parameter Group A Group B Median birth weight (kg) 2.6 kg (IQRa 2.4–3.0) 2.5 kg (IQR 2.4–2.8) Term gestational age 10/11 (91%) 8/8 (100%) Associated malformations 4/11 (36.4%) 3/8 (37.5%) Pneumonitis on presentation 9/11 (81.8%) 6/8 (75%) Median age at presentation 7 days (IQR 4–14) 10.5 days (IQR 2.5–15.8) Parameter Group A Group B Median birth weight (kg) 2.6 kg (IQRa 2.4–3.0) 2.5 kg (IQR 2.4–2.8) Term gestational age 10/11 (91%) 8/8 (100%) Associated malformations 4/11 (36.4%) 3/8 (37.5%) Pneumonitis on presentation 9/11 (81.8%) 6/8 (75%) Median age at presentation 7 days (IQR 4–14) 10.5 days (IQR 2.5–15.8) a IQR, interquartile range. Treatment The overall mean age at definitive treatment was 22.6 days (range 3–50 days). A total of four (21%) patients had primary repair and comprised two patients from each group. Six (31.6%) cases (Group A 5; Group B 1) had delayed primary repair after treatment of pneumonitis. The remaining nine (47.4%) patients had initial gastrostomy at a median age of 18 days (range 12–28 days). Of these cases, four patients (Group A) had Stamm gastrostomy, while five cases (Group B) had temporary cardia banding in addition to Stamm gastrostomy. After a period of 4–6 weeks, these cases had delayed primary repair. The average weight gain following gastrostomy in Group A patients was 0.4 kg, and Group B patients 0.9 kg. Outcome A total of eight (42.1%) patients developed postoperative complications, which includes surgical wound infection (n =  5; Group A 3; Group B 2) and anastomotic leak (n = 3; Group A 2; Group B 1). For anaesthesia, in Group A, four of the babies could not wake up from anaesthesia, and three of those who woke up had repeated apnoea in the absence of identifiable chest pathology, while in Group B, only one of the patients had postoperative apnoea. Overall, six patients (31.6%) survived. In Group A, 1 of 11 children (9%) survived, while in Group B, 5 of 8 children (62.5%) survived (1 of 11 in Group A; 5 of 8 in Group B; p = 0.024). The distribution of the survived cases is shown in Table 2 and the distribution and causes of mortality in the two groups are shown in Table 3. The surviving patients have been followed up for an average of 28 months (range 12–58 months). Only one of the patients (16.7%) developed oesophageal stricture, which responded to dilatation. Table 2 The distribution of the survived cases according to the definitive treatment in the two groups of patients with OA Survival Definitive treatment n Group A Group B p-value Primary repair 4 1/2 (50%) 1/2 (50%) 0.833 Delayed primary repair after treatment of pneumonitis 6 0/5 (0%) 1/1 (100%) 0.166 Delayed primary repair after gastrostomy 9 0/4 (0%) 3/5 (60%) 0.119 Total 19 1/11 (9.1%) 5/8 (62.5%) 0.024* Survival Definitive treatment n Group A Group B p-value Primary repair 4 1/2 (50%) 1/2 (50%) 0.833 Delayed primary repair after treatment of pneumonitis 6 0/5 (0%) 1/1 (100%) 0.166 Delayed primary repair after gastrostomy 9 0/4 (0%) 3/5 (60%) 0.119 Total 19 1/11 (9.1%) 5/8 (62.5%) 0.024* * p<0.05 (Significant). Table 2 The distribution of the survived cases according to the definitive treatment in the two groups of patients with OA Survival Definitive treatment n Group A Group B p-value Primary repair 4 1/2 (50%) 1/2 (50%) 0.833 Delayed primary repair after treatment of pneumonitis 6 0/5 (0%) 1/1 (100%) 0.166 Delayed primary repair after gastrostomy 9 0/4 (0%) 3/5 (60%) 0.119 Total 19 1/11 (9.1%) 5/8 (62.5%) 0.024* Survival Definitive treatment n Group A Group B p-value Primary repair 4 1/2 (50%) 1/2 (50%) 0.833 Delayed primary repair after treatment of pneumonitis 6 0/5 (0%) 1/1 (100%) 0.166 Delayed primary repair after gastrostomy 9 0/4 (0%) 3/5 (60%) 0.119 Total 19 1/11 (9.1%) 5/8 (62.5%) 0.024* * p<0.05 (Significant). Table 3 Distribution and causes of mortality following management of OA in the two groups of patients Groups Mortality Causes of morta1lity Uncontrolled sepsis Anaesthesia-related complications Group A 10/11 (90.9%) 3 7 Group B 3/8 (37.5%) 2 1 Groups Mortality Causes of morta1lity Uncontrolled sepsis Anaesthesia-related complications Group A 10/11 (90.9%) 3 7 Group B 3/8 (37.5%) 2 1 Table 3 Distribution and causes of mortality following management of OA in the two groups of patients Groups Mortality Causes of morta1lity Uncontrolled sepsis Anaesthesia-related complications Group A 10/11 (90.9%) 3 7 Group B 3/8 (37.5%) 2 1 Groups Mortality Causes of morta1lity Uncontrolled sepsis Anaesthesia-related complications Group A 10/11 (90.9%) 3 7 Group B 3/8 (37.5%) 2 1 DISCUSSION Given the reported global incidence of OA [1, 13, 14], the small number of cases in the present report may not indicate the actual incidence of OA in our setting. Apparently, some of these cases may not be captured in the tertiary referral centres [10, 15, 16]. The treatment of OA depends on the type of OA, the weight and state of the patient on presentation and the presence and severity of associated anomalies [1–5, 17]. In the present report, these parameters were similar in the two groups of patients, and these cases would have been better managed by primary repair. However, because of delayed presentation, majority of our cases presented with pneumonitis, undernutrition, sepsis or a combination of these. As a result, a substantial number of them required resuscitation and stabilization before definitive treatment. Similar management has been reported in some other developing countries [9, 10, 18]. The lack of total parenteral nutrition in our setting compounds the challenges and demands that we find a way to administer enteral nutrition. Gastrostomy has been used in cases of pure OA, and in OA-TEF that will require staged treatment [19]. In the latter, there is accompanying thoracotomy to ligate the fistula. The use of gastrostomy for feeding may be fraught with significant respiratory morbidity. The incorporation of temporary cardia banding though reported to be useful in extremely premature cases of OA with severe respiratory distress [11, 12] may be valuable in our setting as a way to reduce gastric aspiration and improve nutrition. With improvement in nutrition and weight gain, and reduction in airway contamination, these cases with complications of late presentation may be suitable for delayed primary repair. Anaesthetic management of OA has been shown to significantly affect the outcome of treatment [2, 4, 17]. Reports from HICs indicate that anaesthesia challenges have been systematically addressed [1, 20], while in some developing countries [8–10], there is report of persistence of inadequate trained personnel and lack of facilities. Lessons from HICs point to advantages of training in neonatal anaesthesia to sufficiently understand the peculiarities of these cases and adequately manage them. The marked reduction of mortality from anaesthesia and respiratory complications following improved anaesthetic management in the present report may attest to that. The overall outcome of OA management in the present report is poor compared with the >90% survival reported in most HICs [1–5, 14, 21, 22]. Although there was a significant improvement of outcome with better anaesthesia, improved nutrition and reduced airway contamination with cardia banding compared with earlier cases managed in the setting [23], the outcome still fell short of the current results from more developed countries where current mortality is limited to cases with associated life-threatening anomalies [1–3]. The absence of these anomalies in our cases and the high mortality in the present report indicate that there are inherent deficiencies in our management of OA that need to be addressed. In the first place, delayed presentation and late intervention are major problems in our setting and have also been reported in some other LMICs [7–10]. Addressing the delays may require improvement of antenatal and perinatal cares, and concerted efforts to educate the healthcare workers at the primary and secondary care centres where most of these cases are delivered, on recognizing this and other anomalies and the need for prompt referral to appropriate centres. Secondly, intensive care facilities and trained personnel need to be improved. Though similar deficiency has been reported in some LMICs [6, 9, 10], the importance of intensive care in the management of OA cannot be overemphasized. Improving healthcare funding and international collaboration may aid provision of the facility and training of specialized staff. Finally, septicaemia has been recognized as a major contributor to morbidity and mortality in OA in some developing countries [9, 10, 18, 24]. In this study, it was responsible for almost 40% of the mortality and persisted in the Group B cases. Training the staff on infection control, establishing protocol for routine cleaning of cots, incubators and minimizing thoroughfare in newborn wards may help to combat sepsis [25, 26]. Long-term follow-up of these patients is crucial to recognize and address long-term sequel of treatment of OA, such as gastroesophageal reflux and stricture [3]. Though only one of our surviving cases has developed oesophageal stricture, the rest have to be monitored as the follow-up period increases. CONCLUSION In our setting, despite persisting barriers to care, outcome of OA in our setting may be improved with better anaesthesia and incorporation of temporary cardia banding to gastrostomy in cases that present late. Efforts to ensure early diagnosis, provision of intensive care facilities and infection control may further improve outcome. References 1 Pinheiro PF , Simões e Silva AC , Pereira RM. Current knowledge on esophageal atresia . World J Gastroenterol 2012 ; 18 : 3662 – 72 . 10.3748/wjg.v18.i28.3662. Google Scholar CrossRef Search ADS PubMed 2 Lal DR , Gadepalli SK , Downard CD , et al. Perioperative management and outcomes of esophageal atresia and tracheoesophageal fistula . J Pediatr Surg 2017 ; 52 : 1245 – 51 . 10.1016/j.jpedsurg.2016.11.046. Google Scholar CrossRef Search ADS PubMed 3 Donoso F , Kassa AM , Gustafson E , et al. Outcome and management in infants with esophageal atresia—a single centre observational study . 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Esophageal atresia and tracheo-esophageal fistula: a review . Indian J Pediatr 1999 ; 66 : 759 – 72 . http://dx.doi.org/10.1007/BF02726269 Google Scholar CrossRef Search ADS PubMed 19 Hosseini SM , Davani SZ , Sabet B , et al. The role of gastrostomy in the staged operation of esophageal atresia . J Indian Assoc Pediatr Surg 2008 ; 13 : 7 – 10 . 10.4103/0971-9261.42565. Google Scholar CrossRef Search ADS PubMed 20 Broemling N , Campbell F. Anesthetic management of congenital tracheoesophageal fistula . Paediatr Anaesth 2010 ; 21 : 1092 – 9 . 10.1111/j.1460-9592.2010.03377.x. Google Scholar CrossRef Search ADS PubMed 21 Sfeir R , Bonnard A , Khen-Dunlop N , et al. Esophageal atresia: data from a national cohort . J Pediatr Surg 2013 ; 48 : 1664 – 9 . 10.1016/j.jpedsurg.2013.03.075. Google Scholar CrossRef Search ADS PubMed 22 Jawaid W , Chan B , Jesudason EC. Subspecialization may improve an esophageal atresia service but has not addressed declining trainee experience . J Pediatr Surg 2012 ; 47 : 1363 – 8 . 10.1016/j.jpedsurg.2011.12.003. Google Scholar CrossRef Search ADS PubMed 23 Nwosu JN , Onyekwulu FA. Oesophageal atresia and tracheooesophageal fistula: a 12 years experience in a developing nation . Niger J Med 2013 ; 22 : 295 – 8 . Google Scholar PubMed 24 Vukadin M , Savic D , Malikovic A , et al. Analysis of prognostic factors and mortality in children with esophageal atresia . Indian J Pediatr 2015 ; 82 : 586 – 90 . 10.1007/s12098-015-1730-6. Google Scholar CrossRef Search ADS PubMed 25 Izquierdo-Blasco J , Campins-Martí M , Soler-Palacín P , et al. Impact of the implementation of an interdisciplinary infection control program to prevent surgical wound infection in pediatric heart surgery . Eur J Pediatr 2015 ; 174 : 957 – 63 . Google Scholar CrossRef Search ADS PubMed 26 Segal I , Kang C , Albersheim SG , et al. Surgical site infections in infants admitted to the neonatal intensive care unit . J Pediatr Surg 2014 ; 49 : 381 – 4 . http://dx.doi.org/10.1016/j.jpedsurg.2013.08.001 Google Scholar CrossRef Search ADS PubMed © The Author [2017]. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Tropical Pediatrics Oxford University Press

Effect of cardia banding and improved anaesthetic care on outcome of oesophageal atresia in a developing country

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Abstract

Abstract We comparatively analysed cases of oesophageal atresia (OA) managed in Enugu, south-eastern Nigeria from October 2010 to September 2015 to evaluate our short-term outcome with management following incorporation of temporary cardia banding to gastrostomy for late presenting cases and improved anaesthesia in 2013. Overall, 19 cases were analysed. The clinical parameters did not differ in the cases managed before (Group A) and after (Group B) these introductions. Four (21.1%) cases had primary repair (2 per group), six (31.6%) had delayed primary repair after treatment of pneumonitis (Group A 5; Group B 1) and nine (47.3%) had delayed primary repair after gastrostomy (Group A 4; Group B 5). Anaesthesia-related mortality dropped from 53.8 to 7.7% and survival improved from 9.1 to 62.5% following the introductions. Despite persisting barriers to care, outcome of OA in our setting may improve with better anaesthesia and incorporation of temporary cardia banding to gastrostomy. oesophageal atresia, anaesthesia, cardia banding, outcome, developing country INTRODUCTION Oesophageal atresia (OA) is a rare malformation with a prevalence of 2.43 per 10 000 births [1]. Refinements in surgical techniques, anaesthesia and neonatal intensive care have improved outcome [1–5]. Studies indicate that management of OA in most high-income countries (HICs) results in significantly higher survival compared with the outcome from most low- and middle-income countries (LMICs) [6, 7]. Factors reported to contribute to the poorer outcome of OA in the latter include late diagnosis with its attendant problems, and inadequate perioperative and anaesthetic care [6–9, 10]. Temporary gastric banding has been previously used in cases of premature babies with oesophageal atresia with tracheo-oesophageal fistula (OA-TEF) and severe respiratory distress [11, 12]. In 2013, we incorporated temporary cardia banding to gastrostomy in OA cases with complications of late presentation, before delayed primary repair. In the temporary banding, a purse-string suture (non-absorbable) is applied to the cardia of the stomach (avoiding the vagus nerve and other structures in the oesophagogastric junction) during gastrostomy procedure. The suture is removed during definitive repair of OA-TEF. In addition to this modification of procedure, one of our anaesthesiologists underwent additional training on neonatal anaesthesia. This study is a preliminary report of our experience with the management OA following improved anaesthesia care and incorporation of cardia banding. MATERIALS AND METHODS We retrospectively analysed the cases of OA managed from October 2010 to September 2015 at the University of Nigeria Teaching Hospital, and Mother of Christ Specialist Hospital, Enugu. Health research and ethics committee gave approval for the study (# NHREC/05/01/2008B-FWA00002458-1RB00002323). For the study, we collected the following data from the case notes, theatre records and discharge summaries: age at presentation and age at operation; gestational age at birth; birth weight, weight on presentation and weight at operation; sex; and findings on evaluation, type of atresia, operative procedures, outcome and follow-up. We compared the cases managed before September 2013 (Group A) with those managed from October 2013 (Group B) following improved anaesthesia care and incorporation of cardia banding during gastrostomy. We used Statistical Package for Social Sciences (SPSS 21.0 version, SPSS Inc., Chicago, Ill) for data entry and analysis. Results were expressed as percentages, or mean. Data were analysed by chi-square test with the significance level set at p < 0.05. RESULTS Overall, there were 19 patients (11 in Group A; 8 in Group B). In total, 11 (57.9%) cases were female and 8 (42.1%) were male. Their mean age at presentation was 8.3 ± 7 days (range 2–26 days). All the cases had proximal atresia and distal tracheaoesophageal fistula, and were delivered outside the two hospitals. The Patients’ characteristics are summarized in Table 1. Table 1 Summary of the patients’ characteristics in the two groups Parameter Group A Group B Median birth weight (kg) 2.6 kg (IQRa 2.4–3.0) 2.5 kg (IQR 2.4–2.8) Term gestational age 10/11 (91%) 8/8 (100%) Associated malformations 4/11 (36.4%) 3/8 (37.5%) Pneumonitis on presentation 9/11 (81.8%) 6/8 (75%) Median age at presentation 7 days (IQR 4–14) 10.5 days (IQR 2.5–15.8) Parameter Group A Group B Median birth weight (kg) 2.6 kg (IQRa 2.4–3.0) 2.5 kg (IQR 2.4–2.8) Term gestational age 10/11 (91%) 8/8 (100%) Associated malformations 4/11 (36.4%) 3/8 (37.5%) Pneumonitis on presentation 9/11 (81.8%) 6/8 (75%) Median age at presentation 7 days (IQR 4–14) 10.5 days (IQR 2.5–15.8) a IQR, interquartile range. Table 1 Summary of the patients’ characteristics in the two groups Parameter Group A Group B Median birth weight (kg) 2.6 kg (IQRa 2.4–3.0) 2.5 kg (IQR 2.4–2.8) Term gestational age 10/11 (91%) 8/8 (100%) Associated malformations 4/11 (36.4%) 3/8 (37.5%) Pneumonitis on presentation 9/11 (81.8%) 6/8 (75%) Median age at presentation 7 days (IQR 4–14) 10.5 days (IQR 2.5–15.8) Parameter Group A Group B Median birth weight (kg) 2.6 kg (IQRa 2.4–3.0) 2.5 kg (IQR 2.4–2.8) Term gestational age 10/11 (91%) 8/8 (100%) Associated malformations 4/11 (36.4%) 3/8 (37.5%) Pneumonitis on presentation 9/11 (81.8%) 6/8 (75%) Median age at presentation 7 days (IQR 4–14) 10.5 days (IQR 2.5–15.8) a IQR, interquartile range. Treatment The overall mean age at definitive treatment was 22.6 days (range 3–50 days). A total of four (21%) patients had primary repair and comprised two patients from each group. Six (31.6%) cases (Group A 5; Group B 1) had delayed primary repair after treatment of pneumonitis. The remaining nine (47.4%) patients had initial gastrostomy at a median age of 18 days (range 12–28 days). Of these cases, four patients (Group A) had Stamm gastrostomy, while five cases (Group B) had temporary cardia banding in addition to Stamm gastrostomy. After a period of 4–6 weeks, these cases had delayed primary repair. The average weight gain following gastrostomy in Group A patients was 0.4 kg, and Group B patients 0.9 kg. Outcome A total of eight (42.1%) patients developed postoperative complications, which includes surgical wound infection (n =  5; Group A 3; Group B 2) and anastomotic leak (n = 3; Group A 2; Group B 1). For anaesthesia, in Group A, four of the babies could not wake up from anaesthesia, and three of those who woke up had repeated apnoea in the absence of identifiable chest pathology, while in Group B, only one of the patients had postoperative apnoea. Overall, six patients (31.6%) survived. In Group A, 1 of 11 children (9%) survived, while in Group B, 5 of 8 children (62.5%) survived (1 of 11 in Group A; 5 of 8 in Group B; p = 0.024). The distribution of the survived cases is shown in Table 2 and the distribution and causes of mortality in the two groups are shown in Table 3. The surviving patients have been followed up for an average of 28 months (range 12–58 months). Only one of the patients (16.7%) developed oesophageal stricture, which responded to dilatation. Table 2 The distribution of the survived cases according to the definitive treatment in the two groups of patients with OA Survival Definitive treatment n Group A Group B p-value Primary repair 4 1/2 (50%) 1/2 (50%) 0.833 Delayed primary repair after treatment of pneumonitis 6 0/5 (0%) 1/1 (100%) 0.166 Delayed primary repair after gastrostomy 9 0/4 (0%) 3/5 (60%) 0.119 Total 19 1/11 (9.1%) 5/8 (62.5%) 0.024* Survival Definitive treatment n Group A Group B p-value Primary repair 4 1/2 (50%) 1/2 (50%) 0.833 Delayed primary repair after treatment of pneumonitis 6 0/5 (0%) 1/1 (100%) 0.166 Delayed primary repair after gastrostomy 9 0/4 (0%) 3/5 (60%) 0.119 Total 19 1/11 (9.1%) 5/8 (62.5%) 0.024* * p<0.05 (Significant). Table 2 The distribution of the survived cases according to the definitive treatment in the two groups of patients with OA Survival Definitive treatment n Group A Group B p-value Primary repair 4 1/2 (50%) 1/2 (50%) 0.833 Delayed primary repair after treatment of pneumonitis 6 0/5 (0%) 1/1 (100%) 0.166 Delayed primary repair after gastrostomy 9 0/4 (0%) 3/5 (60%) 0.119 Total 19 1/11 (9.1%) 5/8 (62.5%) 0.024* Survival Definitive treatment n Group A Group B p-value Primary repair 4 1/2 (50%) 1/2 (50%) 0.833 Delayed primary repair after treatment of pneumonitis 6 0/5 (0%) 1/1 (100%) 0.166 Delayed primary repair after gastrostomy 9 0/4 (0%) 3/5 (60%) 0.119 Total 19 1/11 (9.1%) 5/8 (62.5%) 0.024* * p<0.05 (Significant). Table 3 Distribution and causes of mortality following management of OA in the two groups of patients Groups Mortality Causes of morta1lity Uncontrolled sepsis Anaesthesia-related complications Group A 10/11 (90.9%) 3 7 Group B 3/8 (37.5%) 2 1 Groups Mortality Causes of morta1lity Uncontrolled sepsis Anaesthesia-related complications Group A 10/11 (90.9%) 3 7 Group B 3/8 (37.5%) 2 1 Table 3 Distribution and causes of mortality following management of OA in the two groups of patients Groups Mortality Causes of morta1lity Uncontrolled sepsis Anaesthesia-related complications Group A 10/11 (90.9%) 3 7 Group B 3/8 (37.5%) 2 1 Groups Mortality Causes of morta1lity Uncontrolled sepsis Anaesthesia-related complications Group A 10/11 (90.9%) 3 7 Group B 3/8 (37.5%) 2 1 DISCUSSION Given the reported global incidence of OA [1, 13, 14], the small number of cases in the present report may not indicate the actual incidence of OA in our setting. Apparently, some of these cases may not be captured in the tertiary referral centres [10, 15, 16]. The treatment of OA depends on the type of OA, the weight and state of the patient on presentation and the presence and severity of associated anomalies [1–5, 17]. In the present report, these parameters were similar in the two groups of patients, and these cases would have been better managed by primary repair. However, because of delayed presentation, majority of our cases presented with pneumonitis, undernutrition, sepsis or a combination of these. As a result, a substantial number of them required resuscitation and stabilization before definitive treatment. Similar management has been reported in some other developing countries [9, 10, 18]. The lack of total parenteral nutrition in our setting compounds the challenges and demands that we find a way to administer enteral nutrition. Gastrostomy has been used in cases of pure OA, and in OA-TEF that will require staged treatment [19]. In the latter, there is accompanying thoracotomy to ligate the fistula. The use of gastrostomy for feeding may be fraught with significant respiratory morbidity. The incorporation of temporary cardia banding though reported to be useful in extremely premature cases of OA with severe respiratory distress [11, 12] may be valuable in our setting as a way to reduce gastric aspiration and improve nutrition. With improvement in nutrition and weight gain, and reduction in airway contamination, these cases with complications of late presentation may be suitable for delayed primary repair. Anaesthetic management of OA has been shown to significantly affect the outcome of treatment [2, 4, 17]. Reports from HICs indicate that anaesthesia challenges have been systematically addressed [1, 20], while in some developing countries [8–10], there is report of persistence of inadequate trained personnel and lack of facilities. Lessons from HICs point to advantages of training in neonatal anaesthesia to sufficiently understand the peculiarities of these cases and adequately manage them. The marked reduction of mortality from anaesthesia and respiratory complications following improved anaesthetic management in the present report may attest to that. The overall outcome of OA management in the present report is poor compared with the >90% survival reported in most HICs [1–5, 14, 21, 22]. Although there was a significant improvement of outcome with better anaesthesia, improved nutrition and reduced airway contamination with cardia banding compared with earlier cases managed in the setting [23], the outcome still fell short of the current results from more developed countries where current mortality is limited to cases with associated life-threatening anomalies [1–3]. The absence of these anomalies in our cases and the high mortality in the present report indicate that there are inherent deficiencies in our management of OA that need to be addressed. In the first place, delayed presentation and late intervention are major problems in our setting and have also been reported in some other LMICs [7–10]. Addressing the delays may require improvement of antenatal and perinatal cares, and concerted efforts to educate the healthcare workers at the primary and secondary care centres where most of these cases are delivered, on recognizing this and other anomalies and the need for prompt referral to appropriate centres. Secondly, intensive care facilities and trained personnel need to be improved. Though similar deficiency has been reported in some LMICs [6, 9, 10], the importance of intensive care in the management of OA cannot be overemphasized. Improving healthcare funding and international collaboration may aid provision of the facility and training of specialized staff. Finally, septicaemia has been recognized as a major contributor to morbidity and mortality in OA in some developing countries [9, 10, 18, 24]. In this study, it was responsible for almost 40% of the mortality and persisted in the Group B cases. Training the staff on infection control, establishing protocol for routine cleaning of cots, incubators and minimizing thoroughfare in newborn wards may help to combat sepsis [25, 26]. Long-term follow-up of these patients is crucial to recognize and address long-term sequel of treatment of OA, such as gastroesophageal reflux and stricture [3]. Though only one of our surviving cases has developed oesophageal stricture, the rest have to be monitored as the follow-up period increases. CONCLUSION In our setting, despite persisting barriers to care, outcome of OA in our setting may be improved with better anaesthesia and incorporation of temporary cardia banding to gastrostomy in cases that present late. Efforts to ensure early diagnosis, provision of intensive care facilities and infection control may further improve outcome. References 1 Pinheiro PF , Simões e Silva AC , Pereira RM. Current knowledge on esophageal atresia . World J Gastroenterol 2012 ; 18 : 3662 – 72 . 10.3748/wjg.v18.i28.3662. Google Scholar CrossRef Search ADS PubMed 2 Lal DR , Gadepalli SK , Downard CD , et al. Perioperative management and outcomes of esophageal atresia and tracheoesophageal fistula . J Pediatr Surg 2017 ; 52 : 1245 – 51 . 10.1016/j.jpedsurg.2016.11.046. Google Scholar CrossRef Search ADS PubMed 3 Donoso F , Kassa AM , Gustafson E , et al. Outcome and management in infants with esophageal atresia—a single centre observational study . 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Journal of Tropical PediatricsOxford University Press

Published: Dec 14, 2017

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