Management and outcomes of gastrointestinal congenital anomalies in low, middle and high income countries: protocol for a multicentre, international, prospective cohort study

Naomi Jane Wright, Global PaedSurg Research Collaboration, Naomi Wright, Andrew Leather, Nick Sevdalis, Niyi Ade-Ajayi, Adesoji Ademuyiwa, Emmanuel Ameh, Justine Davies, Kokila Lakhoo, Dan Poenaru, Emily Smith, Harmony Ubhi, Samuel Parker, Godfrey Sama Philipo, Sadi Abukhalaf, Nana Adofo-Ansong, Melika Akhbari, Ahmad Alhamid, Osaid H Alser, Emrah Aydin, Yousra-Imane Benaskeur, Shrouk M Elghazaly, Safa Abdal Elrais, Sophia Hashim, Laura Herrera, Gabriella Hyman, Henang Kwasau, Yang Liu, Bruno Martinez-Leo, Kelly Naranjo, Ibrahim Nour, Cristiana Riboni, Mahmoud Saleh, Hosni Khairy Salem, Patricia Shinondo, Marcus Sim, Hannah Thompson, Agota Vaitkiene, Dominique Vervoort, Isabelle Williams, Aayenah Yunus, Muhammad Amjad Chaudhary, Muhammad Adnan Khan Khattak, Muhammad Bin Amjad, Marlene Dominguez Anaya, Samiul Hasan, Sabbir Karim, Ashrarur Rahman Mitul, Paolo Bragagnini, Segundo Rite, Hana Arbab, Lubna Samad, Aqil Soomro, Niveshni Maistry, Raed Nael Al-Taher, Ibrahim Rabi Nour, Osama Abdul Kareen Sarhan, Muhammad Arshad, Taimur Qureshi, Hina Yousaf, Candy S C Choo, Doris Mae Dimatatac, Shireen Anne Nah, Vijay Anand Ismavel, Ann Miriam, Shajin T, Monica Ivanov, Andreea Serban, Eva Blazquez-Gomez, Luis Garcia-Aparicio, Marti Iriondo, Jordi Prat, Xavier Tarrado, Lars Hagander, Emma Svensson, Alhassan Abdul-Mumin, Dominic Bagbio, Sheila Owusu, Stephen Tabiri, Dayang Anita Abdul Aziz, Naomi Jane Wright, Global PaedSurg Research Collaboration, Naomi Wright, Andrew Leather, Nick Sevdalis, Niyi Ade-Ajayi, Adesoji Ademuyiwa, Emmanuel Ameh, Justine Davies, Kokila Lakhoo, Dan Poenaru, Emily Smith, Harmony Ubhi, Samuel Parker, Godfrey Sama Philipo, Sadi Abukhalaf, Nana Adofo-Ansong, Melika Akhbari, Ahmad Alhamid, Osaid H Alser, Emrah Aydin, Yousra-Imane Benaskeur, Shrouk M Elghazaly, Safa Abdal Elrais, Sophia Hashim, Laura Herrera, Gabriella Hyman, Henang Kwasau, Yang Liu, Bruno Martinez-Leo, Kelly Naranjo, Ibrahim Nour, Cristiana Riboni, Mahmoud Saleh, Hosni Khairy Salem, Patricia Shinondo, Marcus Sim, Hannah Thompson, Agota Vaitkiene, Dominique Vervoort, Isabelle Williams, Aayenah Yunus, Muhammad Amjad Chaudhary, Muhammad Adnan Khan Khattak, Muhammad Bin Amjad, Marlene Dominguez Anaya, Samiul Hasan, Sabbir Karim, Ashrarur Rahman Mitul, Paolo Bragagnini, Segundo Rite, Hana Arbab, Lubna Samad, Aqil Soomro, Niveshni Maistry, Raed Nael Al-Taher, Ibrahim Rabi Nour, Osama Abdul Kareen Sarhan, Muhammad Arshad, Taimur Qureshi, Hina Yousaf, Candy S C Choo, Doris Mae Dimatatac, Shireen Anne Nah, Vijay Anand Ismavel, Ann Miriam, Shajin T, Monica Ivanov, Andreea Serban, Eva Blazquez-Gomez, Luis Garcia-Aparicio, Marti Iriondo, Jordi Prat, Xavier Tarrado, Lars Hagander, Emma Svensson, Alhassan Abdul-Mumin, Dominic Bagbio, Sheila Owusu, Stephen Tabiri, Dayang Anita Abdul Aziz

Abstract

Introduction: Congenital anomalies are the fifth leading cause of death in children <5 years of age globally, contributing an estimated half a million deaths per year. Very limited literature exists from low and middle income countries (LMICs) where most of these deaths occur. The Global PaedSurg Research Collaboration aims to undertake the first multicentre, international, prospective cohort study of a selection of common congenital anomalies comparing management and outcomes between low, middle and high income countries (HICs) globally.

Methods and analysis: The Global PaedSurg Research Collaboration consists of surgeons, paediatricians, anaesthetists and allied healthcare professionals involved in the surgical care of children globally. Collaborators will prospectively collect observational data on consecutive patients presenting for the first time, with one of seven common congenital anomalies (oesophageal atresia, congenital diaphragmatic hernia, intestinal atresia, gastroschisis, exomphalos, anorectal malformation and Hirschsprung's disease).Patient recruitment will be for a minimum of 1 month from October 2018 to April 2019 with a 30-day post-primary intervention follow-up period. Anonymous data will be collected on patient demographics, clinical status, interventions and outcomes using REDCap. Collaborators will complete a survey regarding the resources and facilities for neonatal and paediatric surgery at their centre.The primary outcome is all-cause in-hospital mortality. Secondary outcomes include the occurrence of post-operative complications. Chi-squared analysis will be used to compare mortality between LMICs and HICs. Multilevel, multivariate logistic regression analysis will be undertaken to identify patient-level and hospital-level factors affecting outcomes with adjustment for confounding factors.

Ethics and dissemination: At the host centre, this study is classified as an audit not requiring ethical approval. All participating collaborators have gained local approval in accordance with their institutional ethical regulations. Collaborators will be encouraged to present the results locally, nationally and internationally. The results will be submitted for open access publication in a peer reviewed journal.

Trial registration number: NCT03666767.

Keywords: neonatal intensive and critical care; neonatology; paediatric anaesthesia; paediatric intensive and critical care; paediatric surgery; paediatrics.

Conflict of interest statement

Competing interests: NS is the director of the London Safety and Training Solutions, which offers training in patient safety, implementation solutions and human factors to healthcare organisations. No other conflicts of interest are declared.

© Author(s) (or their employer(s)) 2019. Re-use permitted under CC BY. Published by BMJ.

References

    1. Wang H, Bhutta ZA, Coates MM, et al. . Global, regional, national, and selected subnational levels of stillbirths, neonatal, infant, and under-5 mortality, 1980–2015: a systematic analysis for the global burden of disease study 2015. The Lancet 2016;388:1725–74. 10.1016/S0140-6736(16)31575-6
    1. Boyle B, Addor M-C, Arriola L, et al. . Estimating global burden of disease due to congenital anomaly: an analysis of European data. Arch Dis Child Fetal Neonatal Ed 2018;103:F22–F28. 10.1136/archdischild-2016-311845
    1. Flores A, Valencia D, Sekkarie A, et al. . Building capacity for birth defects surveillance in Africa: implementation of an intermediate birth defects surveillance workshop. J Glob Health Perspect 2015;2015
    1. Sitkin NA, Ozgediz D, Donkor P, et al. . Congenital anomalies in low- and middle-income countries: the unborn child of global surgery. World J Surg 2015;39:36–40. 10.1007/s00268-014-2714-9
    1. Ozgediz D, Langer M, Kisa P, et al. . Pediatric surgery as an essential component of global child health. Semin Pediatr Surg 2016;25:3–9. 10.1053/j.sempedsurg.2015.09.002
    1. Farmer D, Sitkin N, Lofberg K, et al. . Surgical Interventions for Congenital Anomalies : Debas HT, Donkor P, Gawande A, et al., Essential surgery: disease control priorities. Volume 1 Third Edition Washington, DC, 2015.
    1. Sakonidou S, Ali K, Farmer I, et al. . Mortality and short-term morbidity in infants with Exomphalos. Pediatr Int 2018;60:438–41. 10.1111/ped.13537
    1. Ameh EA, Seyi-Olajide JO, Sholadoye TT. Neonatal surgical care: a review of the burden, progress and challenges in sub-Saharan Africa. Paediatr Int Child Health 2015;35:243–51. 10.1179/2046905515Y.0000000033
    1. Ekenze SO, Ajuzieogu OV, Nwomeh BC. Challenges of management and outcome of neonatal surgery in Africa: a systematic review. Pediatr Surg Int 2016;32:291–9. 10.1007/s00383-016-3861-x
    1. Wright NJ, Zani A, Ade-Ajayi N. Epidemiology, management and outcome of gastroschisis in sub-Saharan Africa: results of an international survey. Afr J Paediatr Surg 2015;12:1–6. 10.4103/0189-6725.150924
    1. PaedSurg Africa Research Collaboration Paediatric surgery across sub-Saharan Africa: a multi-centre prospective cohort study. Available: [Accessed 26 Jan 2019].
    1. Bradnock TJ, Marven S, Owen A, et al. . Gastroschisis: one year outcomes from national cohort study. BMJ 2011;343 10.1136/bmj.d6749
    1. Krishnaswami S, Nwomeh BC, Ameh EA. The pediatric surgery workforce in low- and middle-income countries: problems and priorities. Semin Pediatr Surg 2016;25:32–42. 10.1053/j.sempedsurg.2015.09.007
    1. Okoye MT, Ameh EA, Kushner AL, et al. . A pilot survey of pediatric surgical capacity in West Africa. World J Surg 2015;39:669–76. 10.1007/s00268-014-2868-5
    1. United Nations Sustainable development goals, 2015. Available: [Accessed 27 Feb 2018].
    1. Wright NJ, Anderson JE, Ozgediz D, et al. . Addressing paediatric surgical care on world birth defects day. The Lancet 2018;391 10.1016/S0140-6736(18)30501-4
    1. GlobalSurg Collaborative Mortality of emergency abdominal surgery in high-, middle- and low-income countries. Br J Surg 2016;103:971–88. 10.1002/bjs.10151
    1. Bhangu A, Ademuyiwa AO, Aguilera ML, et al. . Surgical site infection after gastrointestinal surgery in high-income, middle-income, and low-income countries: a prospective, international, multicentre cohort study. Lancet Infect Dis 2018;18:516–25. 10.1016/S1473-3099(18)30101-4
    1. GlobalSurg Collaborative Laparoscopy in management of appendicitis in high-, middle-, and low-income countries: a multicenter, prospective, cohort study. Surg Endosc 2018;32:3450–66. 10.1007/s00464-018-6064-9
    1. GlobalSurg Collaborative Determinants of morbidity and mortality following emergency abdominal surgery in children in low-income and middle-income countries. BMJ Glob Health 2016;1:e000091 10.1136/bmjgh-2016-000091
    1. Bhangu A, Kolias AG, Pinkney T, et al. . Surgical research Collaboratives in the UK. The Lancet 2013;382:1091–2. 10.1016/S0140-6736(13)62013-9
    1. Long A-M, Bunch KJ, Knight M, et al. . Early population-based outcomes of infants born with congenital diaphragmatic hernia. Arch Dis Child Fetal Neonatal Ed 2018;103:F517–F522. 10.1136/archdischild-2017-313933
    1. Ruttenstock E, Wright N, Barrena S, et al. . Best oxygenation index on day 1: a reliable marker for outcome and survival in infants with congenital diaphragmatic hernia. Eur J Pediatr Surg 2015;25:3–8. 10.1055/s-0034-1393960
    1. Burjonrappa S, Crete E, Bouchard S. Comparative outcomes in intestinal atresia: a clinical outcome and pathophysiology analysis. Pediatr Surg Int 2011;27:437–42. 10.1007/s00383-010-2729-8
    1. Forrester MB, Merz RD. Structural birth defects associated with omphalocele and gastroschisis, Hawaii, 1986–2001. Congenit Anom 2008;48:87–91. 10.1111/j.1741-4520.2008.00184.x
    1. Kunz SN, Tieder JS, Whitlock K, et al. . Primary fascial closure versus staged closure with silo in patients with gastroschisis: a meta-analysis. J Pediatr Surg 2013;48:845–57. 10.1016/j.jpedsurg.2013.01.020
    1. Ross AR, Eaton S, Zani A, et al. . The role of preformed silos in the management of infants with gastroschisis: a systematic review and meta-analysis. Pediatr Surg Int 2015;31:473–83. 10.1007/s00383-015-3691-2
    1. Levitt MA, Peña A. Anorectal malformations. Orphanet J Rare Dis 2007;2:33 10.1186/1750-1172-2-33
    1. van der Steeg HJJ, Schmiedeke E, Bagolan P, et al. . European consensus meeting of ARM-Net members concerning diagnosis and early management of newborns with anorectal malformations. Tech Coloproctol 2015;19:181–5. 10.1007/s10151-015-1267-8
    1. Holschneider A, Hutson J, Peña A, et al. . Preliminary report on the International Conference for the development of standards for the treatment of anorectal malformations. J Pediatr Surg 2005;40:1521–6. 10.1016/j.jpedsurg.2005.08.002
    1. Bradnock TJ, Knight M, Kenny S, et al. . British Association of Paediatric Surgeons Congenital Anomalies Surveillance System (BAPS-CASS). Hirschsprung's disease in the UK and Ireland: incidence and anomalies. Arch Dis Child 2017;102:722–7.
    1. Ross AR, Hall NJ. Outcome reporting in randomized controlled trials and systematic reviews of gastroschisis treatment: a systematic review. J Pediatr Surg 2016;51:1385–9. 10.1016/j.jpedsurg.2016.05.008
    1. Allin BSR, Hall NJ, Ross AR, et al. . Development of a gastroschisis core outcome set. Arch Dis Child Fetal Neonatal Ed 2019;104:F76–F82. 10.1136/archdischild-2017-314560
    1. Allin BSR, Irvine A, Patni N, et al. . Variability of outcome reporting in Hirschsprung’s Disease and gastroschisis: a systematic review. Sci Rep 2016;6 10.1038/srep38969
    1. Watanabe S, Suzuki T, Hara F, et al. . Omphalocele and gastroschisis in newborns: over 16 years of experience from a single clinic. J Neonatal Surg 2017;6 10.21699/jns.v6i2.530
    1. Tan KB, Tan KH, Chew SK, et al. . Gastroschisis and omphalocele in Singapore: a ten-year series from 1993 to 2002. Singapore Med J 2008;49:31–6.
    1. Schneider A, Blanc S, Bonnard A, et al. . Results from the French national esophageal atresia register: one-year outcome. Orphanet J Rare Dis 2014;9:206 10.1186/s13023-014-0206-5
    1. World Bank World bank country and lending groups, 2018. Available: [Accessed 26 Jan 2019].

Source: PubMed

赞助者和合作者

医疗条件

药物干预

3
订阅