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Mortality from gastrointestinal congenital anomalies at 264 hospitals in 74 low-income, middle-income, and high-income countries : a multicentre, international, prospective cohort study

Wright, Naomi Jane ; Ademuyiwa, Adesoji ; Ameh, Emmanuel ; Davies, Justine ; Lakhoo, Kokila ; Poenaru, Dan ; Smith, Emily Rose ; Ade-Ajayi, Niyi ; Sevdalis, Nick and Leather, Andrew J.M. , et al. (2021) In The Lancet 398(10297). p.325-339
Abstract

Background: Congenital anomalies are the fifth leading cause of mortality in children younger than 5 years globally. Many gastrointestinal congenital anomalies are fatal without timely access to neonatal surgical care, but few studies have been done on these conditions in low-income and middle-income countries (LMICs). We compared outcomes of the seven most common gastrointestinal congenital anomalies in low-income, middle-income, and high-income countries globally, and identified factors associated with mortality. Methods: We did a multicentre, international prospective cohort study of patients younger than 16 years, presenting to hospital for the first time with oesophageal atresia, congenital diaphragmatic hernia, intestinal atresia,... (More)

Background: Congenital anomalies are the fifth leading cause of mortality in children younger than 5 years globally. Many gastrointestinal congenital anomalies are fatal without timely access to neonatal surgical care, but few studies have been done on these conditions in low-income and middle-income countries (LMICs). We compared outcomes of the seven most common gastrointestinal congenital anomalies in low-income, middle-income, and high-income countries globally, and identified factors associated with mortality. Methods: We did a multicentre, international prospective cohort study of patients younger than 16 years, presenting to hospital for the first time with oesophageal atresia, congenital diaphragmatic hernia, intestinal atresia, gastroschisis, exomphalos, anorectal malformation, and Hirschsprung's disease. Recruitment was of consecutive patients for a minimum of 1 month between October, 2018, and April, 2019. We collected data on patient demographics, clinical status, interventions, and outcomes using the REDCap platform. Patients were followed up for 30 days after primary intervention, or 30 days after admission if they did not receive an intervention. The primary outcome was all-cause, in-hospital mortality for all conditions combined and each condition individually, stratified by country income status. We did a complete case analysis. Findings: We included 3849 patients with 3975 study conditions (560 with oesophageal atresia, 448 with congenital diaphragmatic hernia, 681 with intestinal atresia, 453 with gastroschisis, 325 with exomphalos, 991 with anorectal malformation, and 517 with Hirschsprung's disease) from 264 hospitals (89 in high-income countries, 166 in middle-income countries, and nine in low-income countries) in 74 countries. Of the 3849 patients, 2231 (58·0%) were male. Median gestational age at birth was 38 weeks (IQR 36–39) and median bodyweight at presentation was 2·8 kg (2·3–3·3). Mortality among all patients was 37 (39·8%) of 93 in low-income countries, 583 (20·4%) of 2860 in middle-income countries, and 50 (5·6%) of 896 in high-income countries (p<0·0001 between all country income groups). Gastroschisis had the greatest difference in mortality between country income strata (nine [90·0%] of ten in low-income countries, 97 [31·9%] of 304 in middle-income countries, and two [1·4%] of 139 in high-income countries; p≤0·0001 between all country income groups). Factors significantly associated with higher mortality for all patients combined included country income status (low-income vs high-income countries, risk ratio 2·78 [95% CI 1·88–4·11], p<0·0001; middle-income vs high-income countries, 2·11 [1·59–2·79], p<0·0001), sepsis at presentation (1·20 [1·04–1·40], p=0·016), higher American Society of Anesthesiologists (ASA) score at primary intervention (ASA 4–5 vs ASA 1–2, 1·82 [1·40–2·35], p<0·0001; ASA 3 vs ASA 1–2, 1·58, [1·30–1·92], p<0·0001]), surgical safety checklist not used (1·39 [1·02–1·90], p=0·035), and ventilation or parenteral nutrition unavailable when needed (ventilation 1·96, [1·41–2·71], p=0·0001; parenteral nutrition 1·35, [1·05–1·74], p=0·018). Administration of parenteral nutrition (0·61, [0·47–0·79], p=0·0002) and use of a peripherally inserted central catheter (0·65 [0·50–0·86], p=0·0024) or percutaneous central line (0·69 [0·48–1·00], p=0·049) were associated with lower mortality. Interpretation: Unacceptable differences in mortality exist for gastrointestinal congenital anomalies between low-income, middle-income, and high-income countries. Improving access to quality neonatal surgical care in LMICs will be vital to achieve Sustainable Development Goal 3.2 of ending preventable deaths in neonates and children younger than 5 years by 2030. Funding: Wellcome Trust.

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The Lancet
volume
398
issue
10297
pages
15 pages
publisher
Elsevier
external identifiers
  • pmid:34270932
  • scopus:85110686338
ISSN
0140-6736
DOI
10.1016/S0140-6736(21)00767-4
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English
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Funding Information: NJW is funded by the Wellcome Trust through a Clinical PhD in Global Health undertaken at King's College London (203905/Z/16/Z). The Wellcome Trust had no input into the study protocol other than to recommend open-access publication in a peer-reviewed journal and to make the anonymised dataset publicly available. NS receives research support from the National Institute for Health Research (NIHR) Applied Research Collaboration South London at King's College Hospital National Health System (NHS) Foundation Trust. NS is a member of King's Improvement Science, which offers co-funding to the NIHR Appilied Research Collaboration South London and is funded by King's Health Partners (Guy's and St Thomas' NHS Foundation Trust, King's College Hospital NHS Foundation Trust, King's College London and South London and Maudsley NHS Foundation Trust), Guy's and St Thomas' Charity, and the Maudsley Charity. NS and AL are also supported by the NIHR Global Health Research Unit on Health System Strengthening in Sub-Saharan Africa, King's College London (GHRU 16/136/54) and by the ASPIRES research programme in low-income and middle-income countries (Antibiotic use across Surgical Pathways?Investigating, Redesigning and Evaluating Systems), funded by the Economic and Social Research Council (ESRC). The views expressed are those of the authors and not necessarily those of the Wellcome Trust, NHS, the NIHR, the ESRC or the Department of Health and Social Care. The authors thank Bolaji Coker and Alexandra Vincent for the REDCap administration and management; Beverley Power and team (CDH UK) for representing patients, parents, and families through feedback on the study design during the pilot study and for contributing to data interpretation and manuscript content; and Xiya Ma and Dylan Goh for helping with the Chinese translation of study documentation. We thank the following individuals for dissemination and local investigator recruitment: Dawn M Torrence Ireland (CDH International), Lawal-Aiyedun Olubunmi (Spina Bifida and Hydrocephalus Care Foundation, Nigeria), Matthijs Botman (Global Surgey Amsterdam, Netherlands), Rinse Meester (Netherlands Society for International Surgery, Netherlands), Amanda J Neville (EUROCAT Association), Greg Ryan (ONE in 5000 Foundation), and Hassan Ali Daoud and Mohamed Hassan Nour (InciSion Somaliland, Somaliland). Editorial note: the Lancet Group takes a neutral position with respect to territorial claims in published maps and institutional affiliations. Funding Information: NJW is funded by the Wellcome Trust through a Clinical PhD in Global Health undertaken at King's College London (203905/Z/16/Z). The Wellcome Trust had no input into the study protocol other than to recommend open-access publication in a peer-reviewed journal and to make the anonymised dataset publicly available. NS receives research support from the National Institute for Health Research (NIHR) Applied Research Collaboration South London at King's College Hospital National Health System (NHS) Foundation Trust. NS is a member of King's Improvement Science, which offers co-funding to the NIHR Appilied Research Collaboration South London and is funded by King's Health Partners (Guy's and St Thomas' NHS Foundation Trust, King's College Hospital NHS Foundation Trust, King's College London and South London and Maudsley NHS Foundation Trust), Guy's and St Thomas' Charity, and the Maudsley Charity. NS and AL are also supported by the NIHR Global Health Research Unit on Health System Strengthening in Sub-Saharan Africa, King's College London (GHRU 16/136/54) and by the ASPIRES research programme in low-income and middle-income countries (Antibiotic use across Surgical Pathways—Investigating, Redesigning and Evaluating Systems), funded by the Economic and Social Research Council (ESRC). The views expressed are those of the authors and not necessarily those of the Wellcome Trust, NHS, the NIHR, the ESRC or the Department of Health and Social Care. The authors thank Bolaji Coker and Alexandra Vincent for the REDCap administration and management; Beverley Power and team (CDH UK) for representing patients, parents, and families through feedback on the study design during the pilot study and for contributing to data interpretation and manuscript content; and Xiya Ma and Dylan Goh for helping with the Chinese translation of study documentation. We thank the following individuals for dissemination and local investigator recruitment: Dawn M Torrence Ireland (CDH International), Lawal-Aiyedun Olubunmi (Spina Bifida and Hydrocephalus Care Foundation, Nigeria), Matthijs Botman (Global Surgey Amsterdam, Netherlands), Rinse Meester (Netherlands Society for International Surgery, Netherlands), Amanda J Neville (EUROCAT Association), Greg Ryan (ONE in 5000 Foundation), and Hassan Ali Daoud and Mohamed Hassan Nour (InciSion Somaliland, Somaliland). Publisher Copyright: © 2021 The Author(s). Published by Elsevier Ltd. This is an Open Access article under the CC BY 4.0 license Copyright: Copyright 2021 Elsevier B.V., All rights reserved.
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date added to LUP
2021-09-10 12:10:12
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2024-06-29 17:13:42
@article{10f7c5b0-7223-4f35-ab31-566873382ace,
  abstract     = {{<p>Background: Congenital anomalies are the fifth leading cause of mortality in children younger than 5 years globally. Many gastrointestinal congenital anomalies are fatal without timely access to neonatal surgical care, but few studies have been done on these conditions in low-income and middle-income countries (LMICs). We compared outcomes of the seven most common gastrointestinal congenital anomalies in low-income, middle-income, and high-income countries globally, and identified factors associated with mortality. Methods: We did a multicentre, international prospective cohort study of patients younger than 16 years, presenting to hospital for the first time with oesophageal atresia, congenital diaphragmatic hernia, intestinal atresia, gastroschisis, exomphalos, anorectal malformation, and Hirschsprung's disease. Recruitment was of consecutive patients for a minimum of 1 month between October, 2018, and April, 2019. We collected data on patient demographics, clinical status, interventions, and outcomes using the REDCap platform. Patients were followed up for 30 days after primary intervention, or 30 days after admission if they did not receive an intervention. The primary outcome was all-cause, in-hospital mortality for all conditions combined and each condition individually, stratified by country income status. We did a complete case analysis. Findings: We included 3849 patients with 3975 study conditions (560 with oesophageal atresia, 448 with congenital diaphragmatic hernia, 681 with intestinal atresia, 453 with gastroschisis, 325 with exomphalos, 991 with anorectal malformation, and 517 with Hirschsprung's disease) from 264 hospitals (89 in high-income countries, 166 in middle-income countries, and nine in low-income countries) in 74 countries. Of the 3849 patients, 2231 (58·0%) were male. Median gestational age at birth was 38 weeks (IQR 36–39) and median bodyweight at presentation was 2·8 kg (2·3–3·3). Mortality among all patients was 37 (39·8%) of 93 in low-income countries, 583 (20·4%) of 2860 in middle-income countries, and 50 (5·6%) of 896 in high-income countries (p&lt;0·0001 between all country income groups). Gastroschisis had the greatest difference in mortality between country income strata (nine [90·0%] of ten in low-income countries, 97 [31·9%] of 304 in middle-income countries, and two [1·4%] of 139 in high-income countries; p≤0·0001 between all country income groups). Factors significantly associated with higher mortality for all patients combined included country income status (low-income vs high-income countries, risk ratio 2·78 [95% CI 1·88–4·11], p&lt;0·0001; middle-income vs high-income countries, 2·11 [1·59–2·79], p&lt;0·0001), sepsis at presentation (1·20 [1·04–1·40], p=0·016), higher American Society of Anesthesiologists (ASA) score at primary intervention (ASA 4–5 vs ASA 1–2, 1·82 [1·40–2·35], p&lt;0·0001; ASA 3 vs ASA 1–2, 1·58, [1·30–1·92], p&lt;0·0001]), surgical safety checklist not used (1·39 [1·02–1·90], p=0·035), and ventilation or parenteral nutrition unavailable when needed (ventilation 1·96, [1·41–2·71], p=0·0001; parenteral nutrition 1·35, [1·05–1·74], p=0·018). Administration of parenteral nutrition (0·61, [0·47–0·79], p=0·0002) and use of a peripherally inserted central catheter (0·65 [0·50–0·86], p=0·0024) or percutaneous central line (0·69 [0·48–1·00], p=0·049) were associated with lower mortality. Interpretation: Unacceptable differences in mortality exist for gastrointestinal congenital anomalies between low-income, middle-income, and high-income countries. Improving access to quality neonatal surgical care in LMICs will be vital to achieve Sustainable Development Goal 3.2 of ending preventable deaths in neonates and children younger than 5 years by 2030. Funding: Wellcome Trust.</p>}},
  author       = {{Wright, Naomi Jane and Ademuyiwa, Adesoji and Ameh, Emmanuel and Davies, Justine and Lakhoo, Kokila and Poenaru, Dan and Smith, Emily Rose and Ade-Ajayi, Niyi and Sevdalis, Nick and Leather, Andrew J.M. and Douiri, Abdel and Elstad, Maria and Sim, Marcus and Riboni, Cristiana and Martinez-Leo, Bruno and Akhbari, Melika and Tabiri, Stephen and Mitul, Ashrarur and Aziz, Dayang Anita Abdul and Fachin, Camila and Niyukuri, Alliance and Arshad, Muhammad and Salö, Martin}},
  issn         = {{0140-6736}},
  language     = {{eng}},
  month        = {{07}},
  number       = {{10297}},
  pages        = {{325--339}},
  publisher    = {{Elsevier}},
  series       = {{The Lancet}},
  title        = {{Mortality from gastrointestinal congenital anomalies at 264 hospitals in 74 low-income, middle-income, and high-income countries : a multicentre, international, prospective cohort study}},
  url          = {{http://dx.doi.org/10.1016/S0140-6736(21)00767-4}},
  doi          = {{10.1016/S0140-6736(21)00767-4}},
  volume       = {{398}},
  year         = {{2021}},
}