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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
The Lancet ( IF 168.9 ) Pub Date : 2021-07-13 , DOI: 10.1016/s0140-6736(21)00767-4
Naomi Jane Wright , Andrew J.M. Leather , Niyi Ade-Ajayi , Nick Sevdalis , Justine Davies , Dan Poenaru , Emmanuel Ameh , Adesoji Ademuyiwa , Kokila Lakhoo , Emily Rose Smith , Abdel Douiri , Maria Elstad , Marcus Sim , Cristiana Riboni , Bruno Martinez-Leo , Melika Akhbari , Stephen Tabiri , Ashrarur Mitul , Dayang Anita Abdul Aziz , Camila Fachin , Alliance Niyukuri , Muhammad Arshad , Fowzia Ibrahim , Natalie Moitt , Mohamed Fahmy Doheim , Hannah Thompson , Harmony Ubhi , Isabelle Williams , Sophia Hashim , Godfrey Sama Philipo , Laura Herrera , Aayenah Yunus , Dominique Vervoort , Samuel Parker , Yousra-Imane Benaskeur , Osaid H. Alser , Nana Adofo-Ansong , Ahmad Alhamid , Hosni khairy Salem , Mahmoud Saleh , Safa Abdal Elrais , Sadi Abukhalaf , Patricia Shinondo , Ibrahim Nour , Emrah Aydin , Agota Vaitkiene , Kelly Naranjo , Andile Maqhawe Dube , Sodumisa Ngwenya , Mina A. Yacoub , Henang Kwasau , Gabriella Hyman , Shrouk Mahmoud Elghazaly , Ibrahim Al-Slaibi , Intisar Hisham , Helena Franco , Hana Arbab , Lubna Samad , Aqil Soomro , Muhammad Amjad Chaudhry , Safina Karim , Muhammad Adnan Khan Khattak , Shireen Anne Nah , Doris Mae Dimatatac , Candy SC Choo , Niveshni Maistry , Ashrarur Rahman Mitul , Samiul Hasan , Sabbir Karim , Hina Yousuf , Taimur Qureshi , Ibrahim Rabi Nour , Raed Nael Al-Taher , Osama Abdul Kareem Sarhan , Luis Garcia-Aparicio , Jordi Prat , Eva Blazquez-Gomez , Xavier Tarrado , Martí Iriondo , Paolo Bragagnini , Segundo Rite , Lars Hagander , Emma Svensson , Sheila Owusu , Alhassan Abdul-Mumin , Dominic Bagbio , Vijay Anand Ismavel , Ann Miriam , Shajin T , Marlene Anaya Dominguez , Monica Ivanov , Andreea Madalina Serban , Miliard Derbew , Mahmoud Elfiky , Maricarmen Olivos Perez , Marcia Abrunhosa Matias , Alexis P Arnaud , Ahmed Negida , Sebastian King , Mohamad Rafi Fazli , Nadia Hamidi , Souhem Touabti , Rossana Francisco Chipalavela , Pablo Lobos , Brendan Jones , Damir Ljuhar , Georg Singer , Samiul Hasan , Annelien Cordonnier , Lorena Jáuregui , Zlatan Zvizdic , Janice Wong , Etienne St-Louis , Qiang Shu , Yang Lui , Catalina Correa , Lucie Pos , Elvyn Alcántara , Erick Féliz , Luis Enrique Zea-Salazar , Liza Ali , Matthieu Peycelon , Nzanzu Kipata Anatole , Cherno S. 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Alijla , Mohamed Anwer El Tallaa , Adham Ashraf Abuattaya , Bisan D.M. Wishah , MOHAMMED A.M. ALDIRAWI , Ahmed S Darwish , Sulaiman T. Alzerei , Nidal Wishah , Sharif Alijla , Isidora Garcia , Marlene Diaz Echegaray , Veronica Raquel Cañapataña Sahuanay , Fernando Trigoso Mori , Jackelyne Alvarado Zelada , Juan Jose Salinas Barreto , Porfirio Rivera Altamirano , Cesar Torres Miranda , Rocio Anicama Elias , Julio Rivera Alvarez , Juan Pedro Vasquez Matos , Fernando Ayque Rosas , Jesmarina Ledesma Peraza , Andrea Gutarra Palomino , Stephany Vega Centen , Victor Casquero , María Rosa Ortiz Argomedo , Francisco Lapouble , Genaro Llap Unchón , Florangel Patricia Delgado Malaga , Luis Ortega Sotelo , Segundo Gamboa Kcomt , Araceli Villalba Villalba , Nancy Rossana Mendoza Leon , Loreley Raquel Cardenas Alva , Maria Susana Loo Neyra , Cathy Lee Alanguia Chipana , Cintya Maria de Jesus Torres Picón , Natalia Huaytalla Quiroz , Danny Dominguez , Carlos Segura Calle , Jenny Arauco , Luis Ormeño Calderón , Ximena Ghilardi Silva , Miriam Daniela Fernandez Wilson , Joan Elizabeth Gutierrez Maldonado , Cesar Diaz Leon , Waldo Berrocal Anaya , Patricia Chavez Galvez , Prince Pamela Aguilar Gargurevich , Flor de Maria Diaz Castañeda , Carmen Guisse , Erika Ramos Paredes , Jose Luis Apaza Leon , Faye Aguilar Aguilar , Raul Ramirez De La Cruz , Lenny Flores Carbajal , Carlos Mendoza Chiroque , Gladys Johana Sulca Cruzado , Natalia Tovar Gutierrez , Jennifer Sotelo Sanchez , Carolina Paz Soldan , Karina Hernández Córdova , Edgar Fernando Delgado Quinteros , Luz Mery Brito Quevedo , Juan Jose Mendoza Oviedo , Angel Samanez Obeso , Patricia Paredes Espinoza , Johann de Guzman , Raisa Yu , Vlad Cosoreanu , Sebastian Ionescu , Aurel Mironescu , Lucian Vida , Adrian Papa , Roxana Verdeata , Bogdan Gavrila , Liviu Muntean , Marija Lukac , Miona Stojanovic , Djordje Toplicic , Milan Slavkovic , Andjelka Slavkovi , Dragoljub Zivanovic , Ana Kostic , Maja Raicevic , Delphine Nkuliza , Daniel Sidler , Corné de Vos , Elmarie vd Merwe , David Tasker , Omar Khamag , Cecilia Rengura , Thozama Siyotula , Uzair Jooma , Dirk von Delft , Marion Arnold , Hansraj Mangray , Shamaman Harilal , Sanele Madziba , Naveen Wijekoon , Tharanga Gamage , Benedict Paul Bright , Alaa Abdulrahman , Ola Ahmed Abdulmjeed Mohammed , Mohammed Salah , Ahmad Elian Abu Ajwa , Mohammed Morjan , Mohammad Mohannad Batal , Vivian Faks , Mohamad Bassel Mouti , Ahmadfateh Assi , Ahmad Al-Mouakeh , Ahmad Sankari Tarabishi , Ziad Aljarad , Aos Alhamid , Jiraporn Khorana , Wannisa Poocharoen , Sirima Liukitithara , Anan Sriniworn , Wasun Nuntasunti , Monawat Ngerncham , Ratiyaporn Phannua , Kanokrat Thaiwatcharamas , Patchareeporn Tanming , Lassaad Sahnoun , Nahla Kchiche , Roua Abdelmoumen , Egemen Eroğlu , Mehmet Ali Ozen , Hatice Sonay Yalçın Cömert , Mustafa İmamoğlu , Haluk Sarıhan , Şebnem Kader , Mehmet Mutlu , Yakup Aslan , Ahmet Beşir , Şükran Geze , Bahanur Çekiç , Ali Yalcinkaya , Kaan Sönmez , Ramazan Karabulut , Zafer Türkyılmaz , Kıvanç Şeref , Merve Altın , Merve Aykut , M.Eren Akan , Melisa Erdem , Ebru Ergenekon , Canan Türkyılmaz , Elif Keleş , Ali Canözer , Aslı Öztürk Yeniay , Elif Eren , İlknur Banlı Cesur , Zerrin Özçelik , Gökmen Kurt , Mustafa Kurthan Mert , Hatice Kaya , Müge Çelik , Suleyman Cuneyt Karakus , Nazile Erturk , Alev Suzen , Nilay Hakan , Fatih Akova , Mehmet Pasaoglu , Shukurali Eshkabilov , Rustam Z. Yuldashev , Dekhkonboev Avazjon Abdunomonovich , Aliev Makhmudjan Muslimovich , Azad Patel , Chisengo Kapihya , Nicholas Ensar , Ramesh M Nataraja , Mithila Sivasubramaniam , Matthew Jones , Warwick Teague , Sharman Tan Tanny , Gordon Thomas , Kiera Roberts , Soundappan Sannappa Venkatraman , Holger Till , Manon Pigeolet , Martine Dassonville , Anas Shikha , Win Sabai Phyu Win , Zahidah Adlynee Haji Ahmad , Léamarie Meloche-Dumas , Louise Caouette-Laberge , Dickens St-Vil , Ann Aspirot , Nelson Piché , Shahrzad Joharifard , Nadia Safa , Jean-Martin Laberge , Sherif Emil , Pramod Puligandla , Kenneth Shaw , Hussein Wissanji , Eileen Duggan , Elena Guadagno , Maria Consuelo Puentes , Paola Osses Leal , Carolina Mendez Benavente , Michal Rygl , Barbora Trojanová , Klára Berková , Tereza Racková , Ladislav Planka , Jan Škvařil , Radek Štichhauer , Shahad Sabti , Alex Macdonald , Nordeen Bouhadiba , Dorothy Kufeji , Caroline Pardy , Simon Mccluney , Alireza Keshtgar , Rebecca Roberts , Hannah Rhodes , Kate Burns , Robin Garrett-Cox , Kat Ford , Hannah Cornwall , Krithi Ravi , Felicity Arthur , Paul Losty , Tony Lander , Ingo Jester , Suren Arul , Oliver Gee , Giampiero Soccorso , Michael Singh , Max Pachl , Benjamin Martin , Afnan Alzubair , Arun Kelay , Jonathan Sutcliffe , Thomas Middleton , Amy Hughes Thomas , Merina Kurian , Fraser Cameron , Jayaram Sivaraj , Mark C Thomas , Dean Rex , Ceri Jones , Kate Bradshaw , Arnaud Bonnard , Xavier Delforge , Camille Duchesne , Caroline Le Gall , Coralie Defert , Samia Laraqui Hossini , Florent Guerin , Géraldine Hery , Virginie Fouquet-Languillat , Jules Kohaut , Aline Broch , Thomas Blanc , Luke Harper , Thomas Delefortrie , Quentin Ballouhey , Laurent Fourcade , Céline Grosos , Benoit Parmentier , Guillaume Levard , Maria Giovanna Grella , Mariette Renaux Petel , Lucie Grynberg , Olivier Abbo , Sofia Mouttalib , Mélodie Juricic , Aurelien Scalabre , Elodie Haraux , Anke Rissmann , Hardy Krause , Peter Goebel , Ludwig Patzer , Udo Rolle , Andrea Schmedding , Alexandra Antunez-Mora , Bernd Tillig , Sylvester von Bismarck , Patricia Reis Barbosa , Christian Knorr , Domitille Stark , Marco Brunero , Luigi Avolio , Francesco Manni , Matilde Molinelli , Marinella Guazzotti , Alessandro Raffaele , Piero Giovanni Romano , Silvia Cavaiuolo , Gian Battista Parigi , Laszlo Juhasz , Anna Rieth , Arunas Strumila , Rūta Dagilytė , Arunas Liubsys , Pranas Gurskas , Dalius Malcius , Agne Mikneviciute , Asta Vinskaite , Vidmantas Barauskas , Liam Vierboom , Timothy Hall , Spencer Beasley , Lucy Goddard , Mark Stringer , Naveen Weeratunga , Stephen Adams , Jitoko Cama , Marilyn Wong , Sridharan Jayaratnam , Askar Kukkady , Udaya Samarakkody , Sylwester Gerus , Dariusz Patkowski , Agnieszka Wolny , Tomasz Koszutski , Szymon Tobor , Marta Osowicka , Piotr Czauderna , Dariusz Wyrzykowski , Hanna Garnier , Stefan Anzelewicz , Osowicka Marta , Agata Knurowska , Alicja Weiszewsk , Andrzej Grabowski , Wojciech Korlacki , Michal Pasierbek , Przemyslaw Wolak , Aneta Piotrowska , Anna Roszkiewicz , Piotr Kalicińsk , Agata Trypens , Grzegorz Kowalewsk , David Sigalet , Amer Alsaied , Mansour Ali , Ameen Alsaggaf , Alaa Ghallab , Yazeed Owiwi , Ali Zeinelabdeen , Mohamed Fayez , Ahmed Atta , Mazen Zidan , Asaad saleh Radwan , Hanin Shalaby , Reem Abdelbaqi , Khalid Alattas , Yar Kano , Omar Sindi , Abdullah Alshehri , Tariq Altokhais , Fahad Alturki , Mohammad Almosaibli , Dasha Krisanova , Wisam Abbas , Hee-Beom Yang , Hyun-Young Kim , Joong Kee Youn , Jae Hee Chung , Seok Hyeon Cho , In ji Hwang , Ju yeon Lee , Eung song Song , Jenny Arboleda , Mercedes Ruiz de Temiño Bravo , Alexander Siles Hinojosa , Miriam García , Isabel Casal Beloy , Detlef Oliu San Miguel , Maria Elena Molina Vazquez , Verónica Alonso , Alberto Sanchez , Oscar Gomez , Isabel Carrillo , Tomas Wester , Carmen Mesas Burgos , Lars Hagander , Martin Salö , Erik Omling , Niclas Rudolfson , Christina Granéli , Helena Arnadóttir , Emma Grottling , Kate Abrahamsson , Vladimir Gatzinsky , Michaela Dellenmark Blom , Daniel Borbonet , Paul Puglia , Vinicio Jimenez Morejon , Gaston Acuna , Mario Moraes , Jonathan Chan , Pavan Brahmamdam , Alan Tom , Karen Sherer , Brandy Gonzales , Aaron Cunningham , Sanjay Krishnaswami , Reto Baertschiger , Mary Leech , Regan Williams , Lauren Camp , Ankush Gosain , Maria Mora , Bailey D. Lyttle , Jeremy Chang , Lydia McColl Makepeace , Kathryn L Fowler , Sara Mansfield , Erica Hodgman , Chukwubinyelum Amaechi , Alana Beres , Mark N. Pernik , Luke J. Dosselman , Murad Almasri , Sunil Jain , Varun Modi , Marianelly Fernandez Ferrer , John Coon , Joann Gonzalez , Medhavi Honhar , Nensi Ruzgar , Griffin Coghill , Sarah Ullrich , Maija Cheung , Katrine Løfberg , Jodie Greenberg , Kate Davenport , Samir Gadepalli , Sarah Fox , Stephanie Johnson , Mercedes Pilkington , April Hamilton , Nicole Lin , Juan Sola , Yang Yao , Jenna Kylene Davis , Monica Langer , Jonathan Vacek , Fizan Abdullah , Julie Khlevner , William Middlesworth , Marc Levitt , Hira Ahmad , Sabina M Siddiqui , Alex Bowder , Terry Derks , Afua Amoabin Amoabin , Brooke Pinar , Frank Owusu-Sekyere , Benmanseur Saousen , Rasika Naidoo , Azra Karamustafic , Danielle Paula de Oliveira , Sarah Bueno Motter , Jerhy Andrade , Antonín Šafus , Jason Langley , Alexandra Wilke , Corazone Deya , Habib Mansour Murtadi , Mindaugas Berzanskis , Nwachukwu Calistus , Olalekan S. Ajiboye , Michael Felix , Osagie O Olabisi , Seçil Erçin , Teymursha Muradi , Stephen S. Burks , Sergio Lerma , Jillian Jacobson , Calin Calancea , Rafael Valerio-Vazquez , Guigui Sikwete , Owusu Sekyere , Akhona Mbonisweni , Shahnoor Syed , Cho Seok Hyeon , Fatemeh Pajouhandeh , Sheba Mary Pognaa Kunfah

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.



中文翻译:

74 个低收入、中等收入和高收入国家 264 家医院的胃肠道先天性异常死亡率:一项多中心、国际、前瞻性队列研究

背景

先天性异常是全球 5 岁以下儿童死亡的第五大原因。许多胃肠道先天性异常在没有及时获得新生儿外科护理的情况下是致命的,但在低收入和中等收入国家 (LMICs) 对这些疾病的研究很少。我们比较了全球低收入、中等收入和高收入国家七种最常见的胃肠道先天性异常的结果,并确定了与死亡率相关的因素。

方法

我们对 16 岁以下的患者进行了一项多中心、国际前瞻性队列研究,这些患者首次就诊时患有食管闭锁、先天性膈疝、肠闭锁、腹裂、食道外裂、肛门直肠畸形和先天性巨结肠。在 2018 年 10 月至 2019 年 4 月期间连续招募至少 1 个月的患者。我们使用 REDCap 平台收集了有关患者人口统计、临床状态、干预和结果的数据。患者在初次干预后随访 30 天,如果未接受干预,则在入院后 30 天随访。主要结果是所有疾病合并和每种疾病单独的全因住院死亡率,按国家收入状况分层。我们做了一个完整的案例分析。

发现

我们纳入了来自 264 家医院(89高收入国家,中等收入国家 166 个,低收入国家 9 个)在 74 个国家。3849例患者中,男性2231例(58·0%)。出生时的中位胎龄为 38 周(IQR 36-39),出生时的中位体重为 2·8 公斤(2·3-3·3)。所有患者的死亡率分别为:低收入国家 93 名患者中的 37 名(39·8%)、中等收入国家 2860 名患者中的 583 名(20·4%)和高收入国家 896 名患者中的 50 名(5·6%) (所有国家收入组之间的 p<0·0001)。腹裂在国家收入阶层之间的死亡率差异最大(低收入国家 10 个国家中有 9 个 [90·0%],中等收入国家 304 个国家中有 97 个 [31·9%],两个 [1·4%]高收入国家的 139 个;所有国家收入组之间的 p≤0·0001)。与所有患者较高死亡率显着相关的因素包括国家收入状况(低收入高收入国家相比,风险比 2·78 [95% CI 1·88–4·11],p<0·0001;中等收入高收入国家,2·11 [1·59–2·79],p<0·0001),就诊时败血症 (1·20 [1·04–1·40],p=0· 016),美国麻醉医师协会 (ASA) 在主要干预中的得分较高(ASA 4-5 vs ASA 1-2, 1·82 [1·40-2·35], p<0·0001;ASA 3 vsASA 1–2, 1·58, [1·30–1·92], p<0·0001]),未使用手术安全检查表 (1·39 [1·02–1·90], p=0· 035),并且在需要时无法进行通气或肠外营养(通气 1·96,[1·41–2·71],p=0·0001;肠外营养 1·35,[1·05–1·74],p =0·018)。给予肠外营养 (0·61, [0·47–0·79], p=0·0002) 和使用外周插入的中央导管 (0·65 [0·50–0·86], p=0 ·0024) 或经皮中心线 (0·69 [0·48–1·00], p=0·049) 与较低的死亡率相关。

解释

低收入、中等收入和高收入国家之间的胃肠道先天性异常死亡率存在不可接受的差异。改善中低收入国家优质新生儿外科护理的可及性对于实现到 2030 年结束新生儿和 5 岁以下儿童可预防死亡的可持续发展目标 3.2 至关重要。

资金

惠康信托。

更新日期:2021-07-23
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