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This study is a multi-centre, international, prospective cohort study of congenital anomalies to compare outcomes between LMICs and high-income countries (HICs) globally.
Background: Congenital anomalies have risen to become the 5th leading cause of death in children under 5-years of age globally, yet limited literature exists, particularly from low- and middle-income countries (LMICs) where most of these deaths occur.
Aim: To undertake a multi-centre prospective cohort study of congenital anomalies to compare outcomes between LMICs and high-income countries (HICs) globally.
Methods: The Global PaedSurg Research Collaboration will be established consisting of children's surgical care providers from around the world to participate in the study; collaborators will be co-authors of resulting presentations and publication(s). Data will be collected on patients presenting primarily with seven congenital anomalies (oesophageal atresia, congenital diaphragmatic hernia, intestinal atresia, gastroschisis, exomphalos, anorectal malformation and Hirschsprung's disease) for a minimum of one month between Oct 2018 - April 2019. Anonymous data will be collected on patient demographics, clinical status, interventions and outcome. Data will be captured using the secure, online data collection tool REDCap.
The primary outcome will be all-cause in-hospital mortality and the secondary outcomes will be 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. P<0.05 will be deemed significant. Study approval will be sought from all participating centres. Funding has been granted by the Wellcome Trust.
Outcomes: The study aims to be the first large-scale, geographically comprehensive, multi-centre prospective cohort study of a selection of common congenital anomalies to define current management and outcomes globally. Results will be used to aid advocacy and global health prioritisation and inform future interventional studies aimed at improving outcomes.
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| Label | Type | Description | Intervention Names |
|---|---|---|---|
| Oesophageal atresia (OA) +/- tracheo-oesophageal fistula (TOF) |
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| Congenital diaphragmatic hernia (CDH) |
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| Intestinal atresia (IA) |
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| Gastroschisis |
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| Exomphalos |
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| Anorectal malformation (ARM) |
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| Hirschsprung's disease |
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| Name | Type | Description | Arm Group Labels | Other Names |
|---|---|---|---|---|
| Comparisons will be made between LMICs and HICs | Other | Countries will be defined as low, middle or high-income using the World Bank classification. |
|
| Measure | Description | Time Frame |
|---|---|---|
| All-cause, in-hospital mortality | This will include all patients in the study, both those who did not receive an intervention and those that did. For patient's hospitalised for over 30-days following primary intervention, a 30-day post-primary intervention mortality rate will be utilised. For patients who do not receive a primary intervention (conservative generic ward care only) but remain alive and hospitalised at 30-days following primary admission will have this time point used for recording their mortality status for the primary outcome. | Mortality whilst in hospital during primary admission, up to a maximum of 30-days following primary intervention or 30-days following presentation for those who do not receive an intervention and are still in hospital. |
| Measure | Description | Time Frame |
|---|---|---|
| Surgical site-infection | This is defined by the Centre for Disease Control as including one or more of the following within 30-days of surgery: 1) purulent drainage from the superficial or deep (fascia or muscle) incision, but not within the organ/ space component of the surgical site OR 2) at least two of: pain or tenderness; localised swelling; redness; heat; fever; AND the incision is opened deliberately to manage infection, spontaneously dehisces or the clinician diagnoses a SSI (negative culture swab excludes this criterion) OR 3) there is an abscess within the wound (clinically or radiologically detected). |
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Inclusion Criteria:
Exclusion Criteria:
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Neonates, infants and children up to 16-years of age presenting for the first time with one of the seven congenital anomalies in the study to institutions in low-, middle- and high-income countries globally.
All institutions that provide surgical care for the patients being studied can contribute data. Participating institutions will be recruited through convenience sampling with snowballing.
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| Name | Affiliation | Role |
|---|---|---|
| Naomi J Wright, MBChB BSc MRCS DCH MSc | King's College London | Principal Investigator |
| Facility | Status | City | State | ZIP | Country | Contacts |
|---|---|---|---|---|---|---|
| King's College London | London | United Kingdom |
| PubMed Identifier | Type | Citation | Retractions |
|---|---|---|---|
| 31481373 | Derived | Wright NJ; Global PaedSurg Research Collaboration. Management and outcomes of gastrointestinal congenital anomalies in low, middle and high income countries: protocol for a multicentre, international, prospective cohort study. BMJ Open. 2019 Sep 3;9(8):e030452. doi: 10.1136/bmjopen-2019-030452. |
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Following publication of the main study results, the full anonymised dataset will be shared with all collaborators and made publicly available. At no time during presentation or publication of the study will individual collaborators, institutions or countries be independently identifiable. For the main study publication, all data within low, middle and high-income countries will be pooled for analysis. Following publication of the main study, collaborators from within a country can undertake a sub-analysis of the data from their country, but only if all collaborators who have contributed data from that country agree. Individual country names will not be identifiable on the dataset made publicly available - each country will be represented by a random number. The publicly available anonymised data will be identifiable by continent allowing for continental sub-analyses to be undertaken.
Following publication of the main results.
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| Occurring within 30-days of primary intervention |
| Wound dehiscence | All layers of the wound open post-operatively | Occurring within 30-days of primary intervention |
| Need for re-intervention | Need for a second unplanned intervention within 30-days of the primary intervention. | Occurring within 30-days of primary intervention |
| Condition specific complications | OA: pneumonia, mediastinitis, pneumothorax, chylothorax, haemothorax, anastomotic leak, anastomotic stricture, recurrent TOF, other. CDH: air leak, chylothorax, recurrence, adhesional obstruction. IA: anastomotic leak/ stenosis, short-gut, missed additional atresia, adhesive bowel obstruction. Gastroschisis: ischaemic bowel, abdominal compartment syndrome, necrotising enterocolitis. Exomphalos: ruptured sac. ARM: electrolyte disturbance, high stoma output (over 20mls/kg/day), stoma prolapse/ retraction/ herniation, peri-stoma skin breakdown (or perianal if primary reconstruction was undertaken without a covering stoma), anal stenosis. Hirschsprung's disease: enterocolitis, electrolyte disturbance, high stoma output (over 20mls/kg/day), stoma prolapse/ retraction/ herniation, peri-stoma skin breakdown (or perianal if primary pull-through was undertaken without covering stoma), anal stenosis, post-operative obstruction, anastomotic leak. | Occurring within 30-days of primary intervention |
| Condition specific outcome variables | Oesophageal atresia: time to first oral feed (in days) and time to full oral feeds (in days) | Occurring within 30-days of primary intervention |
| Length of hospital stay | In days, including the first and last day. Time from admission to death in patients who do not survive. | Maximum 30-days post-intervention or following presentation for those who do not receive an intervention |
| 30-day post primary intervention mortality | Death within 30-days of primary intervention or 30-days of presentation in those who do not receive an intervention |
| Ventilation requirement | Need for any ventilation (excluding during anaesthetic for interventions) and duration of ventilation in days | Occurring within 30-days of primary intervention or admission for those not receiving an intervention |
| Time to first enteral feed | In days, including the day of the primary intervention and the day when enteral feeds were started. | Occurring within 30-days of primary intervention or admission for those not receiving an intervention |
| Time to full enteral feeds | In days, including the day of the primary intervention and the day when full enteral feeds were achieved. | Occurring within 30-days of primary intervention or admission for those not receiving an intervention |
| Parenteral nutrition requirement | Need for any parenteral nutrition and total duration of parenteral nutrition in days for those who receive it. | Occurring within 30-days of primary intervention or admission for those not receiving an intervention |
| ID | Term |
|---|---|
| D004933 | Esophageal Atresia |
| D065630 | Hernias, Diaphragmatic, Congenital |
| D007409 | Intestinal Atresia |
| D020139 | Gastroschisis |
| D006554 | Hernia, Umbilical |
| D000071056 | Anorectal Malformations |
| D006627 | Hirschsprung Disease |
| ID | Term |
|---|---|
| D004065 | Digestive System Abnormalities |
| D004066 | Digestive System Diseases |
| D004935 | Esophageal Diseases |
| D005767 | Gastrointestinal Diseases |
| D000013 | Congenital Abnormalities |
| D009358 | Congenital, Hereditary, and Neonatal Diseases and Abnormalities |
| D006548 | Hernia, Diaphragmatic |
| D000082122 | Internal Hernia |
| D006547 | Hernia |
| D020763 | Pathological Conditions, Anatomical |
| D013568 | Pathological Conditions, Signs and Symptoms |
| D007410 | Intestinal Diseases |
| D009139 | Musculoskeletal Abnormalities |
| D009140 | Musculoskeletal Diseases |
| D046449 | Hernia, Abdominal |
| D007232 | Infant, Newborn, Diseases |
| D006555 | Hernia, Ventral |
| D008531 | Megacolon |
| D003108 | Colonic Diseases |
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