Gastroschisis survival improvement and early intervention: experience in a developing country
Abstract
Background Gastroschisis is the most common abdominal congenital defect worldwide. While the mortality rate is 5–10% in developed countries, the rate escalates in developing countries due to less access to surgical care, and studies on the condition are scarce. Gastroschisis mortality and morbidity rates in Indonesia are unknown or unclear; little is also known about influencing factors that reveal epidemiological characteristics.
Objective To identify the rate and factors associated with gastroschisis mortality in Indonesia.
Methods A retrospective cohort study was conducted at Dr. Cipto Mangunkusumo Hospital, which included neonates with gastroschisis who underwent defect closure surgery from January 2015 to September 2020. We explored possible influencing risk factors, including gestational age, birth weight, number of surgeries, age at closure, and presence of gastroschisis complications. Bivariate analysis was done using Chi-square or Fisher’s test.
Results Of 49 neonates with gastroschisis, 42 were included in the study. Seven neonates were excluded due to incomplete medical records. The mortality rate of gastroschisis based on our data was 69%. The age at closure (<1 day) was significantly associated with lower mortality rate (P=0.005). In contrast, other factors, including gestational age, birth weight, number of surgeries, and the presence of complicated gastroschisis were not the risk factors of gastroschisis mortality.
Conclusion The mortality rate of gastroschisis is high in Indonesia, specifically at Dr. Cipto Mangunkusumo Hospital. Immediate closure is significantly associated with a decreased likelihood of death. Awareness, diagnosis, and efficient referral of gastroschisis from remote areas to a tertiary facility must be encouraged to reduce the high mortality rate.
References
2. Eggink BH, Richardson CJ, Malloy MH, Angel CA. Outcome of gastroschisis: a 20-year case review of infants with gastroschisis born in galveston, texas. J Pediatr Surg. 2006;41:1103-8. DOI: https://doi.org/10.1016/j.jpedsurg.2006.02.008
3. Global PaedSurg Research Collaboration. Mortality from gastrointestinal congenital anomalies at 264 hospitals in 74 low-income, middle-income, and high-income countries: a multicentre, international, prospective cohort study. Lancet. 2021;398:325-39. DOI: https://doi.org/10.1016/s0140-6736(21)00767-4
4. Wang Y, Liu G, Canfield MA, Mai CT, Gilboa SM, Meyer SE, et al. Racial/ethnic differences in survival of United States children with birth defects: a population-based study. J Pediatr. 2015;166:819-26.e1-2. DOI: https://doi.org/10.1016/j.jpeds.2014.12.025
5. Fillingham A, Rankin J. Prevalence, prenatal diagnosis and survival of gastroschisis. Prenat Diagn. 2008;28:1232-7. DOI: https://doi.org/10.1002/pd.2153
6. Nembhard WN, Waller DK, Sever LE, Canfield MA. Patterns of first-year survival among infants with selected congenital anomalies in Texas, 1995-1997. Teratology. 2001;64:267-75. DOI: https://doi.org/10.1002/tera.1073
7. Islam S. Congenital abdominal wall defects. In: Holcomb GW III, Murphy JP SPS, editor. Holcomb and Ashcraft’s Pediatric Surgery. 7th edition ed. Philadelphia: Elsevier Saunders;2020. p. 763-79.
8. Niles SGM, Mitchell-Fearon K, Gill MI, DeSouza CJ, Fearon IC, Abel CA, et al. Mortality-related factors in gastroschisis - a Jamaican perspective. J Pediatr Surg. 2017;52:530-33. DOI: https://doi.org/10.1016/j.jpedsurg.2016.10.045
9. Clark RH, Walker MW, Gauderer MW. Factors associated with mortality in neonates with gastroschisis. Eur J Pediatr Surg. 2011;21:21-4. DOI: https://doi.org/10.1055/s-0030-1262791
10. Sekabira J, Hadley GP. Gastroschisis: a third world perspective. Pediatr Surg Int. 2009;25:327-9. DOI: https://doi.org/10.1007/s00383-009-2348-4
11. Vilela PC, Ramos De Amorim MM, Falbo GH, Santos LC. Risk factors for adverse outcome of newborns with gastroschisis in a Brazilian hospital. J Pediatr Surg. 2001;36:559-64. DOI: https://doi.org/10.1053/jpsu.2001.22282
12. Vo LU, Langlois PH. Time trends in prevalence of gastroschisis in Texas, 1999 to 2011: subgroup analyses by maternal and infant characteristics. Birth Defects Res A Clin Mol Teratol. 2015;103:928-40. DOI: https://doi.org/10.1002/bdra.23438
13. Quinn JA, Munoz FM, Gonik B, Frau L, Cutland C, Mallett-Moore T, et al. Preterm birth: case definition & guidelines for data collection, analysis, and presentation of immunisation safety data. Vaccine. 2016;34:6047-56. DOI: https://doi.org/10.1016/j.vaccine.2016.03.045
14. Cutland CL, Lackritz EM, Mallett-Moore T, Bardají A, Chandrasekaran R, Lahariya C, et al. Low birth weight: case definition & guidelines for data collection, analysis, and presentation of maternal immunization safety data. Vaccine. 2017;35:6492-500. DOI: https://doi.org/10.1016/j.vaccine.2017.01.049
15. Gupta R, Cabacungan ET. Outcome of neonates with gastroschisis at different gestational ages using a national database. J Pediatr Surg. 2018;53:661-65. DOI: https://doi.org/10.1016/j.jpedsurg.2017.07.015
16. Overcash RT, Deugarte DA, Stephenson ML, Gutkin RM, Norton ME, Parmar Sima,et al. Factors associated with gastroschisis outcomes. Obstet Gynecol. 2014;124:551-7. DOI: https://doi.org/10.1097/aog.0000000000000425
17. Gonzalez DO, Cooper JN, St Peter SD, Minneci PC, Deans KJ. Variability in outcomes after gastroschisis closure across U.S. children's hospitals. J Pediatr Surg. 2018;53:513-20. DOI: https://doi.org/10.1016/j.jpedsurg.2017.04.012
18. Skarsgard ED, Claydon J, Bouchard S, Kim PCW, Lee SK, Laberge JM, et al. Canadian Pediatric Surgical Network: a population-based pediatric surgery network and database for analyzing surgical birth defects. The first 100 cases of gastroschisis. J Pediatr Surg. 2008;43:30-4; discussion 34. DOI: https://doi.org/10.1016/j.jpedsurg.2007.09.011
19. Skarsgard ED. Management of gastroschisis. Curr Opin Pediatr. 2016;28:363-9. DOI: https://doi.org/10.1097/mop.0000000000000336
20. Baird R, Puligandla P, Skarsgard E, Laberge JM. Infectious complications in the management of gastroschisis. Pediatr Surg Int. 2012;28:399-404. DOI: https://doi.org/10.1007/s00383-011-3038-6
21. Carnaghan H, Baud D, Lapidus-Krol E, Ryan G, Shah PS, Pierro A, et al. Effect of gestational age at birth on neonatal outcomes in gastroschisis. J Pediatr Surg. 2016;51:734-8. DOI: https://doi.org/10.1016/j.jpedsurg.2016.02.013
22. Hawkins RB, Raymond SL, St Peter SD, Downard CD, Qureshi FG, Renaud E, et al. Immediate versus silo closure for gastroschisis: results of a large multicenter study. J Pediatr Surg. 2020;55:1280-5. DOI: https://doi.org/10.1016/j.jpedsurg.2019.08.002
23. O'connell RV, Dotters-Katz SK, Kuller JA, Strauss RA. Gastroschisis: a review of management and outcomes. Obstet Gynecol Surv. 2016;71:537-44. DOI: https://doi.org/10.1097/ogx.0000000000000344
24. Owen A, Marven S, Johnson P, Kurinczuk J, Spark P, Draper ES, et al. Gastroschisis: a national cohort study to describe contemporary surgical strategies and outcomes. J Pediatr Surg. 2010;45:1808-16. DOI: https://doi.org/10.1016/j.jpedsurg.2010.01.036
25. Molik KA, Gingalewski CA, West KW, Rescorla FJ, Scherer III LR, Engum SA, et al. Gastroschisis: a plea for risk categorization. J Pediatr Surg. 2001;36:51-5. DOI: https://doi.org/10.1053/jpsu.2001.20004
26. Jager LC, Heij HA. Factors determining outcome in gastroschisis: clinical experience over 18 years. Pediatr Surg Int. 2007;23:731-6. DOI: https://doi.org/10.1007/s00383-007-1960-4
27. Arnold MA, Chang DC, Nabaweesi R, Colombani PM, Bathurst MA, Mon KS, et al. Risk stratification of 4344 patients with gastroschisis into simple and complex categories. J Pediatr Surg. 2007;42:1520-5. DOI: https://doi.org/10.1016/j.jpedsurg.2007.04.032
28. Bergholz R, Boettcher M, Reinshagen K, Wenke K. Complex gastroschisis is a different entity to simple gastroschisis affecting morbidity and mortality-a systematic review and meta-analysis. J Pediatr Surg. 2014;49:1527-32. DOI: https://doi.org/10.1016/j.jpedsurg.2014.08.001
29. Raymond SL, Hawkins RB, St Peter SD, Downard CD, Qureshi FG, Renaud E, et al. Predicting morbidity and mortality in neonates born with gastroschisis. J Surg Res. 2020;245:217-24. DOI: https://doi.org/10.1016/j.jss.2019.07.065
30. Mendez R, Watane A, Farhangi M, Cavuoto KM, Leith T, Budree S, et al. Gut microbial dysbiosis in individuals with sjögren’s syndrome. Microbial Cell Factories. 2020;19:90. DOI: https://doi.org/10.1186/s12934-020-01348-7
31. Pastor AC, Phillips JD, Fenton SJ, Meyers RL, Lamm AW, Raval MV, et al. Routine use of a silastic spring-loaded silo for infants with gastroschisis: a multicenter randomized controlled trial. J Pediatr Surg. 2008;43:1807-12. DOI: https://doi.org/10.1016/j.jpedsurg.2008.04.003
32. Petrosyan M, Sandler AD. Closure methods in gastroschisis. Semin Pediatr Surg. 2018;27:304-8. DOI: https://doi.org/10.1053/j.sempedsurg.2018.08.009
Copyright (c) 2024 Tri Hening Rahayatri, Harsya Dwindaru Gunardi, Rusdah Binti Muhammad Amin, Riana Pauline Tamba
This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License.
Authors who publish with this journal agree to the following terms:
Authors retain copyright and grant the journal right of first publication with the work simultaneously licensed under a Creative Commons Attribution License that allows others to share the work with an acknowledgement of the work's authorship and initial publication in this journal.
Authors are able to enter into separate, additional contractual arrangements for the non-exclusive distribution of the journal's published version of the work (e.g., post it to an institutional repository or publish it in a book), with an acknowledgement of its initial publication in this journal.
This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License.
Accepted 2024-06-10
Published 2024-06-10
