- Email: [email protected]
Assessment of gastroschisis risk factors in Egypt
YJPSU-59454; No of Pages 4 Journal of Pediatric Surgery xxx (xxxx) xxx
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Assessment of gastroschisis risk factors in Egypt☆ Aly Shalaby a,⁎, Alaa Obeida a, Dalia Khairy b, Khaled Bahaaeldin a a b
Department of Pediatric Surgery, Cairo University Specialized Pediatric Hospital Department of Pediatrics, Cairo University Specialized Pediatric Hospital
a r t i c l e
i n f o
Article history: Received 17 October 2019 Accepted 26 October 2019 Available online xxxx Key words: Gastroschisis Abdominal wall defect Low income country Egypt Africa
a b s t r a c t Aim: Mortality in infants born with gastroschisis (GS) in low-to-middle-income countries (LMICs) is high. This study aimed to assess factors which might affect outcome in Egypt in order to improve survival. Methods: A prospective study over a 15-month duration was completed. Variables assessed covered patient, maternal, antenatal, treatment, and complications. The Gastroschisis Prognostic Score (GPS) was used to predict outcome. A validated questionnaire was used to assess socioeconomic status. The main outcome was mortality. Results: Twenty-four cases were studied. Median gestational age was 37 (26–40) weeks, and 9 (38%) were preterm. Mortality occurred in 15 (62%) infants. Median transfer time was 8 (1.5–35) hours, and 64% survived if transferred before 8 h. Median maternal age was 20 (16–27) years. All families were of a low or very-low socioeconomic level. Only 25% had antenatal scans. Most cases were simple GS, and only 3 (12.5%) were complex GS. Median length of stay was 14 (1–52) days, TPN duration was 12 (0–49) days, and days to full feeds was 5 (3–11) days. The GPS score ranged from 0 to 6 in the studied cases and negatively correlated with outcome (rS = −0.98; p = 0.03). Conclusion: The mortality of GS in Egypt is very high, mainly due to sepsis and prematurity. Young maternal age and poor socioeconomic status are linked to GS. The GPS is a good indicator of morbidity and mortality in a LMIC setting. Survival improved with better resuscitation and strict management protocols. More effort is needed to improve antenatal detection, and transfer time should be ideally below 8 h. Level of Evidence: Level IV. © 2019 Elsevier Inc. All rights reserved.
Gastroschisis (GS) results from herniation of the intestines through a right-sided (usually) abdominal wall defect [1–3]. The estimated incidence of GS varies from country to country yet the prevalence has generally been rising worldwide [4,5]. A single center report from Ethiopia calculates that abdominal wall defects made up 1.3% of all pediatric surgery conditions [6] and another report from South Africa put GS at 15% of neonatal surgery admissions [7]. GS is readily detected via antenatal ultrasonography and this may affect outcome [8]. Survival rates in developed countries such as the UK and USA may be N 95% [9] and morbidity is largely related to slow restoration of gastrointestinal function with long-term sequelae being rare [10]. By contrast, the experience in Low-to-Middle income countries (LMICs) is plagued by high mortality rates, in general, in neonates with congenital anomalies [11,12] and specifically in GS sometimes reaching 80% to 100% [13–16]. Most deaths may be attributed to sepsis [15] which may be exacerbated by long delays in transfer to a surgical center [14].
☆ The authors declare no conflict of interest.No funding was received for this study. ⁎ Corresponding author at: Department of Pediatric Surgery, Cairo University Specialized Pediatric Hospital, Ali Ibrahim Street, Mounira 11241, Cairo, Egypt. Tel.: + 20 1223905599; fax: +20 2 23649281. E-mail address: [email protected] (A. Shalaby).
There are several determinants of outcome in GS such as the presence or absence of intestinal complications, namely atresia, necrosis, and perforation. GS cases can be classified into complex GS and simple GS with the former having consistently worse outcomes [17,18]. In the absence of intestinal complications, it is not clear why some simple GS patients have worse outcomes than others, with longer periods of bowel dysfunction [19,20]. Other variables which may be relevant include gestational age, mode of delivery, age at closure, closure method, the degree of bowel matting, necrotizing enterocolitis (NEC), cardiac anomalies, and lung hypoplasia [21,22]. This pilot study aimed to describe and assess, where possible, the impact of these different factors on our local population in an attempt to implement the correct strategies to improve survival. 1. Methods Prospective cohort study including all infants born with GS presenting to the Cairo University Specialized Pediatric Hospital (CUSPH), Surgical Neonatal Intensive Care Unit (SNICU) during the period from July 2017 till September 2018. Patient variables included gender, gestational age, gestational weight, consanguinity, birth place, transfer-time and age at surgical intervention. A Gastroschisis Prognostic Score (GPS) was calculated [22],
https://doi.org/10.1016/j.jpedsurg.2019.10.027 0022-3468/© 2019 Elsevier Inc. All rights reserved.
Please cite this article as: A. Shalaby, A. Obeida, D. Khairy, et al., Assessment of gastroschisis risk factors in Egypt, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.10.027
2
A. Shalaby et al. / Journal of Pediatric Surgery xxx (xxxx) xxx
and outcome recorded as live discharge or death. Maternal variables included age, education, place of residence, delivery mode, smoking, alcohol consumption, and illicit drug use. Antenatal variables included frequency of antenatal scans and the presence of any associated anomaly. Treatment variables included type of surgical closure, duration of mechanical ventilation, duration of SNICU admission, duration of parenteral nutrition (PN) and time to full feeds. Finally, the database included postnatal complications: the incidence of central line-associated blood stream infection (CLABSI), Surgical Site Infections (SSIs), and necrotising enterocolitis (NEC). The Clavien Dindo Classification [24] was used to classify post-operative complications. Parental socio-economic status was calculated using the El-Gilany et al. survey [23]. Maximum score is 84. Families with a score of b22 are classified as a very low socioeconomic level, those scoring 23–43 are of a low socioeconomic level, those scoring 44–65 are of an intermediate socioeconomic level, while those scoring 66–84 are of a high socioeconomic level [23]. A standardized management protocol was implemented 4 months into the study period including naso-gastric tube, covering of exposed intestines with cling film and correction of any arterial in-flow or venous out-flow problems. IV fluids were given as a 20 ml/kg bolus of normal saline and 3 ml/kg bolus of 10% Dextrose and maintenance of 10% Dextrose in normal saline at a volume of 120 ml/kg/day. Overtransfusion was actively avoided. Ampicillin/sulbactam and an aminoglycoside was used prophylactically. Surgical closure was done in a sterile environment (preferably theater) even for sutureless closure.
standardized protocol described above. During the first 4 months, survival was 12.5% (1/8) and increased to 50% (8/16 cases) though this did not reach statistical significance (p = 0.17). 2.2. Maternal variables Maternal age was 20 (16–27) years and 12 (50%) were b 20 years. Education level varied from no schooling [n = 6 (25%), school (secondary) education; n = 14(58%) to a college degree, n = 4 (17%)]. Socioeconomic status of the families varied from very low socioeconomic standard (n = 4, 17%) to low standard (n = 20, 83%). Twelve families (50%) lived in the Cairo/Giza governorates; five (21%) from Beni Sueif, four (17%) from Fayoum, three (12%) from Menofia, Minya, and Qalyubia (one from each). Delivery was by normal vaginal delivery (n = 13, 54%) or caesarian section (n = 11, 46%). None of the mothers gave a history of smoking, alcohol consumption, or illicit drug use. 2.3. Antenatal variables Six mothers (25%) had N3 antenatal ultrasound scans, five cases (21%) had them done for three times, two cases (8%) had them twice, six cases (25%) had a scan done once, and five cases (21%) had no scans at all. There were no associated anomalies in the cases scanned. 2.4. Treatment variables
1.1. Data analysis Descriptive analysis tools that were used for single variables are median and range. Analytical tests were done using correlation coefficient, regression analysis, Fisher exact test and ANOVA tests to correlate different variables with the outcome. Significance was accorded at a P value of b0.05. Data are reported as median (range). 2. Results 2.1. Patient variables Twenty four [n = 15 (62%) male] cases of GS presented during the 15-month study period (July 2017–September 2018) and represented 7% of neonatal surgical admissions. Median gestational age was 37 (26–40) weeks. Birth weight was 2.4 (1–3.5) kg. Nineteen infants (79%) were from non-consanguineous marriage and five (21%) from consanguineous marriage. Eleven cases (46%) were inborn at the Cairo University Hospital, with the others born elsewhere [public hospital, n = 4 (17%); private hospital, n = 4 (17%); private clinic, n = 4 (17%) and one (4.1%) was delivered at home]. Transfer-time was 8 (1.5–35) hours. Age at surgical intervention was 15 (5–36) hours. Ten (42%) infants had surgical intervention in b12 h, 10 (42%) were operated on within 12–24 h and 4 (17%) N24 h. Nine infants (37%) were discharged from the unit alive and 15 infants died (63%) died. The outcome of the first 4 months of the study duration (July to October 2017) was compared to the subsequent months (November 2017 to September 2018) after implementing the
The cohort was composed of simple (n = 21) and complex (n = 3) GS cases. The type of surgical closure was staged surgical silo (n = 10, 42%), primary surgical closure (n = 9, 38%), skin closure alone (n = 3, 12%) and primary sutureless closure (n = 1, 4%). One infant died before reaching theater (Fig. 1). Infants with simple GS fared better especially the ones who had a primary fascial closure (Fig. 1). Complex GS cases didn't survive regardless of choice of surgery (Fig. 1). Length of stay (LOS) in the SNICU was 14 (1–52) days. Surviving infants stayed 20 (12–52) days, non-survivors stayed 9 (1–50) days. LOS for primary closure was 12 (1–37) days, and staged silo was 18 (5–52) days. TPN was given to n = 22 (92%) cases for a duration of 12 (0–49) days. TPN days for staged closure cases were 16 (4–49) days, longer than primary closure cases of 10 (1–29) days. Only 9 (37%) infants actually started enteral feeding with the median days to full feeds (DFF) being 5 (3–11) days. 2.5. Postnatal complications The incidence of CLABSI was 4% (1/24 cases) and SSI was 33% (8/24 cases). One case (4%) was diagnosed with NEC. Clavien Dindo classification of complications included grade II (n = 9, 37%) and grade V (n = 15, 62%). Four infants died within 48 h. 2.5.1. Gastroschisis prognostic score (GPS) Nine (38%) infants had a GPS score of 0, 11 cases (46%) had a score of one, two cases (8%) had a score of four and two cases (8%) had a score of six. Of the two cases with GPS 4, one had closed GS with bowel necrosis
Fig. 1. Types of GS, surgery and outcome. GS: Gastroschisis.
Please cite this article as: A. Shalaby, A. Obeida, D. Khairy, et al., Assessment of gastroschisis risk factors in Egypt, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.10.027
A. Shalaby et al. / Journal of Pediatric Surgery xxx (xxxx) xxx Table 1 Outcome related to transfer time. Transfer time
Alive
Dead
b8 hours ≥8 hours
7 2 9
4 11 15
11 13 24
Alive
Dead
Total
5 4 0 9
5 6 4 15
10 10 4 24
p = 0.03
Table 2 Outcome compared to age at surgery. Outcome
b12 hours 12–24 hours N 24 hours p = 0.29
from the duodeno-jejunal junction till the colon, while the other had severe bowel matting. The former case died, while the latter survived. Both cases with GPS 6 had jejunal atresia associated with severe bowel matting and both died. The relationship between the GPS and outcome showed a strong negative correlation (rS = −0.98; p = 0.03). 2.6. Other factors Eight hours was used as a cut-off to compare the “early” versus the “delayed” transfer. Out of the 11 “early” cases transferred before 8 h, 64% (n = 7) survived. In contrast, out of the 13 “delayed” cases only 15% (n = 2) survived (p = 0.03) (Table 1). Survival was 50% for infants undergoing surgery b12 h of life, 40% for intervention at 12-24 h and 0% at ≥24 h (p = 0.28). (See Table 2.) Outborn cases (n 13, 54%) were transferred from distances varying from 10 to 256 km. The number of inborn and outborn cases was similar (n = 11 vs n = 13). Five inborn and 10 outborn died but this didn't reach statistical significance (p = 0.1). Ventilated cases (n = 3, 18%) survived compared to (n = 6, 86%) non ventilated ones (p = 0.003). 3. Discussion We characterized our series as typically arising from a nonconsanguineous marriage with a slight male predominance [25]. Most were full term and only a third preterm [26,27]. We did have a high overall mortality of N 60%, especially obvious in the premature and in this regard is similar to experience in other LMICs [28]. The GPS [22] used in this study was very easy to use and reliably predicted the outcome. The latter has been echoed by Puligandla et al. [29]. Most of our survivors reached the unit before 8 h [Fig. 2]. The literature is not clear as to what constitutes a delay in transfer. Considering
3
our findings, we propose defining “delayed transfer” as anything at or above 8 h. It has been reported that transport and bowel stabilization times could not predict outcome but were independently predictive of DFF, time on TPN and LOS [30]. Furthermore, an inborn status does not affect outcome but favorably impacts the time to surgery, DFF and the LOS [31]. There are reports of GS clustering and association with poor socioeconomic status [32–34]. In our study, the majority of families were of low or very low status. None of the mothers in our study had a history of smoking, alcohol consumption, or illicit drug use, despite many studies identifying them as risk factors for GS [25,35]. There was poor compliance with antenatal care visits in our study. This could be explained in part by the families' low socioeconomic status. In a broader context only 53% of pregnant women in Egypt seek antenatal care [36]. Furthermore, the drop-off in the number of antenatal visits with increasing parity is steep, dropping from 41% for first births to 13% for sixth or higher-order births. The gap between the richest 20% and the poorest 20% for use of antenatal care in Egypt is large [36]. The ventilated cases in this study had a significantly worse outcome. Ventilated infants in LMIC countries have a mortality rate in the range of 40–60% [37]. GS often need mechanical ventilation at a certain point of the management process. There is no high-level evidence that reduction without anesthesia or an abdominal incision reduces the risks that may occur from ventilation [38]. Low-risk (simple) GS have an ipso facto shorter LOS compared to high-risk (complex) cases [29]. Staged silo infants in our study had a longer LOS which has been independently associated with an increased LOS [40]. Our staged closure cases were on TPN for longer, in keeping with other reports in the literature [41]. TPN cholestasis can reach up to 25% of cases with a higher incidence in pre-terms [42]; however, this was not observed in our study. In the current study 42% started enteral feeding, 90% of whom reached full volumes and survived, while one case (10%) died from sepsis. Our median DFF of 5 days is slightly earlier than the 7 days purported to offer the best outcome, stop TPN and decrease LOS [43]. We reported only one case (4.17%) with NEC. Its incidence after GS repair may reach up to 20% [44]. Unlike the classic associations of prematurity, low birth weight, and cardiac abnormalities with NEC, GS NEC seems to increase if the neonate is outborn or has developed CLABSI [39]. In conclusion, despite GS being an isolated pathology with no comorbidities, there are many extraneous factors affecting its outcome. Its management has been proposed as a reliable indicator for the maturity of neonatal surgical care services in a country [28]. There is a scarcity of published literature on GS from our region and our pilot study has painted a far-from-perfect picture. To the best of our knowledge, it is the first of its kind to outline the factors affecting GS in Egypt: as elsewhere in the world, a young maternal age and poor socioeconomic status are linked to GS. Survival improves with better resuscitation, strict management protocols and effective infection control. More effort is needed to improve antenatal detection. Finally, efficient referral networks need to cut the transfer time below 8 h.
Fig. 2. Outcome of GS related to transfer time. Dashed line at 8 hours.
Please cite this article as: A. Shalaby, A. Obeida, D. Khairy, et al., Assessment of gastroschisis risk factors in Egypt, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.10.027
4
A. Shalaby et al. / Journal of Pediatric Surgery xxx (xxxx) xxx
References [1] Wilson RD, Johnson MP. Congenital abdominal wall defects: an update. Fetal Diagn Ther 2004;19:385–98. https://doi.org/10.1159/000078990. [2] David AL, Tan A, Curry J. Gastroschisis: Sonographic diagnosis, associations, management and outcome. Prenat Diagn 2008;28:633–44. https://doi.org/10.1002/pd.1999. [3] Gow KW, Bhatia A, Saad DF, et al. Left-sided gastroschisis. Am Surg 2006;72:637–40. [4] International Clearinghouse for Birth Defects Surveillance and Research Centre. ICBDSR annual report 2014. International Clearinghouse for Birth Defects Surveillance and Research Centre, 2014. http://www.icbdsr.org/wp-content/annual_ report/Report2014.pdf [accessed 02 August 2019]. [5] Parker SE, Mai CT, Canfield MA, et al. Updated national birth prevalence estimates for selected birth defects in the United States, 2004–2006. Birth Defects Res Part A - Clin Mol Teratol 2010; 88(12):1008–16. doi:https://doi.org/10.1002/bdra.20735. [6] Derbew M. Pediatric surgery in eastern Africa: the unmet need. J Pediatr Surg 2018; 54:21–6. https://doi.org/10.1016/j.jpedsurg.2018.10.028. [7] Sekabira J, Hadley GP. Gastroschisis: a third world perspective. Pediatr Surg Int 2009;25:327–9. https://doi.org/10.1007/s00383-009-2348-4. [8] Bond SJ, Harrison MR, Filly RA, et al. Severity of intestinal damage in gastroschisis: correlation with prenatal sonographic findings. J Pediatr Surg 1988;23:520–5. https://doi.org/10.1016/S0022-3468(88)80360-9. [9] Holland AJ, Walker K, Badawi N. Gastroschisis: an update. Pediatr Surg Int 2010;26: 871. https://doi.org/10.1007/s00383-010-2679-1. [10] Langer JC, Longaker MT, Crombleholme TM, et al. Etiology of intestinal damage in gastroschisis. I: effects of amniotic fluid exposure and bowel constriction in a fetal lamb model. J Pediatr Surg 1989;24:992–7. https://doi.org/10.1016/S0022-3468 (89)80200-3. [11] Murray CJL, Vos T, Lozano R, et al. Disability-adjusted life years (DALYs) for 291 diseases and injuries in 21 regions, 1990–2010: A systematic analysis for the Global Burden of Disease Study 2010. Lancet 2012; 380(9859):2197–223. doi:https://doi. org/10.1016/S0140-6736(12)61689-4. [12] Black RE, Cousens S, Johnson HL, et al. Global, regional, and national causes of child mortality in 2008: a systematic analysis. Lancet 2010; 375(9730):1969–87. doi: https://doi.org/10.1016/S0140-6736(10)60549-1. [13] Arnold M. Is the incidence of gastroschisis rising in South Africa in accordance with international trends? Sth Afr J Surg 2004;42:86–8. [14] Ameh EA, Chirdan LB. Ruptured exomphalos and gastroschisis: a retrospective analysis of morbidity and mortality in Nigerian children. Pediatr Surg Int 2000;16:23–5. https://doi.org/10.1007/s003830050006. [15] Vilela PC, Ramos De Amorim MM, Falbo GH, et al. Risk factors for adverse outcome of newborns with gastroschisis in a Brazilian hospital. J Pediatr Surg 2001;36:559–64. https://doi.org/10.1053/jpsu.2001.22282. [16] Askarpour S, Ostadian N, Javaherizadeh H, et al. Omphalocele, gastroschisis: Epidemiology, survival, and mortality in Imam Khomeini Hospital, Ahvaz-Iran. Pol Prz Chir Polish J Surg 2012; 84: 82–5. doi:https://doi.org/10.2478/v10035-012-0013-4. [17] Bergholz R, Boettcher M, Reinshagen K, et al. 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. https://doi.org/10.1016/j. jpedsurg.2014.08.001. [18] Emil S, Canvasser N, Chen T, et al. Contemporary 2-year outcomes of complex gastroschisis. J Pediatr Surg 2012;47:1521–8. https://doi.org/10.1016/j.jpedsurg. 2011.12.023. [19] Baird R, Eeson G, Safavi A, et al. Institutional practice and outcome variation in the management of congenital diaphragmatic hernia and gastroschisis in Canada: a report from the Canadian pediatric surgery network. J Pediatr Surg 2011;46:801–7. https://doi.org/10.1016/j.jpedsurg.2011.02.008. [20] Lusk LA, Brown EG, Overcash RT, et al. Multi-institutional practice patterns and outcomes in uncomplicated gastroschisis: a report from the University of California Fetal Consortium (UCfC). J Pediatr Surg 2014;49:1782–6. https://doi.org/10.1016/j. jpedsurg.2014.09.018. [21] Arnold MA, Chang DC, Nabaweesi R, et al. Development and validation of a risk stratification index to predict death in gastroschisis. J Pediatr Surg 2007;42:950–6. https://doi.org/10.1016/j.jpedsurg.2007.01.028. [22] Cowan KN, Puligandla PS, Laberge JM, et al. The gastroschisis prognostic score: reliable outcome prediction in gastroschisis. J Pediatr Surg 2012;47:1111–7. https://doi.
org/10.1016/j.jpedsurg.2012.03.010. [23] El-Gilany A, El-Wehady A, El-Wasify M. Updating and validation of the socioeconomic status scale for health research in Egypt. East Mediterr Heal J 2012; 18: 962–68. doi:10.26719/2012.18.9.962. [24] Clavien PA, Barkun J, De Oliveira ML, et al. The Clavien-Dindo classification of surgical complications: five-year experience. Ann Surg 2009;250:187–96. https://doi.org/ 10.1097/SLA.0b013e3181b13ca2. [25] Hougland KT, Hanna AM, Meyers R, et al. Increasing prevalence of gastroschisis in Utah. J Pediatr Surg 2005;40:535–40. https://doi.org/10.1016/j.jpedsurg.2004.11. 026. [26] Abdel-Latif ME, Bolisetty S, Abeywardana S, et al. Mode of delivery and neonatal survival of infants with gastroschisis in Australia and New Zealand. J Pediatr Surg 2008; 43:1685–90. https://doi.org/10.1016/j.jpedsurg.2008.03.053. [27] Lausman AY, Langer JC, Tai M, et al. Gastroschisis: what is the average gestational age of spontaneous delivery? J Pediatr Surg 2007; 42: 1816–21. doi:https://doi. org/10.1016/j.jpedsurg.2007.07.005. [28] Ford K, Poenaru D, Moulot O, et al. Gastroschisis: bellwether for neonatal surgery capacity in low resource settings? J Pediatr Surg 2016;51:1262–7. https://doi.org/10. 1016/j.jpedsurg.2016.02.090. [29] Puligandla PS, Baird R, Skarsgard ED, et al. Outcome prediction in gastroschisis – the gastroschisis prognostic score (GPS) revisited. J Pediatr Surg 2017;52:718–21. https://doi.org/10.1016/j.jpedsurg.2017.01.017. [30] Bucher BT, Mazotas IG, Warner BW, et al. Effect of time to surgical evaluation on the outcomes of infants with gastroschisis. J Pediatr Surg 2012;47:1105–10. https://doi. org/10.1016/j.jpedsurg.2012.03.016. [31] Savoie KB, Huang EY, Aziz SK, et al. Improving gastroschisis outcomes: does birth place matter? J Pediatr Surg 2014;49:1771–5. https://doi.org/10.1016/j.jpedsurg. 2014.09.019. [32] Chircor L, Mehedinţi R, Hîncu M. Risk factors related to omphalocele and gastroschisis. Rom J Morphol Embryol 2009;50:645–9. [33] Root ED, Meyer RE, Emch ME. Evidence of localized clustering of gastroschisis births in North Carolina, 1999–2004. Soc Sci Med 2009;68:1361–7. https://doi.org/10. 1016/j.socscimed.2009.01.034. [34] Rankin J, Pattenden S, Abramsky L, et al. Prevalence of congenital anomalies in five British regions, 1991–99. Arch Dis Child Fetal Neonatal Ed 2005;90:F374–9. https://doi.org/10.1136/adc.2003.047902. [35] Lam PK, Torfs CP. Interaction between maternal smoking and malnutrition in infant risk of gastroschisis. Birth Defects Res Part A - Clin Mol Teratol 2006;76:182–6. https://doi.org/10.1002/bdra.20238. [36] Abou-Zahr I, Lidia C, Wardlaw Tessa M. Antenatal Care in Developing Countries Promises, achievements and missed opportunities. WHO Libr Cat Data Antenatal 2003. doi:25/12/2014. [37] Kambarami R, Chidede O, Chirisa M. Neonatal intensive care in a developing country: outcome and factors associated with mortality. Cent Afr J Med 2000;46:205–7. [38] Davies MW, Kimble RM, Woodgate PG. Ward reduction without general anaesthesia versus reduction and repair under general anaesthesia for gastroschisis in newborn infants. Cochrane Database Syst Rev 2002;3. https://doi.org/10.1002/14651858. cd003671. [39] Youssef F, Laberge JM, Puligandla P, et al. Determinants of outcomes in patients with simple gastroschisis. J Pediatr Surg 2017;52:710–4. https://doi.org/10.1016/j. jpedsurg.2017.01.019. [40] Murthy K, Evans JR, Bhatia AM, et al. The association of type of surgical closure on length of stay among infants with gastroschisis born ≥34 weeks' gestation. J Pediatr Surg 2014;49:1220–5. https://doi.org/10.1016/j.jpedsurg.2013.12.020. [41] Singh SJ, Fraser A, Leditschke JF, et al. Gastroschisis: determinants of neonatal outcome. Pediatr Surg Int 2003;19:260–5. https://doi.org/10.1007/s00383-002-0886-0. [42] Overcash RT, DeUgarte DA, Stephenson ML, et al. Factors associated with gastroschisis outcomes. Obstet Gynecol 2014;124(3):551. https://doi.org/10.1097/ AOG.0000000000000425. [43] Aljahdali A, Mohajerani N, Skarsgard ED. Effect of timing of enteral feeding on outcome in gastroschisis. J Pediatr Surg 2013;48(5):971–6. https://doi.org/10.1016/j. jpedsurg.2013.02.014. [44] Oldham KT, Coran AG, Drongowski RA, et al. The development of necrotizing enterocolitis following repair of gastroschisis: a surprisingly high incidence. J Pediatr Surg 1988;23(10):945–9. https://doi.org/10.1016/S0022-3468(88)80392-0.
Please cite this article as: A. Shalaby, A. Obeida, D. Khairy, et al., Assessment of gastroschisis risk factors in Egypt, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.10.027
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Assessment of gastroschisis risk factors in Egypt☆ Aly Shalaby a,⁎, Alaa Obeida a, Dalia Khairy b, Khaled Bahaaeldin a a b
Department of Pediatric Surgery, Cairo University Specialized Pediatric Hospital Department of Pediatrics, Cairo University Specialized Pediatric Hospital
a r t i c l e
i n f o
Article history: Received 17 October 2019 Accepted 26 October 2019 Available online xxxx Key words: Gastroschisis Abdominal wall defect Low income country Egypt Africa
a b s t r a c t Aim: Mortality in infants born with gastroschisis (GS) in low-to-middle-income countries (LMICs) is high. This study aimed to assess factors which might affect outcome in Egypt in order to improve survival. Methods: A prospective study over a 15-month duration was completed. Variables assessed covered patient, maternal, antenatal, treatment, and complications. The Gastroschisis Prognostic Score (GPS) was used to predict outcome. A validated questionnaire was used to assess socioeconomic status. The main outcome was mortality. Results: Twenty-four cases were studied. Median gestational age was 37 (26–40) weeks, and 9 (38%) were preterm. Mortality occurred in 15 (62%) infants. Median transfer time was 8 (1.5–35) hours, and 64% survived if transferred before 8 h. Median maternal age was 20 (16–27) years. All families were of a low or very-low socioeconomic level. Only 25% had antenatal scans. Most cases were simple GS, and only 3 (12.5%) were complex GS. Median length of stay was 14 (1–52) days, TPN duration was 12 (0–49) days, and days to full feeds was 5 (3–11) days. The GPS score ranged from 0 to 6 in the studied cases and negatively correlated with outcome (rS = −0.98; p = 0.03). Conclusion: The mortality of GS in Egypt is very high, mainly due to sepsis and prematurity. Young maternal age and poor socioeconomic status are linked to GS. The GPS is a good indicator of morbidity and mortality in a LMIC setting. Survival improved with better resuscitation and strict management protocols. More effort is needed to improve antenatal detection, and transfer time should be ideally below 8 h. Level of Evidence: Level IV. © 2019 Elsevier Inc. All rights reserved.
Gastroschisis (GS) results from herniation of the intestines through a right-sided (usually) abdominal wall defect [1–3]. The estimated incidence of GS varies from country to country yet the prevalence has generally been rising worldwide [4,5]. A single center report from Ethiopia calculates that abdominal wall defects made up 1.3% of all pediatric surgery conditions [6] and another report from South Africa put GS at 15% of neonatal surgery admissions [7]. GS is readily detected via antenatal ultrasonography and this may affect outcome [8]. Survival rates in developed countries such as the UK and USA may be N 95% [9] and morbidity is largely related to slow restoration of gastrointestinal function with long-term sequelae being rare [10]. By contrast, the experience in Low-to-Middle income countries (LMICs) is plagued by high mortality rates, in general, in neonates with congenital anomalies [11,12] and specifically in GS sometimes reaching 80% to 100% [13–16]. Most deaths may be attributed to sepsis [15] which may be exacerbated by long delays in transfer to a surgical center [14].
☆ The authors declare no conflict of interest.No funding was received for this study. ⁎ Corresponding author at: Department of Pediatric Surgery, Cairo University Specialized Pediatric Hospital, Ali Ibrahim Street, Mounira 11241, Cairo, Egypt. Tel.: + 20 1223905599; fax: +20 2 23649281. E-mail address: [email protected] (A. Shalaby).
There are several determinants of outcome in GS such as the presence or absence of intestinal complications, namely atresia, necrosis, and perforation. GS cases can be classified into complex GS and simple GS with the former having consistently worse outcomes [17,18]. In the absence of intestinal complications, it is not clear why some simple GS patients have worse outcomes than others, with longer periods of bowel dysfunction [19,20]. Other variables which may be relevant include gestational age, mode of delivery, age at closure, closure method, the degree of bowel matting, necrotizing enterocolitis (NEC), cardiac anomalies, and lung hypoplasia [21,22]. This pilot study aimed to describe and assess, where possible, the impact of these different factors on our local population in an attempt to implement the correct strategies to improve survival. 1. Methods Prospective cohort study including all infants born with GS presenting to the Cairo University Specialized Pediatric Hospital (CUSPH), Surgical Neonatal Intensive Care Unit (SNICU) during the period from July 2017 till September 2018. Patient variables included gender, gestational age, gestational weight, consanguinity, birth place, transfer-time and age at surgical intervention. A Gastroschisis Prognostic Score (GPS) was calculated [22],
https://doi.org/10.1016/j.jpedsurg.2019.10.027 0022-3468/© 2019 Elsevier Inc. All rights reserved.
Please cite this article as: A. Shalaby, A. Obeida, D. Khairy, et al., Assessment of gastroschisis risk factors in Egypt, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.10.027
2
A. Shalaby et al. / Journal of Pediatric Surgery xxx (xxxx) xxx
and outcome recorded as live discharge or death. Maternal variables included age, education, place of residence, delivery mode, smoking, alcohol consumption, and illicit drug use. Antenatal variables included frequency of antenatal scans and the presence of any associated anomaly. Treatment variables included type of surgical closure, duration of mechanical ventilation, duration of SNICU admission, duration of parenteral nutrition (PN) and time to full feeds. Finally, the database included postnatal complications: the incidence of central line-associated blood stream infection (CLABSI), Surgical Site Infections (SSIs), and necrotising enterocolitis (NEC). The Clavien Dindo Classification [24] was used to classify post-operative complications. Parental socio-economic status was calculated using the El-Gilany et al. survey [23]. Maximum score is 84. Families with a score of b22 are classified as a very low socioeconomic level, those scoring 23–43 are of a low socioeconomic level, those scoring 44–65 are of an intermediate socioeconomic level, while those scoring 66–84 are of a high socioeconomic level [23]. A standardized management protocol was implemented 4 months into the study period including naso-gastric tube, covering of exposed intestines with cling film and correction of any arterial in-flow or venous out-flow problems. IV fluids were given as a 20 ml/kg bolus of normal saline and 3 ml/kg bolus of 10% Dextrose and maintenance of 10% Dextrose in normal saline at a volume of 120 ml/kg/day. Overtransfusion was actively avoided. Ampicillin/sulbactam and an aminoglycoside was used prophylactically. Surgical closure was done in a sterile environment (preferably theater) even for sutureless closure.
standardized protocol described above. During the first 4 months, survival was 12.5% (1/8) and increased to 50% (8/16 cases) though this did not reach statistical significance (p = 0.17). 2.2. Maternal variables Maternal age was 20 (16–27) years and 12 (50%) were b 20 years. Education level varied from no schooling [n = 6 (25%), school (secondary) education; n = 14(58%) to a college degree, n = 4 (17%)]. Socioeconomic status of the families varied from very low socioeconomic standard (n = 4, 17%) to low standard (n = 20, 83%). Twelve families (50%) lived in the Cairo/Giza governorates; five (21%) from Beni Sueif, four (17%) from Fayoum, three (12%) from Menofia, Minya, and Qalyubia (one from each). Delivery was by normal vaginal delivery (n = 13, 54%) or caesarian section (n = 11, 46%). None of the mothers gave a history of smoking, alcohol consumption, or illicit drug use. 2.3. Antenatal variables Six mothers (25%) had N3 antenatal ultrasound scans, five cases (21%) had them done for three times, two cases (8%) had them twice, six cases (25%) had a scan done once, and five cases (21%) had no scans at all. There were no associated anomalies in the cases scanned. 2.4. Treatment variables
1.1. Data analysis Descriptive analysis tools that were used for single variables are median and range. Analytical tests were done using correlation coefficient, regression analysis, Fisher exact test and ANOVA tests to correlate different variables with the outcome. Significance was accorded at a P value of b0.05. Data are reported as median (range). 2. Results 2.1. Patient variables Twenty four [n = 15 (62%) male] cases of GS presented during the 15-month study period (July 2017–September 2018) and represented 7% of neonatal surgical admissions. Median gestational age was 37 (26–40) weeks. Birth weight was 2.4 (1–3.5) kg. Nineteen infants (79%) were from non-consanguineous marriage and five (21%) from consanguineous marriage. Eleven cases (46%) were inborn at the Cairo University Hospital, with the others born elsewhere [public hospital, n = 4 (17%); private hospital, n = 4 (17%); private clinic, n = 4 (17%) and one (4.1%) was delivered at home]. Transfer-time was 8 (1.5–35) hours. Age at surgical intervention was 15 (5–36) hours. Ten (42%) infants had surgical intervention in b12 h, 10 (42%) were operated on within 12–24 h and 4 (17%) N24 h. Nine infants (37%) were discharged from the unit alive and 15 infants died (63%) died. The outcome of the first 4 months of the study duration (July to October 2017) was compared to the subsequent months (November 2017 to September 2018) after implementing the
The cohort was composed of simple (n = 21) and complex (n = 3) GS cases. The type of surgical closure was staged surgical silo (n = 10, 42%), primary surgical closure (n = 9, 38%), skin closure alone (n = 3, 12%) and primary sutureless closure (n = 1, 4%). One infant died before reaching theater (Fig. 1). Infants with simple GS fared better especially the ones who had a primary fascial closure (Fig. 1). Complex GS cases didn't survive regardless of choice of surgery (Fig. 1). Length of stay (LOS) in the SNICU was 14 (1–52) days. Surviving infants stayed 20 (12–52) days, non-survivors stayed 9 (1–50) days. LOS for primary closure was 12 (1–37) days, and staged silo was 18 (5–52) days. TPN was given to n = 22 (92%) cases for a duration of 12 (0–49) days. TPN days for staged closure cases were 16 (4–49) days, longer than primary closure cases of 10 (1–29) days. Only 9 (37%) infants actually started enteral feeding with the median days to full feeds (DFF) being 5 (3–11) days. 2.5. Postnatal complications The incidence of CLABSI was 4% (1/24 cases) and SSI was 33% (8/24 cases). One case (4%) was diagnosed with NEC. Clavien Dindo classification of complications included grade II (n = 9, 37%) and grade V (n = 15, 62%). Four infants died within 48 h. 2.5.1. Gastroschisis prognostic score (GPS) Nine (38%) infants had a GPS score of 0, 11 cases (46%) had a score of one, two cases (8%) had a score of four and two cases (8%) had a score of six. Of the two cases with GPS 4, one had closed GS with bowel necrosis
Fig. 1. Types of GS, surgery and outcome. GS: Gastroschisis.
Please cite this article as: A. Shalaby, A. Obeida, D. Khairy, et al., Assessment of gastroschisis risk factors in Egypt, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.10.027
A. Shalaby et al. / Journal of Pediatric Surgery xxx (xxxx) xxx Table 1 Outcome related to transfer time. Transfer time
Alive
Dead
b8 hours ≥8 hours
7 2 9
4 11 15
11 13 24
Alive
Dead
Total
5 4 0 9
5 6 4 15
10 10 4 24
p = 0.03
Table 2 Outcome compared to age at surgery. Outcome
b12 hours 12–24 hours N 24 hours p = 0.29
from the duodeno-jejunal junction till the colon, while the other had severe bowel matting. The former case died, while the latter survived. Both cases with GPS 6 had jejunal atresia associated with severe bowel matting and both died. The relationship between the GPS and outcome showed a strong negative correlation (rS = −0.98; p = 0.03). 2.6. Other factors Eight hours was used as a cut-off to compare the “early” versus the “delayed” transfer. Out of the 11 “early” cases transferred before 8 h, 64% (n = 7) survived. In contrast, out of the 13 “delayed” cases only 15% (n = 2) survived (p = 0.03) (Table 1). Survival was 50% for infants undergoing surgery b12 h of life, 40% for intervention at 12-24 h and 0% at ≥24 h (p = 0.28). (See Table 2.) Outborn cases (n 13, 54%) were transferred from distances varying from 10 to 256 km. The number of inborn and outborn cases was similar (n = 11 vs n = 13). Five inborn and 10 outborn died but this didn't reach statistical significance (p = 0.1). Ventilated cases (n = 3, 18%) survived compared to (n = 6, 86%) non ventilated ones (p = 0.003). 3. Discussion We characterized our series as typically arising from a nonconsanguineous marriage with a slight male predominance [25]. Most were full term and only a third preterm [26,27]. We did have a high overall mortality of N 60%, especially obvious in the premature and in this regard is similar to experience in other LMICs [28]. The GPS [22] used in this study was very easy to use and reliably predicted the outcome. The latter has been echoed by Puligandla et al. [29]. Most of our survivors reached the unit before 8 h [Fig. 2]. The literature is not clear as to what constitutes a delay in transfer. Considering
3
our findings, we propose defining “delayed transfer” as anything at or above 8 h. It has been reported that transport and bowel stabilization times could not predict outcome but were independently predictive of DFF, time on TPN and LOS [30]. Furthermore, an inborn status does not affect outcome but favorably impacts the time to surgery, DFF and the LOS [31]. There are reports of GS clustering and association with poor socioeconomic status [32–34]. In our study, the majority of families were of low or very low status. None of the mothers in our study had a history of smoking, alcohol consumption, or illicit drug use, despite many studies identifying them as risk factors for GS [25,35]. There was poor compliance with antenatal care visits in our study. This could be explained in part by the families' low socioeconomic status. In a broader context only 53% of pregnant women in Egypt seek antenatal care [36]. Furthermore, the drop-off in the number of antenatal visits with increasing parity is steep, dropping from 41% for first births to 13% for sixth or higher-order births. The gap between the richest 20% and the poorest 20% for use of antenatal care in Egypt is large [36]. The ventilated cases in this study had a significantly worse outcome. Ventilated infants in LMIC countries have a mortality rate in the range of 40–60% [37]. GS often need mechanical ventilation at a certain point of the management process. There is no high-level evidence that reduction without anesthesia or an abdominal incision reduces the risks that may occur from ventilation [38]. Low-risk (simple) GS have an ipso facto shorter LOS compared to high-risk (complex) cases [29]. Staged silo infants in our study had a longer LOS which has been independently associated with an increased LOS [40]. Our staged closure cases were on TPN for longer, in keeping with other reports in the literature [41]. TPN cholestasis can reach up to 25% of cases with a higher incidence in pre-terms [42]; however, this was not observed in our study. In the current study 42% started enteral feeding, 90% of whom reached full volumes and survived, while one case (10%) died from sepsis. Our median DFF of 5 days is slightly earlier than the 7 days purported to offer the best outcome, stop TPN and decrease LOS [43]. We reported only one case (4.17%) with NEC. Its incidence after GS repair may reach up to 20% [44]. Unlike the classic associations of prematurity, low birth weight, and cardiac abnormalities with NEC, GS NEC seems to increase if the neonate is outborn or has developed CLABSI [39]. In conclusion, despite GS being an isolated pathology with no comorbidities, there are many extraneous factors affecting its outcome. Its management has been proposed as a reliable indicator for the maturity of neonatal surgical care services in a country [28]. There is a scarcity of published literature on GS from our region and our pilot study has painted a far-from-perfect picture. To the best of our knowledge, it is the first of its kind to outline the factors affecting GS in Egypt: as elsewhere in the world, a young maternal age and poor socioeconomic status are linked to GS. Survival improves with better resuscitation, strict management protocols and effective infection control. More effort is needed to improve antenatal detection. Finally, efficient referral networks need to cut the transfer time below 8 h.
Fig. 2. Outcome of GS related to transfer time. Dashed line at 8 hours.
Please cite this article as: A. Shalaby, A. Obeida, D. Khairy, et al., Assessment of gastroschisis risk factors in Egypt, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.10.027
4
A. Shalaby et al. / Journal of Pediatric Surgery xxx (xxxx) xxx
References [1] Wilson RD, Johnson MP. Congenital abdominal wall defects: an update. Fetal Diagn Ther 2004;19:385–98. https://doi.org/10.1159/000078990. [2] David AL, Tan A, Curry J. Gastroschisis: Sonographic diagnosis, associations, management and outcome. Prenat Diagn 2008;28:633–44. https://doi.org/10.1002/pd.1999. [3] Gow KW, Bhatia A, Saad DF, et al. Left-sided gastroschisis. Am Surg 2006;72:637–40. [4] International Clearinghouse for Birth Defects Surveillance and Research Centre. ICBDSR annual report 2014. International Clearinghouse for Birth Defects Surveillance and Research Centre, 2014. http://www.icbdsr.org/wp-content/annual_ report/Report2014.pdf [accessed 02 August 2019]. [5] Parker SE, Mai CT, Canfield MA, et al. Updated national birth prevalence estimates for selected birth defects in the United States, 2004–2006. Birth Defects Res Part A - Clin Mol Teratol 2010; 88(12):1008–16. doi:https://doi.org/10.1002/bdra.20735. [6] Derbew M. Pediatric surgery in eastern Africa: the unmet need. J Pediatr Surg 2018; 54:21–6. https://doi.org/10.1016/j.jpedsurg.2018.10.028. [7] Sekabira J, Hadley GP. Gastroschisis: a third world perspective. Pediatr Surg Int 2009;25:327–9. https://doi.org/10.1007/s00383-009-2348-4. [8] Bond SJ, Harrison MR, Filly RA, et al. Severity of intestinal damage in gastroschisis: correlation with prenatal sonographic findings. J Pediatr Surg 1988;23:520–5. https://doi.org/10.1016/S0022-3468(88)80360-9. [9] Holland AJ, Walker K, Badawi N. Gastroschisis: an update. Pediatr Surg Int 2010;26: 871. https://doi.org/10.1007/s00383-010-2679-1. [10] Langer JC, Longaker MT, Crombleholme TM, et al. Etiology of intestinal damage in gastroschisis. I: effects of amniotic fluid exposure and bowel constriction in a fetal lamb model. J Pediatr Surg 1989;24:992–7. https://doi.org/10.1016/S0022-3468 (89)80200-3. [11] Murray CJL, Vos T, Lozano R, et al. Disability-adjusted life years (DALYs) for 291 diseases and injuries in 21 regions, 1990–2010: A systematic analysis for the Global Burden of Disease Study 2010. Lancet 2012; 380(9859):2197–223. doi:https://doi. org/10.1016/S0140-6736(12)61689-4. [12] Black RE, Cousens S, Johnson HL, et al. Global, regional, and national causes of child mortality in 2008: a systematic analysis. Lancet 2010; 375(9730):1969–87. doi: https://doi.org/10.1016/S0140-6736(10)60549-1. [13] Arnold M. Is the incidence of gastroschisis rising in South Africa in accordance with international trends? Sth Afr J Surg 2004;42:86–8. [14] Ameh EA, Chirdan LB. Ruptured exomphalos and gastroschisis: a retrospective analysis of morbidity and mortality in Nigerian children. Pediatr Surg Int 2000;16:23–5. https://doi.org/10.1007/s003830050006. [15] Vilela PC, Ramos De Amorim MM, Falbo GH, et al. Risk factors for adverse outcome of newborns with gastroschisis in a Brazilian hospital. J Pediatr Surg 2001;36:559–64. https://doi.org/10.1053/jpsu.2001.22282. [16] Askarpour S, Ostadian N, Javaherizadeh H, et al. Omphalocele, gastroschisis: Epidemiology, survival, and mortality in Imam Khomeini Hospital, Ahvaz-Iran. Pol Prz Chir Polish J Surg 2012; 84: 82–5. doi:https://doi.org/10.2478/v10035-012-0013-4. [17] Bergholz R, Boettcher M, Reinshagen K, et al. 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. https://doi.org/10.1016/j. jpedsurg.2014.08.001. [18] Emil S, Canvasser N, Chen T, et al. Contemporary 2-year outcomes of complex gastroschisis. J Pediatr Surg 2012;47:1521–8. https://doi.org/10.1016/j.jpedsurg. 2011.12.023. [19] Baird R, Eeson G, Safavi A, et al. Institutional practice and outcome variation in the management of congenital diaphragmatic hernia and gastroschisis in Canada: a report from the Canadian pediatric surgery network. J Pediatr Surg 2011;46:801–7. https://doi.org/10.1016/j.jpedsurg.2011.02.008. [20] Lusk LA, Brown EG, Overcash RT, et al. Multi-institutional practice patterns and outcomes in uncomplicated gastroschisis: a report from the University of California Fetal Consortium (UCfC). J Pediatr Surg 2014;49:1782–6. https://doi.org/10.1016/j. jpedsurg.2014.09.018. [21] Arnold MA, Chang DC, Nabaweesi R, et al. Development and validation of a risk stratification index to predict death in gastroschisis. J Pediatr Surg 2007;42:950–6. https://doi.org/10.1016/j.jpedsurg.2007.01.028. [22] Cowan KN, Puligandla PS, Laberge JM, et al. The gastroschisis prognostic score: reliable outcome prediction in gastroschisis. J Pediatr Surg 2012;47:1111–7. https://doi.
org/10.1016/j.jpedsurg.2012.03.010. [23] El-Gilany A, El-Wehady A, El-Wasify M. Updating and validation of the socioeconomic status scale for health research in Egypt. East Mediterr Heal J 2012; 18: 962–68. doi:10.26719/2012.18.9.962. [24] Clavien PA, Barkun J, De Oliveira ML, et al. The Clavien-Dindo classification of surgical complications: five-year experience. Ann Surg 2009;250:187–96. https://doi.org/ 10.1097/SLA.0b013e3181b13ca2. [25] Hougland KT, Hanna AM, Meyers R, et al. Increasing prevalence of gastroschisis in Utah. J Pediatr Surg 2005;40:535–40. https://doi.org/10.1016/j.jpedsurg.2004.11. 026. [26] Abdel-Latif ME, Bolisetty S, Abeywardana S, et al. Mode of delivery and neonatal survival of infants with gastroschisis in Australia and New Zealand. J Pediatr Surg 2008; 43:1685–90. https://doi.org/10.1016/j.jpedsurg.2008.03.053. [27] Lausman AY, Langer JC, Tai M, et al. Gastroschisis: what is the average gestational age of spontaneous delivery? J Pediatr Surg 2007; 42: 1816–21. doi:https://doi. org/10.1016/j.jpedsurg.2007.07.005. [28] Ford K, Poenaru D, Moulot O, et al. Gastroschisis: bellwether for neonatal surgery capacity in low resource settings? J Pediatr Surg 2016;51:1262–7. https://doi.org/10. 1016/j.jpedsurg.2016.02.090. [29] Puligandla PS, Baird R, Skarsgard ED, et al. Outcome prediction in gastroschisis – the gastroschisis prognostic score (GPS) revisited. J Pediatr Surg 2017;52:718–21. https://doi.org/10.1016/j.jpedsurg.2017.01.017. [30] Bucher BT, Mazotas IG, Warner BW, et al. Effect of time to surgical evaluation on the outcomes of infants with gastroschisis. J Pediatr Surg 2012;47:1105–10. https://doi. org/10.1016/j.jpedsurg.2012.03.016. [31] Savoie KB, Huang EY, Aziz SK, et al. Improving gastroschisis outcomes: does birth place matter? J Pediatr Surg 2014;49:1771–5. https://doi.org/10.1016/j.jpedsurg. 2014.09.019. [32] Chircor L, Mehedinţi R, Hîncu M. Risk factors related to omphalocele and gastroschisis. Rom J Morphol Embryol 2009;50:645–9. [33] Root ED, Meyer RE, Emch ME. Evidence of localized clustering of gastroschisis births in North Carolina, 1999–2004. Soc Sci Med 2009;68:1361–7. https://doi.org/10. 1016/j.socscimed.2009.01.034. [34] Rankin J, Pattenden S, Abramsky L, et al. Prevalence of congenital anomalies in five British regions, 1991–99. Arch Dis Child Fetal Neonatal Ed 2005;90:F374–9. https://doi.org/10.1136/adc.2003.047902. [35] Lam PK, Torfs CP. Interaction between maternal smoking and malnutrition in infant risk of gastroschisis. Birth Defects Res Part A - Clin Mol Teratol 2006;76:182–6. https://doi.org/10.1002/bdra.20238. [36] Abou-Zahr I, Lidia C, Wardlaw Tessa M. Antenatal Care in Developing Countries Promises, achievements and missed opportunities. WHO Libr Cat Data Antenatal 2003. doi:25/12/2014. [37] Kambarami R, Chidede O, Chirisa M. Neonatal intensive care in a developing country: outcome and factors associated with mortality. Cent Afr J Med 2000;46:205–7. [38] Davies MW, Kimble RM, Woodgate PG. Ward reduction without general anaesthesia versus reduction and repair under general anaesthesia for gastroschisis in newborn infants. Cochrane Database Syst Rev 2002;3. https://doi.org/10.1002/14651858. cd003671. [39] Youssef F, Laberge JM, Puligandla P, et al. Determinants of outcomes in patients with simple gastroschisis. J Pediatr Surg 2017;52:710–4. https://doi.org/10.1016/j. jpedsurg.2017.01.019. [40] Murthy K, Evans JR, Bhatia AM, et al. The association of type of surgical closure on length of stay among infants with gastroschisis born ≥34 weeks' gestation. J Pediatr Surg 2014;49:1220–5. https://doi.org/10.1016/j.jpedsurg.2013.12.020. [41] Singh SJ, Fraser A, Leditschke JF, et al. Gastroschisis: determinants of neonatal outcome. Pediatr Surg Int 2003;19:260–5. https://doi.org/10.1007/s00383-002-0886-0. [42] Overcash RT, DeUgarte DA, Stephenson ML, et al. Factors associated with gastroschisis outcomes. Obstet Gynecol 2014;124(3):551. https://doi.org/10.1097/ AOG.0000000000000425. [43] Aljahdali A, Mohajerani N, Skarsgard ED. Effect of timing of enteral feeding on outcome in gastroschisis. J Pediatr Surg 2013;48(5):971–6. https://doi.org/10.1016/j. jpedsurg.2013.02.014. [44] Oldham KT, Coran AG, Drongowski RA, et al. The development of necrotizing enterocolitis following repair of gastroschisis: a surprisingly high incidence. J Pediatr Surg 1988;23(10):945–9. https://doi.org/10.1016/S0022-3468(88)80392-0.
Please cite this article as: A. Shalaby, A. Obeida, D. Khairy, et al., Assessment of gastroschisis risk factors in Egypt, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.10.027