Hospital Readmission Among Infants with Gastroschisis

AP South1,2, JJ Wessel1,2, A Sberna1, M Patel1 and AL Morrow1 1Division of Neonatology, Perinatal Institute, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH, USA and 2Intestinal Rehabilitation Program, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH, USA

Introduction Objective: Infants with gastroschisis have significant perinatal morbidity Gastroschisis is a congenital abdominal wall defect that results in including long hospitalizations and feeding intolerance. Two thirds are evisceration of the bowel into the amniotic space. The birth premature and 20% are growth restricted. Despite these known risk factors prevalence is increasing, affecting B4.5 per 10 000 births.1 While for post-natal complications, little is known about readmission for infants in-hospital morbidity and mortality are well described, there is with gastroschisis. Our objective was to determine the frequency and limited information regarding post-discharge outcomes. Infants indication for hospital readmission after initial discharge among infants with gastroschisis have multiple risk factors for poor long-term with gastroschisis. outcome, including prematurity in two thirds,2 and poor in utero Study Design: Retrospective cohort study. All surviving infants treated growth in 20%.3 Despite absence of extreme prematurity in most for gastroschisis at Cincinnati Children’s Hospital Medical Center, born cases, all infants with gastroschisis are at risk for the development between January 2006 and December 2008 were included. Main outcome of necrotizing enterocolitis with subsequent bowel injury or loss. In measures included the frequency and indication for readmission. addition, 11% of infants have complex gastrointestinal involvement Associated neonatal risk factors also were assessed. (atresia, stenosis, perforation, necrosis or volvulus) which requires 4 Result: Fifty-eight patients were analyzed. Twenty-three (40%) subjects surgical resection of bowel. After repair of the abdominal wall were readmitted (five with multiple readmissions); 65% of readmissions defect, infants with or without a complex lesion experience a period occurred in the first year and 70% involved complications directly related of bowel dysmotility, resulting in prolonged time on parenteral to gastroschisis. The most common reasons for readmission were bowel nutrition and subsequent prolonged hospitalizations lasting 5,6 obstruction and abdominal distention/pain. Median time to readmission 4 to 8 weeks. directly related to gastroschisis was 23 weeks (range 5 to 92). All three Changes in bowel function over time have not been formally infants with home parenteral nutrition were readmitted. Readmission was evaluated among infants with gastroschisis, but persistent not associated with gestational age, birth weight or length of initial dysfunction could contribute to post-discharge morbidity and hospitalization. rehospitalization. Our objective was to identify the frequency of readmissions after initial hospital discharge among infants with Conclusion: Readmission after initial hospitalization is common in gastroschisis and identify modifiable perinatal risk factors for gastroschisis patients. Parental counseling should include education readmission. regarding the possibility of complications requiring readmission. Determinants of readmission require further study. Journal of Perinatology (2011) 31, 546–550; doi:10.1038/jp.2010.206; Methods published online 10 February 2011 After obtaining Institutional Review Board approval from Keywords: abdominal wall defect; gastroschisis; readmission; long-term Cincinnati Children’s Hospital Medical Center (CCHMC), a outcomes; bowel obstruction; surgical complications retrospective cohort study was performed. Subjects included all neonates treated for gastroschisis at CCHMC, born between 1 January 2006 and 31 December 2008, and who survived until initial hospital discharge. Infants were not excluded for comorbid conditions including bowel resection or other congenital anomalies. Infants whose initial surgical procedure was not Correspondence: Dr AP South, Cincinnati Children’s Hospital Medical Center, 3333 Burnet performed at CCHMC were excluded. Subjects were identified Avenue, ML 7009, Cincinnati, OH 45229, USA. through review of the diagnosis codes assigned by the attending E-mail: [email protected] Received 26 August 2010; revised 31 October 2010; accepted 2 November 2010; published online neonatologist over the specified time period. For each identified 10 February 2011 patient, surgical procedure notes and attending neonatologist gastroschisis and hospital readmission AP South et al 547 progress notes were evaluated to confirm the diagnosis of 33 weeks (range 28 to 36) and birth weight of 1870 g (range 950 to gastroschisis. Complete ascertainment of subjects was confirmed 2480). Three infants died of bowel-related morbidity, including by comparing identified patients with hospital billing records. total necrosis, necrotizing enterocolitis totalis and intestinal failure. All data were abstracted retrospectively from the medical record. Two infants died secondary to pulmonary morbidity (pulmonary Perinatal management including delivery hospital, mode of hypertension with alveolar–capillary dysplasia). delivery, method of repair and nutritional management were Demographic characteristics and initial medical course are determined. Neonatal characteristics including gestational age, presented in Table 1. Seventy-five percent of infants were preterm. birthweight and gender were determined. Medical and surgical The mean gestational age of all infants was 35.6 weeks. Infant complications, including the presence of complex bowel birth weight was below expected for gestational age (mean, 32nd involvement (atresia, stenosis, necrosis, volvulus or perforation) and bowel resection, and length of initial hospitalization were recorded. Readmission after initial hospital discharge was Table 1 Demographics and initial medical course of surviving infants with determined by reviewing the complete medical record for each gastroschisis identified infant. Details recorded include the primary indication Maternal/infant demographics and date of readmission. Gestational age (n ¼ 56) During the time period of study, the surgical management plan Mean (s.d.), weeks 35.6 (2.0) was determined by the attending pediatric surgeon. There was no Preterma, n/N (%) 42/56 (75%) standard practice or protocol among the group of >15 surgeons. A Birth weight (n ¼ 58) primary closure was attempted only when viscero-abdominal Mean (s.d.), g 2463 (508) disproportion was not significant. Birth weight for gestational age (n ¼ 56) All statistical analysis was performed with Stata 10.1 (College Percentile, mean (s.d.) 32.0 (22.9) Station, TX, USA). Both primary outcomes and co-variates are z-score, mean (s.d.) À0.6 (0.8) described using mean values and standard deviations for normally Small for gestational ageb, n/N (%) 10/56 (18%) distributed data and median values with inter-quartile range for Male gender, n/N (%) 34/58 (59%) non-normal distributions. Birth at level 3 Neonatal Intensive Care Unit, n/N (%) 48/57 (84%) Kaplan–Meier survival estimates were determined using Maternal age (n ¼ 58) Mean (s.d.), years 22.2 (4.6) readmission as the censored variable. Infants were censored at the Teenage motherc, n/N (%) 17/58 (29%) time of readmission, or on 31 December 2009, whichever came Prenatal diagnosis, n/N (%) 53/58 (91%) first. Variables analyzed included preterm birth, small for Vaginal delivery, n/N (%) 33/58 (57%) gestational age (birth weight less than the 10th percentile), gender, birth at a hospital with a level three neonatal intensive care unit, Medical/surgical care and complications mode of delivery, prenatal diagnosis, method of surgical Primary closure, n/N (%) 12/58 (21%) management, age of enteral feeding initiation, days on parenteral Complex defectd, n/N (%) 4/58 (7%) nutrition, length of hospitalization, development of necrotizing Age enteral feeds initiated (n ¼ 58) enterocolitis, complex gastrointestinal involvement. Cox Median (IQR), days 16 (13–19) proportional hazards models were performed to determine Range, days 6–104 statistically significant differences in readmission rates between Enteral feeds initiated at p30 days after birth, n/N (%) 55/58 (95%) dichotomous variables. For co-variates found to be statistically Days on parenteral nutrition (n ¼ 55) Median (IQR), days 31 (23–41) associated with readmission rate, baseline neonatal demographics Range, days 14–176 were compared using Student’s t-test for continuous data, Length of initial perinatal hospitalization 2 and w or Fisher’s exact test for dichotomous data. Tests were Median (IQR), days 44 (36–72) considered statistically significant for P-values <0.05 using Range, days 27–178 a two-sided distribution. Necrotizing enterocolitis, n/N (%) 4/58 (7%) Discharged with home parenteral nutrition, n/N (%) 3/58 (5%) Discharged with home nasogastric or gastrostomy 13/58 (22%) Results tube feedings, n/N (%)

Sixty-three neonates with gastroschisis had their initial Abbreviations: IQR, interquartile range; s.d., standard deviation. gastroschisis repair at CCHMC from January 2006 through aPreterm defined as gestational age <37 weeks. b December 2008. Five (8%) infants died during hospitalization, Small for gestational age defined as weight <10th percentile at birth. cTeenage mother defined as maternal age <20 years. leaving 58 infants who survived until discharge and were included dComplex defect defined as gastroschisis with atresia, stenosis, necrosis, volvulus or in this evaluation. Infants who died had a mean gestation age of perforation within the first 2 weeks after birth.

Journal of Perinatology gastroschisis and hospital readmission AP South et al 548

Table 2 Readmission characteristics Table 3 Details of readmission

Total Directly related to gastroschisis Not directly related to gastroschisis (n ¼ 17) (n ¼ 6) Readmitted at least one time, n/N (%) 23/58 (40%) Readmitted for problem directly related to gastroschisis, n/N (%) 17/58 (29%) Bowel obstruction (n ¼ 5) Orchiopexy (n ¼ 3) Readmitted for bowel obstruction, n/N (%) 5/58 (9%) Abdominal distention (n ¼ 2) Mastoiditis (n ¼ 1) Catheter-related infection (n ¼ 2) Pressure equalization tubes (n ¼ 1) Time to readmission for a problem directly related to gastroschisisa Gastroenteritis (n ¼ 2) Median (IQR), weeks 23 (12, 41) Ventral hernia repair (n ¼ 2) Readmitted for problem not directly related to gastroschisis, 6/58 (10%) Necrotizing enterocolitis (n ¼ 1) n/N (%) Abdominal pain (n ¼ 1) Failure to thrive (n ¼ 1) Time to readmission for a problem not directly related to gastroschisisa Gastrostomy tube replacement (n ¼ 1) Median (IQR), weeks 65 (41, 74)

Abbreviation: IQR, interquartile range. Frequency and Timing of All Readmissions aWilcoxon rank-sum P ¼ 0.025. 0.00

percentile, z-score ¼À0.6); 18% were small for gestational age 0.25 (less than the 10th percentile). Only 12 infants (21%) had a primary closure and all others had a silo repair. Median time to initiate feeding was 16 days, and the median duration of parenteral 0.50 nutrition was 31 days. The median length of initial hospitalization was 44 days. Proportion Readmitted 0.75 Forty percent of the cohort was readmitted (Table 2), with 17 (29%) of the cohort readmitted for problems directly related to gastroschisis (Table 3). Median time to readmission was 23 weeks 1.00 (interquartile range ¼ 12, 41) for problems directly related to Birth 1 Year 2 Years 3 Years 4 Years Analysis Time gastroschisis, and 65 weeks (interquartile range ¼ 41, 74) for problems not directly related to gastroschisis (Wilcoxon rank-sum Timing of Readmission by Relation to Gastroschisis P ¼ 0.025). 0.00 A Kaplan–Meier plot representing the percentage of infants readmitted over time for the entire cohort is presented in Figure 1a. 0.25 No infants were readmitted after 2 years of age. Figure 1b is a

Kaplan–Meier plot representing the timing of readmission based 0.50 on whether or not the readmission was directly related to gastroschisis, and includes only those infants readmitted. Kaplan–

Proportion Readmitted 0.75 Meier estimates and Cox proportional hazards estimates were calculated for speciﬁc risk factors. There were no differences in all readmissions, or in readmission for causes directly related to 1.00 gastroschisis based on birth at a level 3 vs level 1 or 2 birth 1 Year 2 Years 3 Years 4 Years hospital; bowel resection during hospitalization; complex Analysis Time gastrointestinal involvement; small for gestational age at birth Not Gastroschisis Related Gastroschisis Related (weight <10th percentile); mode of delivery (cesarean vs vaginal); Figure 1 Frequency and timing of all readmissions (a) and of all readmissions preterm delivery (<37 weeks); timing of initiation of enteral by reason for readmission (b). feedings (<20 days vs X20 days); parenteral nutrition duration (<60 days vs X60 days); length of hospitalization (<56 days vs X56 days); gender; maternal age (<20 years vs X20 years); and Eight (67%) of 12 infants managed with initial primary closure prenatal diagnosis. were readmitted, while only 9 (20%) of 46 infants managed with a Survival analysis with Cox proportional hazards estimates silo were readmitted (Fisher’s exact P ¼ 0.003). Readmission for indicates that infants managed with primary closure were more bowel obstruction occurred in 2 (17%) of 12 infants closed likely to be readmitted for any reason (log-rank test, P ¼ 0.027). primarily and 3 (7%) of 46 infants closed with a silo (Fisher’s exact

Journal of Perinatology gastroschisis and hospital readmission AP South et al 549

P ¼ 0.273). Baseline demographic and clinical conditions did not management of gastroschisis. The ideal method of abdominal wall differ based on the mode of initial repair. closure has been previously evaluated in terms of mortality, length of hospitalization, time on parenteral nutrition and time to initiate or achieve full enteral feeds.12–15 Yet, one could argue that aside Discussion from mortality, none of those outcomes are relevant if they come at The present study demonstrates that infants with gastroschisis are the expense of continued, prolonged morbidity after discharge. In commonly readmitted within the first 2 years of life. Information our sample, infants undergoing primary closure were more likely on post-discharge outcomes of children with gastroschisis is to be readmitted for bowel obstruction, however, this did not reach limited, as most studies report only perinatal outcomes. Two statistical significance. This finding is surprising, given that these publications report widely variant readmission rates. We previously patients are subjectively perceived as less seriously affected, and reported that half of the infants prospectively evaluated at 18 to 24 therefore with a better prognosis. Our findings raise the possibility months had been readmitted, with half of the readmissions directly that the method of abdominal wall closure may have important related to bowel pathology.5 Piper and Jaksic7 reported only on implications for readmission rates and deserves additional study. abdominal complications requiring readmission, citing a 15% This study should prompt three changes in the way we care for readmission rate among 27 infants with gastroschisis. While infants with gastroschisis. First, prenatal and pre-discharge prenatal bowel dilation may have been a risk factor for counseling should include discussions of long-term morbidities readmission,7 details regarding perinatal risk factors for including readmissions, and the high-likelihood for bowel readmission were not reported. Results of the present study are obstruction. Secondly, a larger multi-center analysis to consistent with our previous work.5 prospectively evaluate methods of abdominal wall closure should Forty percent of the 58 infants in our study were readmitted, and be performed, with outcomes focused on growth, neurodevelopment more than one-quarter were readmitted with problems directly and bowel function. Finally, further evaluation is needed to related to gastroschisis. These included 7% with bowel obstruction determine how modification in outpatient follow-up of infants with and 5% with abdominal distention or pain. Bowel obstruction is gastroschisis could reduce the frequency of rehospitalization. a known comorbidity associated with abdominal surgery, and may Our study has several limitations. It was retrospective in design, be related to the formation of adhesions.8 Furthermore, adhesions allowing us only to access information already recorded. In could impair proper bowel function, causing constipation, addition, we may have underestimated the frequency of diarrhea or abdominal pain. Previous studies on bowel obstruction readmission secondary to readmission to other outside hospitals. after neonatal laparotomy support that readmission for bowel We believe that this is unlikely to occur given that our center is the obstruction among infants with gastroschisis is common. van Eijck sole provider of pediatric surgical, gastrointestinal and neonatology et al.8 reported that 25% of 55 infants with gastroschisis developed services in the area. Thus, only those patients who moved away bowel obstruction, much higher than the rate seen in our from the region after their initial hospitalization would not be evaluation. In their retrospective case series, the cumulative identified. hazard for developing small bowel obstruction was 27% at 6 We have shown that readmission after gastroschisis is common, months. No additional cases of bowel obstruction were seen and that bowel obstruction is the most frequent complication between 6 months and 4 years. Reasons for the increased frequency directly related to the abdominal wall defect. Further evaluation of of readmission in the van Eijck’s study are not obvious. One neonatal management strategies including surgical management, possibility is that we have underestimated the frequency of route and timing of delivery, and enteral feeding strategies is readmission secondary to bowel obstruction. However, Koivusalo imperative, with emphasis on long-term outcomes including et al.9 reported only 3 of 11 children with gastroschisis developed readmission, growth, nutrition, and bowel disorders. Results of this subsequent obstruction. study should inform future randomized clinical trials designed to Review of the literature suggests a general consensus that improve outcomes among children with gastroschisis. infants with gastroschisis generally do well without long-term morbidities other than occasional short bowel syndrome with malabsorption.10,11 These conclusions are based on individual Conflict of interest physician experience, small numbers of patients and studies with The authors declare no conflict of interest. methodological limitations. The present study does not support that consensus. Rather, our results demonstrate that infants with gastroschisis are at risk for significant morbidity after initial Acknowledgments hospital discharge. We thank the Perinatal Institute and Intestinal Rehabilitation Program at Our results also point to the importance of identifying and Cincinnati Children’s Hospital Medical Center for financially supporting this evaluating more pertinent outcome measures in evaluating the research effort.

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