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Nationwide analysis of mortality and hospital readmissions in esophageal atresia
YJPSU-59569; No of Pages 6 Journal of Pediatric Surgery xxx (xxxx) xxx
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Nationwide analysis of mortality and hospital readmissions in esophageal atresia Hallie J. Quiroz a, Alexa Turpin b, Brent A. Willobee a, Anthony Ferrantella a, Joshua Parreco c, David Lasko d, Eduardo A. Perez a, Juan E. Sola a, Chad M. Thorson a,⁎ a
Dewitt-Daughtry Family Department of Surgery, Division of Pediatric Surgery, University of Miami Miller School of Medicine University of Miami Miller School of Medicine Dewitt-Daughtry Family Department of Surgery, Division of Trauma and Acute Care Surgery, University of Miami Miller School of Medicine d South Florida Pediatric Surgeons b c
a r t i c l e
i n f o
Article history: Received 14 January 2020 Accepted 25 January 2020 Available online xxxx Key words: Esophageal atresia Outcomes Readmission Infants
a b s t r a c t Purpose: The purpose of this study is to identify determinants of mortality and hospital readmission in infants born with esophageal atresia ± tracheoesophageal fistula. Methods: The Nationwide Readmissions Database (2010–2014) was queried for newborns with a diagnosis of esophageal atresia. Outcomes included mortality and readmissions at 30-day and 1-year. Results: 3157 patients were identified, of which 54% were male. 81% had an additional congenital anomaly, and 35% had VACTERL association. Overall mortality at index hospitalization was 11% (n = 360) and was significantly higher with additional congenital anomalies (13%), VACTERL (19%), and Spitz classification II/III (18%) vs. isolated esophageal atresia/tracheoesophageal fistula (4%), all p b 0.001. After esophageal atresia repair (n = 2179), 10% (n = 212) were readmitted within 30 days and 26% (n = 563) within 1 year, with 17% admitted to different hospitals. Common diagnoses during readmission were GERD (54%), infections (42%), failure to thrive (17%), tracheomalacia (14%), and esophageal stricture (10%). Unplanned readmissions accounted for 85% of readmissions. A large number underwent operative procedures, most commonly esophageal dilation (17%) and fundoplication/gastrostomy (12%). Conclusion: Our study has uncovered a high likelihood of complications and unplanned readmission within the first year of life for newborns with esophageal atresia. Coordinated multidisciplinary care may help to decrease unnecessary readmissions and improve outcomes in this vulnerable population. Type of study: Retrospective comparative analysis. Level of evidence: Level III. © 2020 Elsevier Inc. All rights reserved.
Esophageal atresia (EA) is a congenital anomaly characterized by abnormal formation of the primitive foregut leading to discontinuity between the proximal and distal portions of the esophagus, which often occurs in association with tracheoesophageal fistula (TEF). Prior to the first successful operation for EA, this congenital anomaly conferred a 100% mortality rate. [1] With contemporary surgical management and advances in neonatal care, survival is typically dependent upon the presence and severity of associated anomalies or conditions such as low birth weight. [2] Infants with EA ± TEF often suffer complications such as severe gastroesophageal reflux (GERD), esophageal strictures, chronic dysphagia,
⁎ Corresponding author at: University of Miami Miller School of Medicine, Division of Pediatric Surgery, P.O. Box 016960 (R-51), Miami, FL. Tel.: +1 305 243 2247; fax: +1 305 243 5731. E-mail address: [email protected] (C.M. Thorson).
recurrent fistula, pulmonary infections, reactive airway disease, and tracheomalacia. [3–6] The majority of existing literature reporting these complications comes from institutional case series limited by small sample size. Large-scale nationwide analyses have been reported utilizing the Pediatric Health Information System (PHIS) [2] and the Kid's Inpatient Database (KID) [7]. However, these databases track a single inpatient hospitalization leading to paucity of long-term outcomes data regarding hospital readmissions and complications. Programs such as the Children's Health Insurance Program Reauthorization Act (CHIPRA) have identified readmissions as a quality core metric in development [8,9] and the federal Partnership for Patients initiative has challenged hospitals to reduce their readmissions by 20%. [10,11] Given the importance of reducing future readmissions, studies are needed to identify baseline readmissions for complex diseases in order to improve hospital protocols. The present study represents a large, nationwide analysis of risk factors during neonatal and subsequent hospital readmissions in neonates born with EA ± TEF. We
https://doi.org/10.1016/j.jpedsurg.2020.01.025 0022-3468/© 2020 Elsevier Inc. All rights reserved.
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
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hypothesized that patients with EA ± TEF would have a high readmission rate owing to underlying medical complexity. 1. Methods The Nationwide Readmissions Database (NRD) is part of a family of databases developed by the Healthcare Cost and Utilization Project (HCUP). This database was developed to address a large gap in healthcare data — the lack of nationally representative information on hospital readmissions for all ages. The NRD contains approximately 17 million unweighted discharges each year and discharge data from 22 geographically dispersed States, accounting for 49% of all United States hospitalizations [12]. The NRD also has the unique ability to track patients' readmissions to different hospitals by using distinct, patient specific identifiers within the database, thus making it an ideal database for studies on nationwide readmissions. The NRD for 2010–2014 was queried for all newborn patients with a diagnosis of EA ± TEF utilizing the International Classification of Disease
Table 1 Neonates with esophageal atresia, demographic data. Characteristic Demographics Gender Female Male Insurance Status Medicaid Private Insurance Self-Pay Income (Quartile) Highest Quartile Income ($67,000 +) Second Quartile Income ($51,000–$66,999) Third Quartile Income ($41,000–$50,999) Lowest Quartile Income (b$40,999) Hospital Sizea Large Medium Small Perinatal Conditions Birth Weight Extremely Low (b1000 g) Very Low (b1500 g) Low (b2500 g) Normal Birth Term Full Premature (b37 weeks gestation) Congenital Anomalies Esophageal Spitz II or III Isolated EA ± TEF Long Gap VACTERL (2+ anomalies)b VACTERL (3+ anomalies) Cardiacc Persistent Ductus Arteriosus Otolaryngologic Genitourinary Abdominal Anorectal Malformation Small Bowel Atresia Abdominal Wall Defects Other Digestive Brain/Central Nervous System Chromosomal Pulmonary Congenital Diaphragmatic Hernia
n (%)
1449 (46) 1708 (54) 1516 (48) 1507 (48) 129 (4) 764 (24) 858 (27) 761 (24) 743 (24) 2444 (77) 485 (15) 228 (7)
79 (3) 230 (7) 707 (22) 2141 (68) 1973 (62) 1184 (38)
1828 (58) 596 (19) 92 (3) 2181 (69) 1113 (35) 1679 (53) 1284 (41) 763 (24) 728 (23) 598 (19) 316 (10) 136 (4) 38 (1) 154 (5) 268 (9) 278 (9) 104 (3) 33 (1)
a Hospital size definition varies by United States geographic region and hospital type (rural, urban nonteaching, urban teaching) [30]. b VACTERL includes two or more features of the association of Vertebral, Anorectal, Cardiac, Tracheoesophageal fistula, Renal and/or Limb anomalies. c Major cardiac structural anomaly (excludes persistent ductus arteriosus).
ninth Revision, Clinical Modification (ICD-9CM) code 750.3. We excluded 1551 patients who were transferred out of the birth hospital for definitive care, as these patients were solely born, resuscitated and no procedures were performed. Initial demographics and mortality statistics utilized all remaining 3157 patients. We then performed analysis on readmissions in infants that underwent definitive esophageal atresia repair and survived their index admissions. ICD-9CM procedure codes (31.73, 42.40, 42.41, 41.42, 42.51, 42.52, 42.53, 42.53, 42.34, 42.55, 42.55, 42.56, 42.58, 42.59, 42.84) were utilized to determine surgical repair of EA ± TEF. Discharge weight variables provided by the NRD were utilized to calculate nationwide estimates. Hospital readmissions were defined as hospital admission within 30 days and 1 year after discharge from a hospital within the same state that the surgical repair occurred (index hospitalization). The NRD provides primary and secondary ICD-9CM diagnoses and procedure codes for these admissions. Associated congenital anomalies, perinatal conditions, operative procedures, complications, and resource utilization with length of stay (LOS) and costs were analyzed. Statistical analysis was performed using IBM SPSS Statistics, version 24 (International Business Machines Corp, Armonk, New York). Demographics are reported as n (%). Quantitative variables are reported as mean ± SD for normally distributed variables or median (IQR) for nonparametric variables. Univariate associations of quantitative variables were analyzed using Student's t-test or Mann–Whitney U test, as appropriate. Qualitative variables were analyzed by cross-tabulation and the chi-square statistic. Statistical significance was defined by p b 0.05. This retrospective comparative analysis was deemed exempt from review from the University of Miami Miller School of Medicine IRB committee.
Table 2 Index hospitalization operative procedures and complications. Characteristic Procedures Major Operative Procedure Esophageal Anastomosis ± Fistula Ligation Excision/Resection Stricture Esophageal Dilation Otolaryngologic Tracheostomy Ear/Mouth/Pharynx Larynx/Trachea Abdominal (excluding gastrostomy) Bowel Repair Small Bowel Resection Large Bowel Resection Fundoplication Gastrostomy Cardiac Genitourinary Neurosurgical ECMO CDH Repair Complications Esophagusa GERD Stricture Perforation/Laceration Respiratory Distress Syndrome Hypotension Pneumonia Intraventricular Hemorrhage Stomachb Disorders of Urinary System Hernia Necrotizing Enterocolitis Hydrocephalus Reopening of Surgical Site a b
n (%) 2516 (80) 2313 (73) 2098 (66) 564 (18) 215 (7) 1216 (39) 60 (2) 131 (4) 2183 (69) 670 (21) 31 (1) 70 (2) 22 (1) 143 (5) 723 (23) 141 (5) 129 (4) 19 (1) 37 (1) 12 (0.5) 815 (26) 685 (22) 89 (3) 23 (1) 626 (20) 268 (9) 120 (4) 120 (4) 105 (3) 99 (3) 92 (3) 75 (2) 62 (2) 25 (1)
Includes achalasia, stricture, perforation, dyskinesia, GERD. Includes gastrostomy infection/complication, gastroparesis, dyspepsia.
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
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conditions during the index hospitalization most commonly involved the esophagus (26%) including GERD (22%) and esophageal stricture (3%). Other complications included neonatal respiratory distress, pneumonia, intraventricular hemorrhage, enterocolitis among others.
2. Results 2.1. Birth hospitalization 3157 newborns with EA ± TEF were identified during the study period. Demographic data are displayed in Table 1. The cohort was 54% (n = 1708) male and had an average gestational age of 34 ± 4 weeks at birth. More than a third of patients were low birth weight and/or premature. Eighty-one percent (n = 2557) had an additional congenital anomaly, with 35% having VACTERL association (includes three or more features of the association of Vertebral, Anorectal, Cardiac, TracheoEsophageal fistula, Renal and/or Limb anomalies). Only 42% (n = 1330) were considered Spitz I classification (N1.5 kg, no major cardiac abnormality), while 53% (n = 1669) were Spitz II (b1.5 kg or major cardiac anomaly), and 5% (n = 158) were Spitz III (b 1.5 kg and major cardiac anomaly). The most common congenital anomalies were cardiac, otolaryngologic, and genitourinary. Operative procedures and complications during the newborn hospitalization are shown in Table 2. The vast majority (80%) underwent a major operative procedure during the newborn period. There were 92 patients (3%) with suspected isolated esophageal atresia without TEF as they only underwent gastrostomy and/or esophagostomy during neonatal hospitalization. Additional operations most commonly were otolaryngologic, abdominal, cardiac and/or genitourinary. Associated
2.2. Mortality The overall mortality during newborn index hospitalization was 11% (n = 360). There were 170 (5%) who died within the first week of life without an operation for the EA ± TEF. Univariate predictors of mortality are shown in Table 3. Premature neonates (b 37 weeks' gestation, 20% mortality) and those who were extremely low birth weight (ELBW, 65% mortality) or very low birth weight (VLBW, 58% mortality) experienced higher mortality (all, p b 0.01). The mean gestational age for survivors was 35 ± 4 weeks vs. 31 ± 5 weeks for those that died, p b 0.001. Associated congenital anomalies involving nearly any additional system conferred higher mortality in the neonatal period. This includes diseases affecting the brain, cardiac, bowel atresia/anorectal malformation, genitourinary, congenital diaphragmatic hernia (CDH), or chromosomal anomalies. Likewise, those with higher Spitz classification (II/III) or VACTERL association experienced higher mortality compared to those without these characteristics. The highest mortality was seen in those with CDH (64%) or chromosomal anomalies (46%).
Table 3 Univariate predictors of morality in neonates with esophageal atresia (NRD, 2010–2014). Variable Demographics Gender VLBW (b1500 g) LBW (b2500 g) Premature (b37 weeks gestation) Congenital Anomalies Any Associated Anomaly Central Nervous System Major Cardiac Small Bowel Atresia Anorectal Malformation Genitourinary CDH Chromosomal VACTERL Spitz II/III Procedures Cardiac Ostomy CDH Repair Genitourinary Blood Transfusion ECMO
Survived n (%)
Died n (%)
p=
Female Male Yes No Yes No Yes No
1286 (89) 1520 (89) 97 (42) 2709 (93) 648 (92) 2158 (88) 943 (80) 1883 (94)
163 (11) 188 (11) 133 (58) 218 (7) 59 (8) 292 (12) 241 (20) 110 (6)
0.83
Yes Isolated EA ± TEF Yes No Yes No Yes No Yes No Yes No Yes No Yes No Yes No Yes Spitz I
2235 (87) 571 (96) 175 (65) 2631 (91) 1466 (87) 1340 (91) 101 (74) 2705 (90) 206 (65) 2600 (92) 441 (75) 2365 (92) 12 (36) 2794 (89) 150 (54) 2656 (92) 896 (81) 980 (92) 1506 (82) 1300 (98)
326 (13) 25 (4) 93 (35) 259 (9) 213 (13) 139 (9) 35 (26) 317 (11) 110 (35) 241 (9) 151 (26) 201 (8) 21 (64) 331 (11) 128 (46) 223 (8) 216 (19) 89 (8) 322 (18) 30 (2)
Yes No Yes No Yes No Yes No Yes No Yes No
92 (65) 2714 (90) 200 (79) 2606 (90) -- (17) 2804 (89) 85 (69) 2721 (90) 412 (88) 2394 (89) 85 (79) 2721 (89)
49 (35) 303 (10) 53 (21) 299 (10) -- (83) 341 (11) 39 (32) 312 (10) 54 (12) 298 (11) 23 (21) 329 (11)
b0.01 b0.01 b0.01
b0.01 b0.01 b0.01 b0.01 b0.01 b0.01 b0.01 b0.01 b0.01 b0.01
b0.01 b0.01 b0.01 b0.01 0.743 b0.01
VLBW, very low birth weight; LBW, low birth weight; CDH, congenital diaphragmatic hernia; ECMO, extracorporeal membrane oxygenation. “–”: signifies data usage per HCUP standards.
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
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2.3. 30-day readmissions
2.4. 1-year readmissions
Of neonates that received surgical repair for their EA ± TEF and survived the index admissions (n = 2179), 10% (n = 212) were readmitted within 30-days. Of these, 90% were readmitted to their index hospital and 10% to a different hospital. Factors associated with readmission are shown in Table 4. Female gender, low birth weight (VLBW, LBW) and prematurity were associated with a higher rate of 30-day readmission. Likewise, those with additional anomalies including otolaryngologic, cardiac, small bowel atresia, CDH, chromosomal anomalies were more likely to be readmitted within 30 days. Readmissions were more common in those receiving additional operations during the neonatal period vs. those without these operations. This included esophagostomies (23% vs. 9%), fundoplication (20% vs. 9%), gastrostomy tubes (19% vs. 7%), or ostomies (24% vs. 9%), all p b 0.001. Not surprisingly, those requiring more than one thoracotomy in the neonatal period had the highest readmission rate vs. those without (44% vs. 9%, p b 0.001). Complications (listed in Table 4) during index hospitalization also increased the risk of being readmitted to the hospital within 30-days of discharge. When compared to neonates without these complications, those with surgical site dehiscence, meconium obstruction, GERD, or intraventricular hemorrhage were more likely to be readmitted within 30 days.
The 1-year readmission rate was 26% with 17% admitted to a different hospital. Children with Medicaid insurance and those within the lower half of the income stratum were more likely to be readmitted than their counterparts (30% vs. 22% and 28% vs. 24%, respectively, all p b 0.05). Additional data on 1-year readmissions are presented in Table 4. The majority of factors associated with an increased 30-day readmission rate were also associated with increased readmission at 1 year. However, patients with brain/ CNS disorders, anorectal malformations, genitourinary anomalies, or VACTERL had an increased 1-year (but not 30-day) readmission rate. Patients eventually readmitted had longer index hospital lengths of stay (36 [22–74] days) compared to those never readmitted (27 [17–58] days), p b 0.001. This increased length of hospital stay translated to higher index hospitalization charges ($286,115 [$180,083–544,486]) for those readmitted vs. those not readmitted ($199,754 [$117,817–303,529]), p b 0.001. 2.5. Readmission characteristics The average time from neonatal discharge to first readmission was 65 ± 59 days. The majority of readmissions (85%) were unplanned for a variety of conditions, data shown in Table 5. Common diagnoses
Table 4 Univariate predictors of readmission based on index hospitalization factors in neonates after esophageal atresia repair (NRD, 2010–2014).
Demographics Gender VLBW (b1500 g) LBW (b2500 g) Premature (b37 weeks gestation) Congenital Anomalies Any Additional Anomaly Central Nervous System Major Cardiac Small Bowel Atresia Anorectal Malformation Genitourinary CDH Chromosomal VACTERL Spitz II/III Complications & Associated Diagnoses Surgical Site Dehiscence Respiratory Distress Syndrome Meconium Obstruction GERD Intraventricular Hemorrhage
30-day readmission 212 (10%)
1-year readmission 563 (26%)
Female Male Yes No Yes No Yes No
125 (13) † 87 (7) 20 (27) † 192 (9) 96 (18) † 115 (7) 112 (15) † 99 (7)
273 (28) 290 (24) 41 (57) † 521 (25) 117 (32) † 386 (24) 251 (34) † 312 (22)
Yes Isolated EA ± TEF Yes No Yes No Yes No Yes No Yes No Yes No Yes No Yes No Yes Spitz I
190 (10) ⁎⁎ 22 (6) -- (5) 206 (10) 162 (13) † 50 (5) 21 (27) † 190 (9) 20 (12) 191 (10) 39 (8) 172 (10) -- (60) † 205 (10) 42 (42) † 169 (8) 73 (10) 110 (13) ⁎⁎
506 (28) ⁎⁎ 56 (16) 44 (38) † 518 (25) 396 (32) † 167 (18) 29 (38) ⁎⁎ 533 (25) 84 (49) † 479 (24) 183 (39) † 379 (24) -- (60) ⁎⁎ 556 (26) 60 (60) † 502 (24) 275 (36) † 210 (25) 399 (32) † 164 (18)
Yes No Yes No Yes No Yes No Yes No
-- (40) † 204 (9) 43 (12) 169 (9) 14 (74) † 197 (9) 89 (15) † 122 (8) 13 (23) † 197 (9)
163 (13) † 49 (5)
12 (63) † 551 (26) 112 (31) ⁎⁎ 451 (25) 14 (74) † 549 (25) 209 (35) † 354 (22) 23 (38) ⁎⁎ 540 (26)
VLBW, very low birth weight; LBW, low birth weight; CDH, congenital diaphragmatic hernia; SIP, spontaneous intestinal perforation; GERD, gastroesophageal reflux disease. “—”: signifies data usage per HCUP standards. ⁎⁎ pb0.05 † pb0.01
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
H.J. Quiroz et al. / Journal of Pediatric Surgery xxx (xxxx) xxx Table 5 Readmission procedures and diagnoses in infants with EA ± TEF after surgical repair.
Readmitted Within 1 Year Elective Readmissiona Unplanned Readmissionb Procedure Major Operative Procedure EGD/Dilation Nissen or Gastrostomy Tube Bronchoscopy Anorectal Malformation Repair Cardiac Repair CDH Repair TEF/Esophageal Repair Diagnoses on Admission GERD Infections Disease of Respiratory System GERD Complicationc Failure to Thrive Tracheomalacia Esophageal Stricture Nausea, vomiting, diarrhea Upper Respiratory Infection Septicemia Gastrostomy Complications Apparent Life-Threatening Event (ALTE) Pneumonia Laryngeal (other) Esophageal Perforation
n
%
563 83 480
27 15 85
264 97 67 49 37 20 12 --
47 17 12 9 7 4 2 b1
305 187 172 102 97 78 55 50 46 39 34 29 28 27 14
54 33 31 18 17 14 10 9 8 7 6 5 5 5 3
EGD, esophagogastroduodenoscopy; CDH, congenital diaphragmatic hernia; GERD, gastroesophageal reflux. “–”: signifies data usage per HCUP standards. a Readmission involving elective planned surgical procedures (correction of anorectal malformation, cardiac surgery, genitourinary procedures, etc.) b Readmission for diagnoses or complications other than elective procedures. c Includes aspiration pneumonia, airway complication attributed to GERD, fundoplication performed at readmission.
during readmission were GERD (54%), infections (33%), respiratory diagnoses (31%), GERD-related complications (18%), failure to thrive (17%), and tracheomalacia (14%). Half of those readmitted underwent a procedure during readmission, most commonly esophagoscopy + dilation (17%) or fundoplication/gastrostomy (12%). Eighty-three patients (15%) were readmitted for elective surgery, most commonly for repair of anorectal malformation (45%, n = 37), cardiac repair (24%, n = 20), neurosurgical procedure (15%, n = 12), or inguinal hernia repair ± orchiopexy (11%, n = 9). The overall median charge for readmission was $31,196 ($11,325–115,294) resulting in total charges for readmissions in the cohort N$64 million. Likewise, the median cost for readmission was $10,012 ($4190–42,688) resulting in total cost of readmission of N$23 million. Readmissions within the first 30 days accounted for nearly a third of the total charges ($20 million) and unplanned readmissions accounted for 90% of the overall readmission hospital charges ($57 million). 3. Discussion Owing to rising medical costs in the United States, many agencies are identifying readmissions as a metric to determine quality of care and adjust reimbursements. While this has not yet been implemented in the pediatric population, programs like CHIPRA consider pediatric readmissions a core quality metric [8,9] and thus an important area for research. This study represents the largest, nationally representative study of infants with surgically repaired esophageal atresia including 30-day and 1-year readmissions. Berry et al. performed a large-scale retrospective study and determined the general 30-day readmission rate for pediatric hospitals was 6.5% [13]. Our study encompassing 2179 surgically repaired neonates with EA ± TEF demonstrated a 30-day readmission rate of 10% with 1
5
in 10 being to a different hospital. Furthermore, our study extends readmission rates up to one year and reveals that one in four children are eventually rehospitalized and 17% of these will be readmitted to a different hospital. Institutional studies have shown that 47% of EA ± TEF are associated with an additional anomaly with half of these owing to recognizable malformation syndromes such as VACTERL association [14–16]. Our large cohort study found that 81% of neonates with EA ± TEF had additional anomalies with 35% VACTERL association and 53% with cardiac anomalies. HCUP databases such as the NRD include ICD-9 codes for up to 30 diagnoses, which allow thorough classification of associated anomalies. Our reported rates align with previous large cohort studies of the PHIS [2] and KID [7]. Our analysis also demonstrated that neonates with Spitz II or III classification of EA ± TEF comprised 92% of all mortalities occurring over the study period, further illustrating that the Spitz classification remains an important prognostic factor for these neonates [17]. Although a 10% 30-day readmission rate is elevated in comparison to the general pediatric population, certain pediatric conditions such as hematological disorders have 30-day readmissions rates as high as 22% [13]. An analysis of the PHIS demonstrated a 17% 30-day readmission rate for children with EA ± TEF [2]. However, this study only captures free-standing children's hospitals that are part of the Children's Hospital Association and they do not clearly identify their methodology for readmission. Upon further stratification of risk for readmission, certain factors significantly increased a patient's likelihood of being readmitted both at 30-day and one-year intervals. Children with Medicaid insurance and those within the lower half of the income stratum were more likely to be readmitted than their counterparts at the oneyear interval. This is consistent with studies of children in lower socioeconomic communities that have also shown that readmissions rates are higher. [18–20] The NRD unfortunately lacks variables on race/ethnicity; however, many studies have shown that race does in fact have an effect on readmission rates [19–21]. This socioeconomic impact is thought to be partially owing to a lack of reliable outpatient care or medical homes to aid in the needs of medically-complex children [22,23]. Previously unreported, our analysis demonstrates that certain congenital and perinatal factors significantly impacted readmission rates. The vast majority of congenital anomalies were associated with increased rates of readmission at both 30-day and 1-year intervals. Likewise, undergoing additional nonesophageal operations and sustaining complications at the index hospitalization were associated with increased likelihood of readmission. The etiology for these findings is likely multifactorial. Often after discharge from the hospital, infants with EA ± TEF are seen by their primary providers and surgeons more frequently than unaffected newborns. Conditions such as EA ± TEF, especially in conjunction with severe associated anomalies, lead to exceedingly complex problems which require carefully coordinated care after discharge. Taken together, patients with EA ± TEF and additional comorbid conditions should be considered “children with medical complexity” and should be considered for coordinated care efforts [24–26]. In addition to detailing the readmission rates for the general EA ± TEF population, we reported the readmission characteristics to better understand the potential pitfalls of the complex care of infants with EA ± TEF. Our study demonstrates that close to 50% of infants with EA ± TEF will eventually undergo a major operative procedure upon readmission, the majority of these being for unplanned readmissions with esophagoscopy and dilation procedures (17%) and secondarily for fundoplication or gastrostomy placement (12%). Fewer (15%) children were readmitted for elective repairs of their concomitant congenital anomalies (ARM, cardiac, inguinal hernias, etc.). While there are no studies that detail one-year reoperative outcomes with readmissions, the previously cited PHIS study reported data on two-year outcomes (whether or not at readmissions). According to this study, 11% will undergo eventual esophageal reconstruction, 12% will undergo fundoplication, and 18%
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
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will require gastrostomy tube placement. [2] Our data present similar rates at 1-year based solely on an inpatient readmission setting. The present study also investigated the financial burden of neonatal hospitalization and readmission in infants with EA ± TEF. Nationwide studies using the National Inpatient Sample (another HCUP database) have reported median hospital cost for gastroschisis of $75,859. [27] Significant regional variation in hospital cost has been demonstrated for infants with hypoplastic left heart syndrome, with higher hospital cost in the Western and Southern United States. [28] In California, the mean cost of hospitalization for all newborns is reported to be $6,389 and is significantly higher for late preterm ($22,102) and very preterm infants ($223,931) compared to term infants ($2,433) [29]. Our study unveiled a median cost for index hospitalization of $72,916 which was significantly higher for those who went on to be readmitted (median index cost $97,812). The observed higher index cost for those eventually readmitted is likely owing to the prolonged index hospital length of stay (36 vs. 27 days). These findings suggest that complex associated anomalies/comorbid perinatal conditions result in longer lengths of stay, higher hospital costs and increased rate of eventual readmission. These readmissions account for a significant overall healthcare cost with total cost of readmission N $21 million, 90% of which is the cost of unplanned readmissions. The limitations of this study are consistent with studies of administrative databases. First, the study design is a retrospective review of the NRD, which lends itself to errors in data sampling, collection measures, usage of the ICD-9 coding scheme, and possible administrative errors during data entry. As with any study using NRD, readmissions across state lines are unable to be captured, though this phenomenon is likely rare. Important perioperative factors such as timing of operative intervention or complications are not recorded in NRD. Likewise, the criteria to determine reason for hospital readmission are left to the analyst using the database and there is some degree of inference made based on the associated ICD-9 diagnostic and procedure codes. Lastly, certain conditions and postoperative complications are treated in the outpatient setting, which is not captured in this inpatient database. Therefore, the 17% esophageal stricture rate is likely an underestimate of the true incidence as scheduled outpatient procedures are not captured by this inpatient database. 4. Conclusion The present study has uncovered a high likelihood of readmission within the first year of life for newborns with esophageal atresia with the majority being unplanned readmissions for a variety of conditions. Coordinated, multidisciplinary care may help to decrease unnecessary readmissions and improve outcomes in this vulnerable population. Funding No funding was provided for the performance of this study. Conflict of interest All authors declare no conflicts of interest. Disclosure NRD and the hospitals utilized for its data accrual are the source of the data used herein; they have not verified and are not responsible for the statistical validity of the data analysis or the conclusions derived by the authors.
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A nationwide analysis of clinical outcomes among newborns with esophageal atresia and tracheoesophageal fistulas in the United States. Journal of Surgical Research 2014;190(2):604–12. [8] Agency for Healthcare Research and Quality. All Pediatric Quality Measures Program (PQMP) measures; 2019. [9] Dougherty D, Schiff J, Mangione-Smith R. The Children's Health Insurance Program Reauthorization Act quality measures initiatives: moving forward to improve measurement, care, and child and adolescent outcomes. Acad Pediatr 2011;11(3, Supplement):S1–S10. [10] Centers for Medicare and Medicaid Services (CMS). Parternship for patients; 2019. [11] Lyren A, Coffey M, Shepherd M, et al. We will not compete on safety: how children's hospitals have come together to hasten harm reduction. Jt Comm J Qual Patient Saf 2018;44(7):377–88. [12] Nolan BW, De Martino RR, Goodney PP, et al. Comparison of carotid endarterectomy and stenting in real world practice using a regional quality improvement registry. 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A retrospective. multicenter study Pediatrics 2009;123(1):286–93. [19] Berry JG, Hall DE, Kuo DZ, et al. Hospital utilization and characteristics of patients experiencing recurrent readmissions within children's hospitals. JAMA 2011;305 (7):682–90. [20] Joynt KE, Orav EJ, Jha AK. Thirty-day readmission rates for Medicare beneficiaries by race and site of care. JAMA 2011;305(7):675–81. [21] Hernandez AF, Curtis LH. Minding the gap between efforts to reduce readmissions and disparities. JAMA 2011;305(7):715–6. [22] Cassedy A, Fairbrother G, Newacheck PW. The impact of insurance instability on children's access, utilization, and satisfaction with health care. Ambulatory pediatrics : the official journal of the Ambulatory Pediatric Association 2008;8(5):321–8. [23] Zickafoose JS, Gebremariam A, Clark SJ, et al. Medical home disparities between children with public and private insurance. Acad Pediatr 2011;11(4):305–10. [24] Cohen E, Friedman J, Nicholas DB, et al. A home for medically complex children: the role of hospital programs. Journal for healthcare quality : official publication of the National Association for Healthcare Quality 2008;30(3):7–15. [25] Cohen E, Jovcevska V, Kuo DZ, et al. Hospital-based comprehensive care programs for children with special health care needs: a systematic review. Arch Pediatr Adolesc Med 2011;165(6):554–61. [26] Mosquera RA, Avritscher EB, Samuels CL, et al. Effect of an enhanced medical home on serious illness and cost of care among high-risk children with chronic illness: a randomized clinical trial. JAMA 2014;312(24):2640–8. [27] Bhatt P, Lekshminarayanan A, Donda K, et al. Trends in incidence and outcomes of gastroschisis in the United States: analysis of the national inpatient sample 20102014. Pediatr Surg Int 2018;34(9):919–29. [28] Essaid L, Strassle PD, Jernigan EG, et al. Regional differences in cost and length of stay in neonates with hypoplastic left heart syndrome. 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Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
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Nationwide analysis of mortality and hospital readmissions in esophageal atresia Hallie J. Quiroz a, Alexa Turpin b, Brent A. Willobee a, Anthony Ferrantella a, Joshua Parreco c, David Lasko d, Eduardo A. Perez a, Juan E. Sola a, Chad M. Thorson a,⁎ a
Dewitt-Daughtry Family Department of Surgery, Division of Pediatric Surgery, University of Miami Miller School of Medicine University of Miami Miller School of Medicine Dewitt-Daughtry Family Department of Surgery, Division of Trauma and Acute Care Surgery, University of Miami Miller School of Medicine d South Florida Pediatric Surgeons b c
a r t i c l e
i n f o
Article history: Received 14 January 2020 Accepted 25 January 2020 Available online xxxx Key words: Esophageal atresia Outcomes Readmission Infants
a b s t r a c t Purpose: The purpose of this study is to identify determinants of mortality and hospital readmission in infants born with esophageal atresia ± tracheoesophageal fistula. Methods: The Nationwide Readmissions Database (2010–2014) was queried for newborns with a diagnosis of esophageal atresia. Outcomes included mortality and readmissions at 30-day and 1-year. Results: 3157 patients were identified, of which 54% were male. 81% had an additional congenital anomaly, and 35% had VACTERL association. Overall mortality at index hospitalization was 11% (n = 360) and was significantly higher with additional congenital anomalies (13%), VACTERL (19%), and Spitz classification II/III (18%) vs. isolated esophageal atresia/tracheoesophageal fistula (4%), all p b 0.001. After esophageal atresia repair (n = 2179), 10% (n = 212) were readmitted within 30 days and 26% (n = 563) within 1 year, with 17% admitted to different hospitals. Common diagnoses during readmission were GERD (54%), infections (42%), failure to thrive (17%), tracheomalacia (14%), and esophageal stricture (10%). Unplanned readmissions accounted for 85% of readmissions. A large number underwent operative procedures, most commonly esophageal dilation (17%) and fundoplication/gastrostomy (12%). Conclusion: Our study has uncovered a high likelihood of complications and unplanned readmission within the first year of life for newborns with esophageal atresia. Coordinated multidisciplinary care may help to decrease unnecessary readmissions and improve outcomes in this vulnerable population. Type of study: Retrospective comparative analysis. Level of evidence: Level III. © 2020 Elsevier Inc. All rights reserved.
Esophageal atresia (EA) is a congenital anomaly characterized by abnormal formation of the primitive foregut leading to discontinuity between the proximal and distal portions of the esophagus, which often occurs in association with tracheoesophageal fistula (TEF). Prior to the first successful operation for EA, this congenital anomaly conferred a 100% mortality rate. [1] With contemporary surgical management and advances in neonatal care, survival is typically dependent upon the presence and severity of associated anomalies or conditions such as low birth weight. [2] Infants with EA ± TEF often suffer complications such as severe gastroesophageal reflux (GERD), esophageal strictures, chronic dysphagia,
⁎ Corresponding author at: University of Miami Miller School of Medicine, Division of Pediatric Surgery, P.O. Box 016960 (R-51), Miami, FL. Tel.: +1 305 243 2247; fax: +1 305 243 5731. E-mail address: [email protected] (C.M. Thorson).
recurrent fistula, pulmonary infections, reactive airway disease, and tracheomalacia. [3–6] The majority of existing literature reporting these complications comes from institutional case series limited by small sample size. Large-scale nationwide analyses have been reported utilizing the Pediatric Health Information System (PHIS) [2] and the Kid's Inpatient Database (KID) [7]. However, these databases track a single inpatient hospitalization leading to paucity of long-term outcomes data regarding hospital readmissions and complications. Programs such as the Children's Health Insurance Program Reauthorization Act (CHIPRA) have identified readmissions as a quality core metric in development [8,9] and the federal Partnership for Patients initiative has challenged hospitals to reduce their readmissions by 20%. [10,11] Given the importance of reducing future readmissions, studies are needed to identify baseline readmissions for complex diseases in order to improve hospital protocols. The present study represents a large, nationwide analysis of risk factors during neonatal and subsequent hospital readmissions in neonates born with EA ± TEF. We
https://doi.org/10.1016/j.jpedsurg.2020.01.025 0022-3468/© 2020 Elsevier Inc. All rights reserved.
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
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hypothesized that patients with EA ± TEF would have a high readmission rate owing to underlying medical complexity. 1. Methods The Nationwide Readmissions Database (NRD) is part of a family of databases developed by the Healthcare Cost and Utilization Project (HCUP). This database was developed to address a large gap in healthcare data — the lack of nationally representative information on hospital readmissions for all ages. The NRD contains approximately 17 million unweighted discharges each year and discharge data from 22 geographically dispersed States, accounting for 49% of all United States hospitalizations [12]. The NRD also has the unique ability to track patients' readmissions to different hospitals by using distinct, patient specific identifiers within the database, thus making it an ideal database for studies on nationwide readmissions. The NRD for 2010–2014 was queried for all newborn patients with a diagnosis of EA ± TEF utilizing the International Classification of Disease
Table 1 Neonates with esophageal atresia, demographic data. Characteristic Demographics Gender Female Male Insurance Status Medicaid Private Insurance Self-Pay Income (Quartile) Highest Quartile Income ($67,000 +) Second Quartile Income ($51,000–$66,999) Third Quartile Income ($41,000–$50,999) Lowest Quartile Income (b$40,999) Hospital Sizea Large Medium Small Perinatal Conditions Birth Weight Extremely Low (b1000 g) Very Low (b1500 g) Low (b2500 g) Normal Birth Term Full Premature (b37 weeks gestation) Congenital Anomalies Esophageal Spitz II or III Isolated EA ± TEF Long Gap VACTERL (2+ anomalies)b VACTERL (3+ anomalies) Cardiacc Persistent Ductus Arteriosus Otolaryngologic Genitourinary Abdominal Anorectal Malformation Small Bowel Atresia Abdominal Wall Defects Other Digestive Brain/Central Nervous System Chromosomal Pulmonary Congenital Diaphragmatic Hernia
n (%)
1449 (46) 1708 (54) 1516 (48) 1507 (48) 129 (4) 764 (24) 858 (27) 761 (24) 743 (24) 2444 (77) 485 (15) 228 (7)
79 (3) 230 (7) 707 (22) 2141 (68) 1973 (62) 1184 (38)
1828 (58) 596 (19) 92 (3) 2181 (69) 1113 (35) 1679 (53) 1284 (41) 763 (24) 728 (23) 598 (19) 316 (10) 136 (4) 38 (1) 154 (5) 268 (9) 278 (9) 104 (3) 33 (1)
a Hospital size definition varies by United States geographic region and hospital type (rural, urban nonteaching, urban teaching) [30]. b VACTERL includes two or more features of the association of Vertebral, Anorectal, Cardiac, Tracheoesophageal fistula, Renal and/or Limb anomalies. c Major cardiac structural anomaly (excludes persistent ductus arteriosus).
ninth Revision, Clinical Modification (ICD-9CM) code 750.3. We excluded 1551 patients who were transferred out of the birth hospital for definitive care, as these patients were solely born, resuscitated and no procedures were performed. Initial demographics and mortality statistics utilized all remaining 3157 patients. We then performed analysis on readmissions in infants that underwent definitive esophageal atresia repair and survived their index admissions. ICD-9CM procedure codes (31.73, 42.40, 42.41, 41.42, 42.51, 42.52, 42.53, 42.53, 42.34, 42.55, 42.55, 42.56, 42.58, 42.59, 42.84) were utilized to determine surgical repair of EA ± TEF. Discharge weight variables provided by the NRD were utilized to calculate nationwide estimates. Hospital readmissions were defined as hospital admission within 30 days and 1 year after discharge from a hospital within the same state that the surgical repair occurred (index hospitalization). The NRD provides primary and secondary ICD-9CM diagnoses and procedure codes for these admissions. Associated congenital anomalies, perinatal conditions, operative procedures, complications, and resource utilization with length of stay (LOS) and costs were analyzed. Statistical analysis was performed using IBM SPSS Statistics, version 24 (International Business Machines Corp, Armonk, New York). Demographics are reported as n (%). Quantitative variables are reported as mean ± SD for normally distributed variables or median (IQR) for nonparametric variables. Univariate associations of quantitative variables were analyzed using Student's t-test or Mann–Whitney U test, as appropriate. Qualitative variables were analyzed by cross-tabulation and the chi-square statistic. Statistical significance was defined by p b 0.05. This retrospective comparative analysis was deemed exempt from review from the University of Miami Miller School of Medicine IRB committee.
Table 2 Index hospitalization operative procedures and complications. Characteristic Procedures Major Operative Procedure Esophageal Anastomosis ± Fistula Ligation Excision/Resection Stricture Esophageal Dilation Otolaryngologic Tracheostomy Ear/Mouth/Pharynx Larynx/Trachea Abdominal (excluding gastrostomy) Bowel Repair Small Bowel Resection Large Bowel Resection Fundoplication Gastrostomy Cardiac Genitourinary Neurosurgical ECMO CDH Repair Complications Esophagusa GERD Stricture Perforation/Laceration Respiratory Distress Syndrome Hypotension Pneumonia Intraventricular Hemorrhage Stomachb Disorders of Urinary System Hernia Necrotizing Enterocolitis Hydrocephalus Reopening of Surgical Site a b
n (%) 2516 (80) 2313 (73) 2098 (66) 564 (18) 215 (7) 1216 (39) 60 (2) 131 (4) 2183 (69) 670 (21) 31 (1) 70 (2) 22 (1) 143 (5) 723 (23) 141 (5) 129 (4) 19 (1) 37 (1) 12 (0.5) 815 (26) 685 (22) 89 (3) 23 (1) 626 (20) 268 (9) 120 (4) 120 (4) 105 (3) 99 (3) 92 (3) 75 (2) 62 (2) 25 (1)
Includes achalasia, stricture, perforation, dyskinesia, GERD. Includes gastrostomy infection/complication, gastroparesis, dyspepsia.
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
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conditions during the index hospitalization most commonly involved the esophagus (26%) including GERD (22%) and esophageal stricture (3%). Other complications included neonatal respiratory distress, pneumonia, intraventricular hemorrhage, enterocolitis among others.
2. Results 2.1. Birth hospitalization 3157 newborns with EA ± TEF were identified during the study period. Demographic data are displayed in Table 1. The cohort was 54% (n = 1708) male and had an average gestational age of 34 ± 4 weeks at birth. More than a third of patients were low birth weight and/or premature. Eighty-one percent (n = 2557) had an additional congenital anomaly, with 35% having VACTERL association (includes three or more features of the association of Vertebral, Anorectal, Cardiac, TracheoEsophageal fistula, Renal and/or Limb anomalies). Only 42% (n = 1330) were considered Spitz I classification (N1.5 kg, no major cardiac abnormality), while 53% (n = 1669) were Spitz II (b1.5 kg or major cardiac anomaly), and 5% (n = 158) were Spitz III (b 1.5 kg and major cardiac anomaly). The most common congenital anomalies were cardiac, otolaryngologic, and genitourinary. Operative procedures and complications during the newborn hospitalization are shown in Table 2. The vast majority (80%) underwent a major operative procedure during the newborn period. There were 92 patients (3%) with suspected isolated esophageal atresia without TEF as they only underwent gastrostomy and/or esophagostomy during neonatal hospitalization. Additional operations most commonly were otolaryngologic, abdominal, cardiac and/or genitourinary. Associated
2.2. Mortality The overall mortality during newborn index hospitalization was 11% (n = 360). There were 170 (5%) who died within the first week of life without an operation for the EA ± TEF. Univariate predictors of mortality are shown in Table 3. Premature neonates (b 37 weeks' gestation, 20% mortality) and those who were extremely low birth weight (ELBW, 65% mortality) or very low birth weight (VLBW, 58% mortality) experienced higher mortality (all, p b 0.01). The mean gestational age for survivors was 35 ± 4 weeks vs. 31 ± 5 weeks for those that died, p b 0.001. Associated congenital anomalies involving nearly any additional system conferred higher mortality in the neonatal period. This includes diseases affecting the brain, cardiac, bowel atresia/anorectal malformation, genitourinary, congenital diaphragmatic hernia (CDH), or chromosomal anomalies. Likewise, those with higher Spitz classification (II/III) or VACTERL association experienced higher mortality compared to those without these characteristics. The highest mortality was seen in those with CDH (64%) or chromosomal anomalies (46%).
Table 3 Univariate predictors of morality in neonates with esophageal atresia (NRD, 2010–2014). Variable Demographics Gender VLBW (b1500 g) LBW (b2500 g) Premature (b37 weeks gestation) Congenital Anomalies Any Associated Anomaly Central Nervous System Major Cardiac Small Bowel Atresia Anorectal Malformation Genitourinary CDH Chromosomal VACTERL Spitz II/III Procedures Cardiac Ostomy CDH Repair Genitourinary Blood Transfusion ECMO
Survived n (%)
Died n (%)
p=
Female Male Yes No Yes No Yes No
1286 (89) 1520 (89) 97 (42) 2709 (93) 648 (92) 2158 (88) 943 (80) 1883 (94)
163 (11) 188 (11) 133 (58) 218 (7) 59 (8) 292 (12) 241 (20) 110 (6)
0.83
Yes Isolated EA ± TEF Yes No Yes No Yes No Yes No Yes No Yes No Yes No Yes No Yes Spitz I
2235 (87) 571 (96) 175 (65) 2631 (91) 1466 (87) 1340 (91) 101 (74) 2705 (90) 206 (65) 2600 (92) 441 (75) 2365 (92) 12 (36) 2794 (89) 150 (54) 2656 (92) 896 (81) 980 (92) 1506 (82) 1300 (98)
326 (13) 25 (4) 93 (35) 259 (9) 213 (13) 139 (9) 35 (26) 317 (11) 110 (35) 241 (9) 151 (26) 201 (8) 21 (64) 331 (11) 128 (46) 223 (8) 216 (19) 89 (8) 322 (18) 30 (2)
Yes No Yes No Yes No Yes No Yes No Yes No
92 (65) 2714 (90) 200 (79) 2606 (90) -- (17) 2804 (89) 85 (69) 2721 (90) 412 (88) 2394 (89) 85 (79) 2721 (89)
49 (35) 303 (10) 53 (21) 299 (10) -- (83) 341 (11) 39 (32) 312 (10) 54 (12) 298 (11) 23 (21) 329 (11)
b0.01 b0.01 b0.01
b0.01 b0.01 b0.01 b0.01 b0.01 b0.01 b0.01 b0.01 b0.01 b0.01
b0.01 b0.01 b0.01 b0.01 0.743 b0.01
VLBW, very low birth weight; LBW, low birth weight; CDH, congenital diaphragmatic hernia; ECMO, extracorporeal membrane oxygenation. “–”: signifies data usage per HCUP standards.
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
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2.3. 30-day readmissions
2.4. 1-year readmissions
Of neonates that received surgical repair for their EA ± TEF and survived the index admissions (n = 2179), 10% (n = 212) were readmitted within 30-days. Of these, 90% were readmitted to their index hospital and 10% to a different hospital. Factors associated with readmission are shown in Table 4. Female gender, low birth weight (VLBW, LBW) and prematurity were associated with a higher rate of 30-day readmission. Likewise, those with additional anomalies including otolaryngologic, cardiac, small bowel atresia, CDH, chromosomal anomalies were more likely to be readmitted within 30 days. Readmissions were more common in those receiving additional operations during the neonatal period vs. those without these operations. This included esophagostomies (23% vs. 9%), fundoplication (20% vs. 9%), gastrostomy tubes (19% vs. 7%), or ostomies (24% vs. 9%), all p b 0.001. Not surprisingly, those requiring more than one thoracotomy in the neonatal period had the highest readmission rate vs. those without (44% vs. 9%, p b 0.001). Complications (listed in Table 4) during index hospitalization also increased the risk of being readmitted to the hospital within 30-days of discharge. When compared to neonates without these complications, those with surgical site dehiscence, meconium obstruction, GERD, or intraventricular hemorrhage were more likely to be readmitted within 30 days.
The 1-year readmission rate was 26% with 17% admitted to a different hospital. Children with Medicaid insurance and those within the lower half of the income stratum were more likely to be readmitted than their counterparts (30% vs. 22% and 28% vs. 24%, respectively, all p b 0.05). Additional data on 1-year readmissions are presented in Table 4. The majority of factors associated with an increased 30-day readmission rate were also associated with increased readmission at 1 year. However, patients with brain/ CNS disorders, anorectal malformations, genitourinary anomalies, or VACTERL had an increased 1-year (but not 30-day) readmission rate. Patients eventually readmitted had longer index hospital lengths of stay (36 [22–74] days) compared to those never readmitted (27 [17–58] days), p b 0.001. This increased length of hospital stay translated to higher index hospitalization charges ($286,115 [$180,083–544,486]) for those readmitted vs. those not readmitted ($199,754 [$117,817–303,529]), p b 0.001. 2.5. Readmission characteristics The average time from neonatal discharge to first readmission was 65 ± 59 days. The majority of readmissions (85%) were unplanned for a variety of conditions, data shown in Table 5. Common diagnoses
Table 4 Univariate predictors of readmission based on index hospitalization factors in neonates after esophageal atresia repair (NRD, 2010–2014).
Demographics Gender VLBW (b1500 g) LBW (b2500 g) Premature (b37 weeks gestation) Congenital Anomalies Any Additional Anomaly Central Nervous System Major Cardiac Small Bowel Atresia Anorectal Malformation Genitourinary CDH Chromosomal VACTERL Spitz II/III Complications & Associated Diagnoses Surgical Site Dehiscence Respiratory Distress Syndrome Meconium Obstruction GERD Intraventricular Hemorrhage
30-day readmission 212 (10%)
1-year readmission 563 (26%)
Female Male Yes No Yes No Yes No
125 (13) † 87 (7) 20 (27) † 192 (9) 96 (18) † 115 (7) 112 (15) † 99 (7)
273 (28) 290 (24) 41 (57) † 521 (25) 117 (32) † 386 (24) 251 (34) † 312 (22)
Yes Isolated EA ± TEF Yes No Yes No Yes No Yes No Yes No Yes No Yes No Yes No Yes Spitz I
190 (10) ⁎⁎ 22 (6) -- (5) 206 (10) 162 (13) † 50 (5) 21 (27) † 190 (9) 20 (12) 191 (10) 39 (8) 172 (10) -- (60) † 205 (10) 42 (42) † 169 (8) 73 (10) 110 (13) ⁎⁎
506 (28) ⁎⁎ 56 (16) 44 (38) † 518 (25) 396 (32) † 167 (18) 29 (38) ⁎⁎ 533 (25) 84 (49) † 479 (24) 183 (39) † 379 (24) -- (60) ⁎⁎ 556 (26) 60 (60) † 502 (24) 275 (36) † 210 (25) 399 (32) † 164 (18)
Yes No Yes No Yes No Yes No Yes No
-- (40) † 204 (9) 43 (12) 169 (9) 14 (74) † 197 (9) 89 (15) † 122 (8) 13 (23) † 197 (9)
163 (13) † 49 (5)
12 (63) † 551 (26) 112 (31) ⁎⁎ 451 (25) 14 (74) † 549 (25) 209 (35) † 354 (22) 23 (38) ⁎⁎ 540 (26)
VLBW, very low birth weight; LBW, low birth weight; CDH, congenital diaphragmatic hernia; SIP, spontaneous intestinal perforation; GERD, gastroesophageal reflux disease. “—”: signifies data usage per HCUP standards. ⁎⁎ pb0.05 † pb0.01
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
H.J. Quiroz et al. / Journal of Pediatric Surgery xxx (xxxx) xxx Table 5 Readmission procedures and diagnoses in infants with EA ± TEF after surgical repair.
Readmitted Within 1 Year Elective Readmissiona Unplanned Readmissionb Procedure Major Operative Procedure EGD/Dilation Nissen or Gastrostomy Tube Bronchoscopy Anorectal Malformation Repair Cardiac Repair CDH Repair TEF/Esophageal Repair Diagnoses on Admission GERD Infections Disease of Respiratory System GERD Complicationc Failure to Thrive Tracheomalacia Esophageal Stricture Nausea, vomiting, diarrhea Upper Respiratory Infection Septicemia Gastrostomy Complications Apparent Life-Threatening Event (ALTE) Pneumonia Laryngeal (other) Esophageal Perforation
n
%
563 83 480
27 15 85
264 97 67 49 37 20 12 --
47 17 12 9 7 4 2 b1
305 187 172 102 97 78 55 50 46 39 34 29 28 27 14
54 33 31 18 17 14 10 9 8 7 6 5 5 5 3
EGD, esophagogastroduodenoscopy; CDH, congenital diaphragmatic hernia; GERD, gastroesophageal reflux. “–”: signifies data usage per HCUP standards. a Readmission involving elective planned surgical procedures (correction of anorectal malformation, cardiac surgery, genitourinary procedures, etc.) b Readmission for diagnoses or complications other than elective procedures. c Includes aspiration pneumonia, airway complication attributed to GERD, fundoplication performed at readmission.
during readmission were GERD (54%), infections (33%), respiratory diagnoses (31%), GERD-related complications (18%), failure to thrive (17%), and tracheomalacia (14%). Half of those readmitted underwent a procedure during readmission, most commonly esophagoscopy + dilation (17%) or fundoplication/gastrostomy (12%). Eighty-three patients (15%) were readmitted for elective surgery, most commonly for repair of anorectal malformation (45%, n = 37), cardiac repair (24%, n = 20), neurosurgical procedure (15%, n = 12), or inguinal hernia repair ± orchiopexy (11%, n = 9). The overall median charge for readmission was $31,196 ($11,325–115,294) resulting in total charges for readmissions in the cohort N$64 million. Likewise, the median cost for readmission was $10,012 ($4190–42,688) resulting in total cost of readmission of N$23 million. Readmissions within the first 30 days accounted for nearly a third of the total charges ($20 million) and unplanned readmissions accounted for 90% of the overall readmission hospital charges ($57 million). 3. Discussion Owing to rising medical costs in the United States, many agencies are identifying readmissions as a metric to determine quality of care and adjust reimbursements. While this has not yet been implemented in the pediatric population, programs like CHIPRA consider pediatric readmissions a core quality metric [8,9] and thus an important area for research. This study represents the largest, nationally representative study of infants with surgically repaired esophageal atresia including 30-day and 1-year readmissions. Berry et al. performed a large-scale retrospective study and determined the general 30-day readmission rate for pediatric hospitals was 6.5% [13]. Our study encompassing 2179 surgically repaired neonates with EA ± TEF demonstrated a 30-day readmission rate of 10% with 1
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in 10 being to a different hospital. Furthermore, our study extends readmission rates up to one year and reveals that one in four children are eventually rehospitalized and 17% of these will be readmitted to a different hospital. Institutional studies have shown that 47% of EA ± TEF are associated with an additional anomaly with half of these owing to recognizable malformation syndromes such as VACTERL association [14–16]. Our large cohort study found that 81% of neonates with EA ± TEF had additional anomalies with 35% VACTERL association and 53% with cardiac anomalies. HCUP databases such as the NRD include ICD-9 codes for up to 30 diagnoses, which allow thorough classification of associated anomalies. Our reported rates align with previous large cohort studies of the PHIS [2] and KID [7]. Our analysis also demonstrated that neonates with Spitz II or III classification of EA ± TEF comprised 92% of all mortalities occurring over the study period, further illustrating that the Spitz classification remains an important prognostic factor for these neonates [17]. Although a 10% 30-day readmission rate is elevated in comparison to the general pediatric population, certain pediatric conditions such as hematological disorders have 30-day readmissions rates as high as 22% [13]. An analysis of the PHIS demonstrated a 17% 30-day readmission rate for children with EA ± TEF [2]. However, this study only captures free-standing children's hospitals that are part of the Children's Hospital Association and they do not clearly identify their methodology for readmission. Upon further stratification of risk for readmission, certain factors significantly increased a patient's likelihood of being readmitted both at 30-day and one-year intervals. Children with Medicaid insurance and those within the lower half of the income stratum were more likely to be readmitted than their counterparts at the oneyear interval. This is consistent with studies of children in lower socioeconomic communities that have also shown that readmissions rates are higher. [18–20] The NRD unfortunately lacks variables on race/ethnicity; however, many studies have shown that race does in fact have an effect on readmission rates [19–21]. This socioeconomic impact is thought to be partially owing to a lack of reliable outpatient care or medical homes to aid in the needs of medically-complex children [22,23]. Previously unreported, our analysis demonstrates that certain congenital and perinatal factors significantly impacted readmission rates. The vast majority of congenital anomalies were associated with increased rates of readmission at both 30-day and 1-year intervals. Likewise, undergoing additional nonesophageal operations and sustaining complications at the index hospitalization were associated with increased likelihood of readmission. The etiology for these findings is likely multifactorial. Often after discharge from the hospital, infants with EA ± TEF are seen by their primary providers and surgeons more frequently than unaffected newborns. Conditions such as EA ± TEF, especially in conjunction with severe associated anomalies, lead to exceedingly complex problems which require carefully coordinated care after discharge. Taken together, patients with EA ± TEF and additional comorbid conditions should be considered “children with medical complexity” and should be considered for coordinated care efforts [24–26]. In addition to detailing the readmission rates for the general EA ± TEF population, we reported the readmission characteristics to better understand the potential pitfalls of the complex care of infants with EA ± TEF. Our study demonstrates that close to 50% of infants with EA ± TEF will eventually undergo a major operative procedure upon readmission, the majority of these being for unplanned readmissions with esophagoscopy and dilation procedures (17%) and secondarily for fundoplication or gastrostomy placement (12%). Fewer (15%) children were readmitted for elective repairs of their concomitant congenital anomalies (ARM, cardiac, inguinal hernias, etc.). While there are no studies that detail one-year reoperative outcomes with readmissions, the previously cited PHIS study reported data on two-year outcomes (whether or not at readmissions). According to this study, 11% will undergo eventual esophageal reconstruction, 12% will undergo fundoplication, and 18%
Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025
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will require gastrostomy tube placement. [2] Our data present similar rates at 1-year based solely on an inpatient readmission setting. The present study also investigated the financial burden of neonatal hospitalization and readmission in infants with EA ± TEF. Nationwide studies using the National Inpatient Sample (another HCUP database) have reported median hospital cost for gastroschisis of $75,859. [27] Significant regional variation in hospital cost has been demonstrated for infants with hypoplastic left heart syndrome, with higher hospital cost in the Western and Southern United States. [28] In California, the mean cost of hospitalization for all newborns is reported to be $6,389 and is significantly higher for late preterm ($22,102) and very preterm infants ($223,931) compared to term infants ($2,433) [29]. Our study unveiled a median cost for index hospitalization of $72,916 which was significantly higher for those who went on to be readmitted (median index cost $97,812). The observed higher index cost for those eventually readmitted is likely owing to the prolonged index hospital length of stay (36 vs. 27 days). These findings suggest that complex associated anomalies/comorbid perinatal conditions result in longer lengths of stay, higher hospital costs and increased rate of eventual readmission. These readmissions account for a significant overall healthcare cost with total cost of readmission N $21 million, 90% of which is the cost of unplanned readmissions. The limitations of this study are consistent with studies of administrative databases. First, the study design is a retrospective review of the NRD, which lends itself to errors in data sampling, collection measures, usage of the ICD-9 coding scheme, and possible administrative errors during data entry. As with any study using NRD, readmissions across state lines are unable to be captured, though this phenomenon is likely rare. Important perioperative factors such as timing of operative intervention or complications are not recorded in NRD. Likewise, the criteria to determine reason for hospital readmission are left to the analyst using the database and there is some degree of inference made based on the associated ICD-9 diagnostic and procedure codes. Lastly, certain conditions and postoperative complications are treated in the outpatient setting, which is not captured in this inpatient database. Therefore, the 17% esophageal stricture rate is likely an underestimate of the true incidence as scheduled outpatient procedures are not captured by this inpatient database. 4. Conclusion The present study has uncovered a high likelihood of readmission within the first year of life for newborns with esophageal atresia with the majority being unplanned readmissions for a variety of conditions. Coordinated, multidisciplinary care may help to decrease unnecessary readmissions and improve outcomes in this vulnerable population. Funding No funding was provided for the performance of this study. Conflict of interest All authors declare no conflicts of interest. Disclosure NRD and the hospitals utilized for its data accrual are the source of the data used herein; they have not verified and are not responsible for the statistical validity of the data analysis or the conclusions derived by the authors.
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Please cite this article as: H.J. Quiroz, A. Turpin, B.A. Willobee, et al., Nationwide analysis of mortality and hospital readmissions in esophageal atresia, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2020.01.025