Prevalence and risk factors of feeding difficulties in children with delayed reconstruction of esophageal atresia: a Swedish nationwide study

Prevalence and risk factors of feeding difficulties in children with delayed reconstruction of esophageal atresia: a Swedish nationwide study | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Research Article Prevalence and risk factors of feeding difficulties in children with delayed reconstruction of esophageal atresia: a Swedish nationwide study Sofie Örnö Ax, Elin Öst, Helene Engstrand Lilja, Erik Omling, and 8 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-6256172/v1 This work is licensed under a CC BY 4.0 License Status: Published Journal Publication published 11 Jun, 2025 Read the published version in Pediatric Surgery International → Version 1 posted 9 You are reading this latest preprint version Abstract Purpose : Children with delayed reconstruction of esophageal atresia (DREA) have a high risk of morbidity. The aim of this study was to determine the prevalence of feeding difficulties in children with DREA, differences to children with primary anastomosis (PA) and identify risk factors for feeding difficulties in children with DREA. Methods : Parents of 30 children with DREA, born between 2001-2018, were recruited nationwide in Sweden and answered an author-developed screening survey about feeding difficulties. Outcomes were statistically compared to that of 105 children with PA, hypothesized as a milder affected group. Relative risk factors for feeding difficulties were investigated using negative binominal regression. Clinical data was retrieved from medical records. Level of significance p<0.05. Results : A majority of children with DREA (83%) had feeding difficulties, most commonly “longer mealtimes than peers”(73%) and “cough/choking during meals”(72%). Food infusion pump(p=0.043), need to avoid specific foods(p=0.049) and need to eat with extra support by an adult(p<0.0001) were more frequent than in PA. The relative risk of feeding difficulties increased with younger child age(p=0.016), number of associated anomalies(p=0.04) number of respiratory(p=0.002) and digestive symptoms(p=0.005). Conclusion : Feeding difficulties in children with DREA are persistent, underlining the need for multidisciplinary and targeted care early in life. Esophageal atresia morbidity feeding surgical outcomes Figures Figure 1 Figure 2 Background Esophageal atresia (EA), is a rare congenital discontinuity of the esophagus affecting 2.43/10 000 newborns( 1 ). In high income countries survival nowadays exceeds 90%( 2 ). A primary anastomosis (PA) of the esophageal ends is reported to be unfeasible in 10% of newborns with EA( 3 ). These children later undergo a delayed reconstruction of EA (DREA). The most common reason for DREA is a long distance between the esophageal ends, referred to as long-gap EA(LGEA)( 3 ). Another reason is frailty of the newborn because of low birth weight and/or prematurity( 4 ). There is no gold standard of operative strategy for DREA. A delayed primary anastomosis(DPA) can be performed in some infants with LGEA after initial insertion of a gastrostomy tube for enteral feeding, and then waiting for some months for growth of the child and the esophageal segments(DPA)( 5 ). Other strategies includes to use elongation traction techniques( 6 , 7 ) or esophageal replacement(ER) with either stomach, jejunum, colon or a gastric tube are techniques used when the gap is too wide to perform a DPA( 8 ). It is well known that the rate of esophageal- and respiratory morbidity is elevated among children with EA( 9 , 10 ). In children with DREA, previous studies indicate an even stronger association with long-term gastrointestinal sequelae such as anastomotic strictures, gastro-esophageal reflux disease and feeding difficulties( 3 , 11 , 12 , 13 , 14 , 15 ). In a recent meta-analysis, the pooled prevalence of studied mealtime adaptations for children with EA encompassed a need to eat slowly(37%), drink fluid to facilitate eating(49%) and to modify textures(28%). Moreover, eating impairments such as difficulty swallowing solids and odynophagia were reported in 45% and 30% respectively( 16 ). Feeding difficulties among children with EA are shown to be associated with chronic respiratory symptoms( 17 ) and may play a role for growth outcomes( 18 ). As eating forms a crucial component of disease-specific health-related quality of life(HRQOL) in children with EA( 19 , 20 , 21 ), there is a distinct association of feeding related morbidity and of caregivers’ anxiety, fear, isolation and sadness( 22 , 23 , 24 , 25 ). Therefore, more knowledge of feeding difficulties in children with DREA is warranted. The first aim of this study was to screen for the prevalence of feeding difficulties in a nationwide recruited cohort of pediatric patients with DREA in Sweden. Secondly, to determine if feeding difficulties among children with DREA differ from children with PA, and the third aim was to identify risk factors of feeding difficulties in children with DREA. Material and Methods This manuscript is part of a nationwide research project in Sweden that explores postoperative morbidity, HRQOL and schooling experiences in children treated with DREA( 26 , 27 , 28 ). Ethical approval was given by the Swedish Ethical Committee in 2019(2019-04,930) and 2020(2020-04,310). Written informed consent for study participation was obtained from parents for all child ages and from children at 15 years of age and older. Assessment of digestive, respiratory and feeding difficulties A standardized survey developed by the authors collected information of the child’s presence of symptoms in the last four weeks and included digestive symptoms and respiratory symptoms. Nine questions regarding the presence of feeding difficulties in children born with EA the past four weeks from the parents’ perspectives were developed from a review of the literature in EA and discussed in a multidisciplinary team. The questions were shown to be feasible as applied in earlier studies published by our group( 29 , 30 ) and enabled comparison of outcomes of children with PA. Additionally, in the nationwide study of children with DREA, six parent-reported questions about swallowing difficulties and food aversion were added (Supplemental material 2 and 3). Children with DREA DREA was defined as when a PA was unattainable during the first surgery, either due to LGEA or frailty because of the infant’s low weight, prematurity and/or other co-morbidities. This meant that all these children’s feeding started with enteral feeding and a(prolonged) delayed establishment of oral feeding. Nationwide recruitment of children with DREA was undertaken through collaboration with all four pediatric surgical centers in Sweden. As detailed in our earlier study( 26 ), children with DREA were considered eligible for recruitment if the child was between 2–18 years of age at the time of the study and had a family that was proficient in written and spoken Swedish. Altogether, 45 children born between 2001 and 2018 who underwent DREA were identified through hospital records at the Karolinska University Hospital, Stockholm(n = 15), the Uppsala University Hospital, Uppsala(n = 14), Sahlgrenska University Hospital, Gothenburg(n = 13) and Skåne University Hospital, Lund(n = 3). Out of the 45 families, one child had died, and three children were lost to follow-up. Two more families were excluded from the study, five declined study participation and four families did not return written informed consent. At the end, 30(67%) families could be included in the study as they gave written informed consent, completed and returned the requested questionnaires. The surgical methods for DREA employed in the study sample were divided into the following; delayed primary anastomosis (DPA)(n = 14), ( 31 ) where esophageal continuity is achieved in a single surgical session much like the one for primary anastomosis(PA). Esophageal replacement(ER) included gastric pull-up(n = 3), ( 32 , 33 ), partial gastric pull-up( n = 3)( 34 ),gastric tube esophagoplasty( n = 8)( 35 ) and colon interposition (n = 2) ( 36 ). In four cases, the reconstruction was delayed due to prematurity/low birth weight only, with a delay of median 137 days(range 34-1221 days). For the 26 patients whose gap length was the reason for DREA, the median time to surgery was 103 days(range 36–207 days). Reference population for comparison One hundred-five children with EA(2–18 years, Gross type C) that underwent PA were included as a reference for comparison from Sahlgrenska University Hospital, Gothenburg. They had participated in earlier studies of generic HRQOL( 37 ), field tests of a condition-specific HRQOL-instrument( 38 , 39 ) and studies about feeding difficulties( 29 , 30 ). Clinical characteristics of children with DREA and PA The clinical characteristics of children with DREA and the reference population has been described for children aged 2–7 and 8–17 years old earlier( 26 ). The median age of the children at the time of the study(DREA, n = 30; median 11 years(range 3–17) vs PA, n = 105; median 8 years, (range 2–17), (p = 0.12), did not differ significantly between the groups. Table 1 presents the sample of children treated with DREA and PA in the total group of children aged 2–18 years. As shown, children with DREA were found to be more commonly born prematurely, with birth weight < 2500 grams and more commonly with anastomotic leakage or sepsis following reconstructive surgery for EA. The prevalence of digestive- and airway symptoms during the last four weeks among children with DREA was reported in a previous study( 26 ). Table 1 Descriptive data of children treated with delayed reconstruction of esophageal atreseia (n = 30) and the reference population of children treated with primary anastomosis (n = 105)( 26 ) n tot Children with delayed reconstruction of esophageal atresia ; n(%) n tot Children with primary anastomosis of esophageal atresia ; n(%) p-value following significance test using Fisher’s exact test Neonatal/Birth characteristics Male sex 30 15(50) 105 63(60) 0.40 Gross subtype 30 A = 12( 40 ) B = 8( 27 ) C = 10( 33 ) 105 C = 105(100) Birth weight in grams, median(range) 30 2133(525–3225) 101 2688(1070–4260) Low birth weight, < 2500 g 30 20(67) 101 39( 39 ) 0.011 Gestational age in weeks, median(range) 30 34( 24 – 40 ) 103 38( 28 – 43 ) Prematurely born 30 18(60) 103 36( 35 ) 0.020 Associated anomalies 30 20(66.7) 105 62(59) 0.53 Cardio-vascular 30 10(33.3) 105 30(28.6) Anorectal 30 7(23.3) 105 10(9.5) Other gastro-intestinal 30 3( 10 ) 104 9(8.7) Uro-genital 30 11(36.7) 105 12(11.4) Limb 30 2(6.7) 105 8(7.6) Vertebrae 30 5(16.7) 105 23(21.9) Choanal atresia 30 1(3.3) 105 1(1.0) Eye 30 2(6.7) 105 6(5.7) Ear 30 1(3.3) 105 3(2.9) Central nervous system 30 2(6.7) 105 10(9.5) Respiratory 30 2(6.7) 105 4(3.8) Other 30 2(6.7) 105 1(1.0) VACTERL* 30 6(20.0) 105 17(16.1) Number of associated anomalies, median (range) 30 1(0–6) 104 1(0–7) Verified genetic deviation 30 5(16.7) 105 10(9.5) Surgical data Major revisional surgery 30 5( 17 ) 105 10(9.5) 0.32 Anastomosis leakage 30 8( 27 ) 103 10(9.7) 0.014 Sepsis verified by blood culture 30 103 14(13.6) 0.028 Days to reconstruction median(range) 30 184(34-1221) Days to discharge median(range) 30 241(62-1235) Fundoplicatio 30 9( 30 ) PPI-treatment 27 19(70.4) *VACTERL-association, vertebral defects, anal atresia, cardiac defects, esophageal atresia, tracheo-esophageal fistula, renal anomalies, and limb abnormalities Data analysis Statistical data were analysed using IBM SPSS Statistics for Windows(version 29.0, Armonk, NY, USA: IBM Corp) and SAS software version 9.4(SAS Institute Inc., Cary, NC, USA). Descriptive statistics were used to present the study population. For categorical variables, frequencies and percentages were used, whereas for continuous variables, median and range were analysed. Where feeding difficulties had been evaluated in both children with DREA and PA, the differences in prevalence were investigated using Fisher’s exact test for binary variables and Pearson Chi Square was used if there were more than two categories. Mann–Whitney U-test were used to analyse differences between two groups, when the dependent variable was continuous. To synchronize with our earlier investigation, we regarded the number of feeding difficulties in a child with DREA as a main outcome of investigation( 29 , 30 ). We used negative binomial regression for number of feeding difficulties to investigate its relative risk in relation to neonatal/birth data(gestational age, birth weight, associated anomalies), surgical complications after reconstructive surgery(anastomotic leakage, major revisional surgery, sepsis verified by blood culture) as well as characteristics of the child at follow up(age, number of respiratory or digestive symptoms respectively). From the negative binomial regression, the relative risk as presented with 95% confidence interval(CI). Due to the low sample size in children with DREA, a multivariable regression analysis and adjustment for possible confounders could not be executed. All tests were two-tailed. Significant level was considered as p < 0.05. Results Prevalence of fifteen feeding difficulties in children with DREA Out of the fifteen investigated aspects of feeding difficulties listed in diagram 1 , 25 children with DREA were reported to have at least one feeding difficulty during the past month. Most commonly, children with DREA reported longer mealtimes than peers and to experience cough or choking during meals. Among the least reported feeding difficulties were the experience of painful mealtimes, texture modified mealtimes and food through infusion pump. Comparison of nine feeding difficulties between children treated with DREA and treated with PA In children with DREA 19/30(63%) had at least one of the nine feeding difficulties that were compared to children with PA. Two families of children that were fed with infusion pump at the time for the study did not fully complete the feeding difficulties questionnaire. Missing data is displayed in Supplemental material, Table 1 . The median number of feeding difficulties in children with DREA was 2(range 0–8) and did not differ compared to children with PA(median 1(0–9, p = 0.25). A significantly higher rate of children with DREA were reported to avoid specific food types that were difficult to eat or swallow compared to children with PA(p = 0.049), as was to have food trough an infusion pump(p = 0.043) and to need assistance by an adult at mealtimes(p < 0.001). diagram 2. Significant results of the comparison of surgical subgroups of DREA and PA are displayed in Table 2 , wheatears complete results are presented in Supplemental material Table 1 . Table 2 Significant results when comparing the frequency of nine feeding difficulties in surgical treatment groups among children with delayed reconstruction of esophageal atresia(n = 30) and primary anastomosis(n = 105)* Frequency in treatment groups n(%) p-value “Eats with extra support by an adult (ex. parent, teacher, assistant” primary anastomosis delayed primary anastomosis 16(15.2) 7(50.0) 0.006 primary anastomosis esophageal replacement 16(15.2) 7(43.8) 0.013 “Eats small portions to facilitate eating ” primary anastomosis delayed primary anastomosis 18(17.1) 4(30.8) 0.031 “Has nutritional intake through gastrostomy” primary anastomosis esophageal replacement 9(8.6) 5(31.3) 0.021 “Has nutritional intake through food infusion pump” primary anastomosis esophageal replacement 5(4.8) 4(25.0) 0.018 *complete results in supplemental material; Table 1 Relative risk for nine feeding difficulties in children with delayed reconstruction of EA Table 3 presents the findings after negative binomial regression of feeding difficulties in children with DREA. A higher number of associated anomalies was associated to increased relative risk for feeding difficulties(p = 0.040), as was lower child age(p = 0.016). Children with DREA(n tot =28) had a median of 2(0–5) respiratory symptoms and 0(range 0–3) digestive symptoms. A higher number of respiratory symptoms(p = 0.002) and digestive symptoms(p = 0.005) significantly increased the relative risk of having feeding difficulties. Table 3 The relationship of neonatal/birth characteristics, complications to reconstructive surgery and respiratory/digestive symptoms at follow up to the number of feeding difficulties* at follow up in children with delayed reconstruction of esophageal atresia when using Negative Binomial Regression. Relative risk (95% CI) P-value NEONATAL/BIRTH CHARACTERISTICS Birth weight Birth weight < 2500g 1.73 (0.64–4.70) 0.28 Birth weight per 500g increase 0.90 (0.65–1.23) 0.50 Gestational age Prematurely born(< 37 gestational weeks at birth) 0.91 (0.34–2.43) 0.86 Gestational Week, per one week increase 0.98 (0.87–1.10) 0.71 Associated anomalies Presence of associated anomaly 1.60 (0.59–4.37) 0.35 Number of associated anomalies, per each additional anomaly 1 1.28 (1.01–1.63) 0.040 RECONSTRUCTIVE SURGERY FOR EA Major revisional surgery after reconstruction of EA 0.59 (0.16–2.17) 0.43 Anastomotic leakage, conservative treatment or revisional surgery 0.97 (0.35–2.70) 0.95 Sepsis verified by blood culture 1.17 (0.42–3.24) 0.76 FOLLOW-UP Child age, per one year increase 0.87 (0.78–0.97) 0.016 Respiratory symptoms, per each additional symptom 2 1.42 (1.14–1.79) 0.002 Digestive symptoms, per each additional symptom 3 1.80 (1.20–2.72) 0.005 *nine feeding difficulties, as stated in diagram 2 1 included are cardio-vascular, anorectal, other gastro-intestinal, uro-genital, skeletal-vertebrae anomalies and those of the central nervous system, ear, eye, respiratory or others 2 cough, wheezing, respiratory infection, chest tightness, dyspnea the past four weeks 3 heartburn, vomiting, swallowing difficulties the past four weeks Discussion To our knowledge this is the first nationwide study of feeding difficulties in children with DREA. We found a high prevalence(83%) of children with feeding difficulties among children with DREA in Sweden. This was despite that these children had taken part in a standardized follow-up care program to monitor health and treatments. Compared to PA, children with DREA reported significantly more often to need assistance at mealtimes, to avoid food that is difficult to eat and to use a food infusion pump. The prevalence of feeding difficulties increased with younger child age, number of associated anomalies and with aerodigestive symptoms. Already in the early nineties Puntis et al. set the tone for how feeding difficulties can be investigated with questionnaires in children with EA and showed that “long mealtimes” was a major issue( 40 ). In this study, “long mealtimes” was considered by two different questions. “Main mealtimes taking longer than 30 minutes” had a prevalence in accordance with current literature( 16 ), whereas having “longer mealtimes than peers” had a surprisingly high prevalence(almost three-quarters) in children with DREA. A possible explanation is that it is easier for parents to recognize that their child finishes late at the table, than quantifying time duration for a mealtime. As having a slower pace when eating has been associated with worse social- and school functioning, negative emotional symptoms( 30 ) and condition-specific HRQOL( 38 ), it’s warrants special attention in clinical practice. The wide range of how prevalent different feeding difficulties are in EA populations suggests a lack of cohesive terminology and common understanding of what condition-specific eating related morbidity looks like for this patient group( 16 ). It complicates matters when studying subgroups in the population, such as DREA. Recently, validated feeding scales like the Montreal Children’s Hospital Feeding Scale have been used( 17 , 41 ), where children with severe EA were found to have more feeding problems. Yet, generic instruments can miss items that forms crucial components of eating related condition-specific HRQOL( 19 , 20 , 21 ) and morbidity( 40 ). Half of the group of children with DREA reported “difficulties eating the same food as peers”, but very few of them needed a modified food texture. A larger proportion of children with DREA than with PA avoided food that was difficult to eat or swallow, which earlier has been defined as a disengagement coping strategy( 42 ) and as a symptom of esophageal dysmotility, stenosis and/or strictures( 43 ). In agreement with these results, children with DREA are at risk of esophageal strictures( 12 , 13 ) and in our study population, the total number of esophageal dilatations at follow-up for children with DREA was significantly higher than in children with P ( 26 ). Esophageal stricture is also a risk factor for ”coughing and choking during mealtimes”( 43 ). Accordingly, coughing and choking during meals was one of the most frequently reported feeding difficulties in this study, and remarkably almost double the estimated pooled prevalence of patients with EA in general( 16 ). Choking, or the risk of choking, is for parents the most anxiety provoking event when feeding a child with DREA( 22 , 23 ). Yet, the need to increase fluid intake to facilitate swallowing was at the same levels as of the PA-group and EA-populations generally( 16 , 29 ). This was unexpected since it is a strategy recommended by caregivers and international support-organizations for EA-families to avoid choking( 44 , 45 ). The greater need for extra adult support at mealtimes in the DREA group compared to PA, may reflect a requirement of supervision at mealtimes for safety concerns or the need for gastrostomy-feeding. This was observed in another study, where the majority of children with LGEA needed support with nutritional intake in school( 27 ). The higher frequency of tube-feeding in the DREA compared to the PA-group could hypothetically explain why more differences between the groups were not found. This was since two families of solely tube-fed children could not answer most feeding difficulty questions. Tube feeding at follow-up was also more common in children with ER vs with PA, which is important to know as it is associated with worse generic HRQOL( 30 ). Swallowing difficulties, heartburn and vomiting as well as respiratory symptoms were associated to increased relative risk of concurrent feeding difficulties, which is not surprising. The relative risk of having feeding difficulties also decreased with higher child age. This all agrees with previous research where respiratory morbidity is found to be associated with feeding difficulties, esophageal dysfunction and dysphagia for children with EA( 9 , 17 ) and that feeding difficulties are more common in young children with EA( 29 , 40 ). Associated anomalies have been regarded as a possible marker of disease severity of EA( 26 ), and in our study more associated anomalies associated with an increased relative risk of feeding difficulties for children with DREA. Yet, the mere presence of associated anomalies was not related to feeding difficulties for children with neither DREA nor EA( 29 ). These results agree with previous research, revealing links between VACTERL association and nutritional intake difficulties in children with EA( 29 ). Interestingly, children with VACTREL displays feeding problems regardless of the presence of EA ( 46 ). As the events that increases the relative risk of feeding difficulties are aligned with previous knowledge, we hypothesise that better knowledge of feeding difficulties in this population can be an accurate tool to determine disease burden. Our results underlines the importance of a multidisciplinary follow up, including a dietician, speech and language therapist and a clinical nurse specialist together with a pediatric surgeon, a gastroenterologist and a pulmonologist. Study limitations and strengths The concept DREA encompasses a group where surgical reconstruction was delayed meanwhile the child was fed enterally( 26 ). Due to retrospective nature of the medical record review, time to oral feeds and how active the oral stimulation was while the child was waiting for reconstruction of the esophagus, could not be identified. Although the term LGEA is more widespread, it has many different definitions( 3 , 4 ). Patients with DREA represent a heterogenous group but enabled us to enlarge the study sample size. Yet, our sample size made it unfeasible to take cofounders or child age into consideration in the statistical analysis and neither could we compare feeding difficulties between the different surgical methods used in the cohort of children with DREA. Yet, with a high inclusion rate and a comparison to children that underwent PA, ( 26 ) our national cohort study is to our knowledge the largest and first of its kind( 16 ). A drawback is the lack of a validated questionnaire about feeding difficulties in the Swedish language. Some of the feeding difficulties that had very high prevalence among children with DREA were unfortunately not included in the comparative analysis of children with PA. Implications for practice and future research Feeding difficulties among children with EA illuminates a cartesian conundrum of congenital disease; patients and their families only report illness when asked condition specific questions, and caregivers are trained to ask about general symptoms of disease( 47 ). In future research, child reported qualitative studies are paramount to properly capture the perception of feeding difficulties. Our study underlines the value of developing a validated questionnaire for children with EA, which encompasses patients’ and their family’s views on what they find troublesome. Furthermore, feeding difficulties should be investigated in relation to anthropometric measures at follow-up as with DREA should receive care at an aerodigestive multidisciplinary clinic. Recognizing its complexity, targeted interventions should be considered to start early in life for children with EA and their families, particularly for children with associated anomalies and persistent aerodigestive morbidity. As an example, pre-operative sham-feed stimulates early oral feeding and may have a positive long-term effect on feeding difficulties( 48 ). Declarations Author Contribution MDB is the principal investigator of the study. SÖA, EÖ, HEL, EO, VG, JFS, AMK, LJ, AT, PS, KA, and MDB contributed to the design and drafting of the study. EÖ, HEL, JFS, AMK, AT, PS, and SÖA were responsible for contacting patients, obtaining informed consent, collecting background data from hospital records, and gathering study materials from patients at their respective surgical centers. SÖA coordinated the national data acquisition process. SÖA and MDB analyzed the data together with a senior statistician. All authors interpreted the data analysis. SÖA drafted the manuscript, which was first critically reviewed for intellectual content by MDB, then by VG, KA, LJ, EÖ, HEL, EO, JFS, AMK, AT, and PS. All authors approved the final manuscript for publication and agree to be accountable for all aspects of the work, ensuring that any questions regarding the accuracy or integrity of the content are thoroughly investigated and resolved. References Pedersen RN, Calzolari E, Husby S, Garne E (2012) Oesophageal atresia: prevalence, prenatal diagnosis and associated anomalies in 23 European regions. Arch Dis Child 97(3):227–232. https://doi.org/10.1136/archdischild-2011-300597 Mortality from gastrointestinal (2021) congenital anomalies at 264 hospitals in 74 low-income, middle-income, and high-income countries: a multicentre, international, prospective cohort study. Lancet 398(10297):325–339. https://doi.org/10.1016/s0140-6736(21)00767-4 Dingemann C, Eaton S, Aksnes G et al (2021) ERNICA Consensus Conference on the Management of Patients with Long-Gap Esophageal Atresia: Perioperative, Surgical, and Long-Term Management. Eur J Pediatr Surg, 31 (3), 214–225. https://doi.org/10.1055/s-0040-1713932 Baird R, Lal DR, Ricca RL et al (2019) Management of long gap esophageal atresia: A systematic review and evidence-based guidelines from the APSA Outcomes and Evidence Based Practice Committee. J Pediatr Surg 54(4):675–687. https://doi.org/10.1016/j.jpedsurg.2018.12.019 Stadil T, Koivusalo A, Pakarinen M et al (2019) Surgical repair of long-gap esophageal atresia: A retrospective study comparing the management of long-gap esophageal atresia in the Nordic countries. J Pediatr Surg 54(3):423–428. https://doi.org/10.1016/j.jpedsurg.2018.07.023 Izadi S, Smithers J, Shieh HF et al (2024) The History and Legacy of the Foker Process for the Treatment of Long Gap Esophageal Atresia. J Pediatr Surg 59(6):1222–1227. https://doi.org/10.1016/j.jpedsurg.2023.12.020 Borselle D, Davidson J, Loukogeorgakis S, De Coppi P, Patkowski D (2024) Thoracoscopic Stage Internal Traction Repair Reduces Time to Achieve Esophageal Continuity in Long Gap Esophageal Atresia. Eur J Pediatr Surg 34(1):36–43. https://doi.org/10.1055/a-2235-8766 Penikis AB, Sescleifer AM, Kunisaki SM (2024) Management of long-gap esophageal atresia. Transl Pediatr 13(2):329–342. https://doi.org/10.21037/tp-23-453 Krishnan U, Mousa H, Dall'Oglio L et al (2016) ESPGHAN-NASPGHAN Guidelines for the Evaluation and Treatment of Gastrointestinal and Nutritional Complications in Children With Esophageal Atresia-Tracheoesophageal Fistula. J Pediatr Gastroenterol Nutr 63(5):550–570. https://doi.org/10.1097/mpg.0000000000001401 Chiang J, Yap T-L, Arif S et al (2023) Gastro-esophageal and respiratory morbidity in children after esophageal atresia repair: a 23-year review from a single tertiary institution in Asia. Annals Pediatr Surg 19(1):19. https://doi.org/10.1186/s43159-023-00251-1 Rayyan M, Embrechts M, Van Veer H et al (2019) Neonatal factors predictive for respiratory and gastro-intestinal morbidity after esophageal atresia repair. Pediatr Neonatol 60(3):261–269. https://doi.org/10.1016/j.pedneo.2018.07.003 Shah R, Varjavandi V, Krishnan U (2015) Predictive factors for complications in children with esophageal atresia and tracheoesophageal fistula. Dis Esophagus 28(3):216–223. https://doi.org/10.1111/dote.12177 Lu YH, Yen TA, Chen CY et al (2021) Risk factors for digestive morbidities after esophageal atresia repair. Eur J Pediatr 180(1):187–194. https://doi.org/10.1007/s00431-020-03733-1 Baird R, Levesque D, Birnbaum R, Ramsay M (2015) A pilot investigation of feeding problems in children with esophageal atresia. Dis Esophagus 28(3):224–228. https://doi.org/10.1111/dote.12178 Bourg A, Gottrand F, Parmentier B et al (2023) Outcome of long gap esophageal atresia at 6 years: A prospective case control cohort study. J Pediatr Surg 58(4):747–755. https://doi.org/10.1016/j.jpedsurg.2022.07.023 Stewart A, Govender R, Eaton S et al (2024) The characteristics of eating, drinking and oro-pharyngeal swallowing difficulties associated with repaired oesophageal atresia/tracheo-oesophageal fistula: a systematic review and meta-proportional analysis. Orphanet J Rare Dis 19(1):253. https://doi.org/10.1186/s13023-024-03259-x Pham A, Ecochard-Dugelay E, Bonnard A et al (2022) Feeding disorders in children with oesophageal atresia: a cross-sectional study. Arch Dis Child 107(1):52–58. https://doi.org/10.1136/archdischild-2020-320609 Traini I, Menzies J, Hughes J, Leach ST, Krishnan U (2020) Oesophageal atresia: The growth gap. World J Gastroenterol 26(12):1262–1272. https://doi.org/10.3748/wjg.v26.i12.1262 The International Ea-, Qol G (2024) An International Collaborative Initiative to Establish a Quality-of-Life Questionnaire for Children and Adolescents with Repair of Esophageal Atresia in 14 Countries. Child (Basel) 11(3). https://doi.org/10.3390/children11030286 Establishment of a (2023) condition-specific quality-of-life questionnaire for children born with esophageal atresia aged 2–7 across 14 countries. Front Pediatr 11:1253892. https://doi.org/10.3389/fped.2023.1253892 Dellenmark-Blom M, Chaplin JE, Gatzinsky V et al (2016) Health-related quality of life experiences among children and adolescents born with esophageal atresia: Development of a condition-specific questionnaire for pediatric patients. J Pediatr Surg 51(4):563–569. https://doi.org/10.1016/j.jpedsurg.2015.09.023 Menzies J, Hughes J (2020) Parental feeding concerns of infants and young children with oesophageal atresia. J Paediatr Child Health 56(11):1791–1794. https://doi.org/10.1111/jpc.14840 Bevilacqua F, Ragni B, Conforti A et al (2020) Fixed the gap, solved the problem? Eating skills in esophageal atresia patients at 3 years. Dis Esophagus 33(1). https://doi.org/10.1093/dote/doz102 Wallace V, Honkalampi K, Korhonen M (2022) Fear, isolation and the importance of support: A qualitative study of parents' experiences of feeding a child born with esophageal atresia. J Pediatr Nurs 67:e9–e15. https://doi.org/10.1016/j.pedn.2022.10.011 Wallace V, Honkalampi K, Sheils E (2021) Anxiety and Depression in Parents of Children Born with Esophageal Atresia: An International Online Survey Study. J Pediatr Nurs 60:77–82. https://doi.org/10.1016/j.pedn.2021.02.016 Dellenmark-Blom M, Örnö Ax S, Öst E et al (2022) Postoperative morbidity and health-related quality of life in children with delayed reconstruction of esophageal atresia: a nationwide Swedish study. Orphanet J Rare Dis 17(1):239. https://doi.org/10.1186/s13023-022-02381-y Dellenmark-Blom M, Reilly C, Öst E et al (2023) Schooling experiences in children with long-gap esophageal atresia compared with children with esophageal atresia and primary anastomosis: a Swedish study. Orphanet J Rare Dis 18(1):233. https://doi.org/10.1186/s13023-023-02846-8 Dellenmark-Blom M, Ax S, Lilja HE et al (2023) Prevalence of Mental Health Problems, Associated Factors, and Health-Related Quality of Life in Children with Long-Gap Esophageal Atresia in Sweden. J Pediatr Surg 58(9):1646–1655. https://doi.org/10.1016/j.jpedsurg.2022.12.004 Ax S, Abrahamsson K, Gatzinsky V, Jönsson L, Dellenmark-Blom M (2021) Parent-Reported Feeding Difficulties among Children Born with Esophageal Atresia: Prevalence and Early Risk Factors. Eur J Pediatr Surg 31(1):69–75. https://doi.org/10.1055/s-0040-1716880 Örnö Ax S, Dellenmark-Blom M, Abrahamsson K, Jönsson L, Gatzinsky V (2023) The association of feeding difficulties and generic health-related quality of life among children born with esophageal atresia. Orphanet J Rare Dis 18(1):237. https://doi.org/10.1186/s13023-023-02836-w Shieh HF, Jennings RW (2017) Long-gap esophageal atresia. Semin Pediatr Surg 26(2):72–77. https://doi.org/https://doi.org/10.1053/j.sempedsurg.2017.02.009 Spitz L, Kiely E, Pierro A (2004) Gastric transposition in children–a 21-year experience. J Pediatr Surg, 39 (3), 276–281; discussion 276–281. https://doi.org/10.1016/j.jpedsurg.2003.11.032 Rubio M, Boglione M, Rührnschopf CG et al (2023) In a Setting of Esophageal Replacement, Total Gastric Pull-Up has Fewer Complications than Partial Gastric Pull-Up. J Pediatr Surg 58(9):1625–1630. https://doi.org/10.1016/j.jpedsurg.2022.11.002 Schärli AF (1996) Esophageal reconstruction by elongation of the lesser gastric curvature. Pediatr Surg Int 11(4):214–217. https://doi.org/10.1007/bf00178419 Zarfati A, Tambucci R, Bagolan P, Conforti A (2023) Isoperistaltic gastric tube for long gap esophageal atresia (LGEA) in newborn, infants, and toddlers: a case-control study from a tertiary center. Front Pediatr 11:1194928. https://doi.org/10.3389/fped.2023.1194928 Bakshi A, Sugarbaker DJ, Burt BM (2017) Alternative conduits for esophageal replacement. Ann Cardiothorac Surg 6(2):137–143. https://doi.org/10.21037/acs.2017.03.07 Flieder S, Dellenmark-Blom M, Witt S et al (2019) Generic Health-Related Quality of Life after Repair of Esophageal Atresia and Its Determinants within a German-Swedish Cohort. Eur J Pediatr Surg 29(1):75–84. https://doi.org/10.1055/s-0038-1672144 Dellenmark-Blom M, Abrahamsson K, Quitmann JH et al (2017) Development and pilot-testing of a condition-specific instrument to assess the quality-of-life in children and adolescents born with esophageal atresia. Dis Esophagus 30(7):1–9. https://doi.org/10.1093/dote/dox017 Dellenmark-Blom M, Dingemann J, Witt S et al (2018) The Esophageal-Atresia-Quality-of-life Questionnaires: Feasibility, Validity and Reliability in Sweden and Germany. J Pediatr Gastroenterol Nutr 67(4):469–477. https://doi.org/10.1097/mpg.0000000000002019 Puntis JW, Ritson DG, Holden CE, Buick RG (1990) Growth and feeding problems after repair of oesophageal atresia. Arch Dis Child 65(1):84–88. https://doi.org/10.1136/adc.65.1.84 Birketvedt K, Mikkelsen A, Hanssen R et al (2024) Feeding difficulties, food intake, and growth in children with esophageal atresia. JPGN Rep 5:462–469. https://doi.org/10.1002/jpr3.12136 Dellenmark-Blom M, Chaplin JE, Quitmann JH et al (2019) The prevalence and role of coping strategies in the nutritional intake of children born with esophageal atresia: a condition-specific approach. Dis Esophagus 32(7). https://doi.org/10.1093/dote/doz004 van Lennep M, Singendonk MMJ, Dall'Oglio L et al (2019) Oesophageal atresia. Nat Rev Dis Primers 5(1):26. https://doi.org/10.1038/s41572-019-0077-0 Federation of Esophageal Atresia and Tracheo-Esophageal Fistula. (n.d.). Long-term esophageal complications. We Are EAT. Retrieved March 18 (2025) from https://www.we-are-eat.org/tell-me-about-esophageal-atresia/long-term-esophageal-complications/ EA/TEF Family Support Connection. (n.d.). A guide for teachers and caregivers. Esophageal Atresia and Tracheoesophageal Fistula Family Support. Retrieved March 18 (2025) from https://www.eatef.org/node/4 Kassa AM, Dahl M, Strinnholm M, Engstrand Lilja H (2020) Attention difficulties and physical dysfunction common in children with complex congenital malformations: a study of preschool children with VACTERL association. Acta Paediatr 109(4):783–789. https://doi.org/10.1111/apa.14566 Hofmann B (2002) On the triad disease, illness and sickness. J Med Philos 27(6):651–673. https://doi.org/10.1076/jmep.27.6.651.13793 Tollne A, Öst E, Nilsson T, Almström M, Svensson JF (2024) Parents caring and sham-feeding their child born with Esophageal atresia at home while waiting for reconstructive surgery. Pediatr Surg Int 40(1):257. https://doi.org/10.1007/s00383-024-05839-1 Additional Declarations No competing interests reported. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-6256172","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":442396271,"identity":"7bf3634d-acd7-425f-8b0a-1d8f19dd18d0","order_by":0,"name":"Sofie Örnö Ax","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA3ElEQVRIiWNgGAWjYFACxgcHEJwKorQwGyBpOUOkFiQb24jQIN/ezHjgA4NNvjl7A+Nn3nm1iQ38hw/g1WJw5jDDwRkMaZY7ew4wS87cdjyxQSItAb8WifwDh3kYDhsY3EhgkPi47RhQC48BXi3yM5IZDv+BaGH+kTgHqIX//Af8nrkB1MIA0cIm8bGhJrGBIQe/DrBfegzSDAzOHGyznHHsgHGbRBoBh7U3M3/4UWFjYHC8+fBtnpo62X7+ww/wWwOxC0QwNgCJwwxsRKhHAXWkahgFo2AUjIIRAABtu0f+ckgBfwAAAABJRU5ErkJggg==","orcid":"","institution":"Queen Silvia Children's Hospital","correspondingAuthor":true,"prefix":"","firstName":"Sofie","middleName":"Örnö","lastName":"Ax","suffix":""},{"id":442396272,"identity":"096ef63e-9d5d-4ac1-8d80-1a74ba72a44f","order_by":1,"name":"Elin Öst","email":"","orcid":"","institution":"Karolinska University Hospital","correspondingAuthor":false,"prefix":"","firstName":"Elin","middleName":"","lastName":"Öst","suffix":""},{"id":442396273,"identity":"498a78f8-5b0d-4c26-92ea-6dae47dd11be","order_by":2,"name":"Helene 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Hospital","correspondingAuthor":false,"prefix":"","firstName":"AnnaMaria","middleName":"","lastName":"Tollne","suffix":""},{"id":442396280,"identity":"403654bb-a1c3-436e-9b09-21b8cc15ed4c","order_by":9,"name":"Pernilla Stenström","email":"","orcid":"","institution":"Lund University, Skane University Hospital","correspondingAuthor":false,"prefix":"","firstName":"Pernilla","middleName":"","lastName":"Stenström","suffix":""},{"id":442396281,"identity":"b10cf465-5253-4ac3-b2df-2d330b0ba2f5","order_by":10,"name":"Kate Abrahamsson","email":"","orcid":"","institution":"Queen Silvia Children's Hospital","correspondingAuthor":false,"prefix":"","firstName":"Kate","middleName":"","lastName":"Abrahamsson","suffix":""},{"id":442396282,"identity":"ef0e3e3c-5aef-47ec-9cc3-ba97c52192ff","order_by":11,"name":"Michaela Dellenmark-Blom","email":"","orcid":"","institution":"Queen Silvia Children's Hospital","correspondingAuthor":false,"prefix":"","firstName":"Michaela","middleName":"","lastName":"Dellenmark-Blom","suffix":""}],"badges":[],"createdAt":"2025-03-18 20:53:12","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-6256172/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-6256172/v1","draftVersion":[],"editorialEvents":[{"content":"https://doi.org/10.1007/s00383-025-06052-4","type":"published","date":"2025-06-11T15:58:00+00:00"}],"editorialNote":"","failedWorkflow":false,"files":[{"id":81018252,"identity":"960ae7db-935b-4cd5-9d6b-8eacc3a534de","added_by":"auto","created_at":"2025-04-21 09:17:24","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":47764,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cem\u003eDiagram 1: \u003c/em\u003eFrequency of feeding difficulties among Swedish children with delayed repair of esophageal atresia (n=30)\u003c/p\u003e","description":"","filename":"pptdiagram2025021111.png","url":"https://assets-eu.researchsquare.com/files/rs-6256172/v1/6e686e64fc4e42b0cb4ecb5d.png"},{"id":81018253,"identity":"f7449a62-5409-4c42-bdb8-47b1577585ab","added_by":"auto","created_at":"2025-04-21 09:17:24","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":42075,"visible":true,"origin":"","legend":"\u003cp\u003e\u003cem\u003eDiagram 2: \u003c/em\u003eFrequency of nine feeding difficulties in children with delayed(n=30) compared to primary(n=105) reconstruction of esophageal atresia\u003c/p\u003e\n\u003cp\u003e\u003cem\u003e* \u003c/em\u003esignificant differences in prevalence between children treated with DREA and the reference population of children treated with PA.\u003c/p\u003e","description":"","filename":"pptdiagram2025021112.png","url":"https://assets-eu.researchsquare.com/files/rs-6256172/v1/0fb06c91579705e2c133c57a.png"},{"id":84726541,"identity":"4b4be326-8b5c-41ed-9c58-27248a809b45","added_by":"auto","created_at":"2025-06-16 16:06:47","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":1134993,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-6256172/v1/d6b18d1b-785b-4cc0-99ab-a2fffe2a269d.pdf"},{"id":81018255,"identity":"b6dd12f5-91e9-4234-9637-f4dfcf028ef3","added_by":"auto","created_at":"2025-04-21 09:17:24","extension":"docx","order_by":0,"title":"","display":"","copyAsset":false,"role":"supplement","size":21414,"visible":true,"origin":"","legend":"","description":"","filename":"Supplementalmaterial20241125.docx","url":"https://assets-eu.researchsquare.com/files/rs-6256172/v1/dceb5b6fe675da667572d940.docx"}],"financialInterests":"No competing interests reported.","formattedTitle":"Prevalence and risk factors of feeding difficulties in children with delayed reconstruction of esophageal atresia: a Swedish nationwide study","fulltext":[{"header":"Background","content":"\u003cp\u003eEsophageal atresia (EA), is a rare congenital discontinuity of the esophagus affecting 2.43/10 000 newborns(\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e). In high income countries survival nowadays exceeds 90%(\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e). A primary anastomosis (PA) of the esophageal ends is reported to be unfeasible in 10% of newborns with EA(\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e). These children later undergo a delayed reconstruction of EA (DREA). The most common reason for DREA is a long distance between the esophageal ends, referred to as long-gap EA(LGEA)(\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e). Another reason is frailty of the newborn because of low birth weight and/or prematurity(\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e). There is no gold standard of operative strategy for DREA. A delayed primary anastomosis(DPA) can be performed in some infants with LGEA after initial insertion of a gastrostomy tube for enteral feeding, and then waiting for some months for growth of the child and the esophageal segments(DPA)(\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e). Other strategies includes to use elongation traction techniques(\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e, \u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e) or esophageal replacement(ER) with either stomach, jejunum, colon or a gastric tube are techniques used when the gap is too wide to perform a DPA(\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eIt is well known that the rate of esophageal- and respiratory morbidity is elevated among children with EA(\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e, \u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e). In children with DREA, previous studies indicate an even stronger association with long-term gastrointestinal sequelae such as anastomotic strictures, gastro-esophageal reflux disease and feeding difficulties(\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e, \u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e, \u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e, \u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e, \u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e, \u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e). In a recent meta-analysis, the pooled prevalence of studied mealtime adaptations for children with EA encompassed a need to eat slowly(37%), drink fluid to facilitate eating(49%) and to modify textures(28%). Moreover, eating impairments such as difficulty swallowing solids and odynophagia were reported in 45% and 30% respectively(\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e). Feeding difficulties among children with EA are shown to be associated with chronic respiratory symptoms(\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e) and may play a role for growth outcomes(\u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e). As eating forms a crucial component of disease-specific health-related quality of life(HRQOL) in children with EA(\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e, \u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e, \u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e), there is a distinct association of feeding related morbidity and of caregivers\u0026rsquo; anxiety, fear, isolation and sadness(\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e, \u003cspan citationid=\"CR23\" class=\"CitationRef\"\u003e23\u003c/span\u003e, \u003cspan citationid=\"CR24\" class=\"CitationRef\"\u003e24\u003c/span\u003e, \u003cspan citationid=\"CR25\" class=\"CitationRef\"\u003e25\u003c/span\u003e). Therefore, more knowledge of feeding difficulties in children with DREA is warranted.\u003c/p\u003e \u003cp\u003eThe first aim of this study was to screen for the prevalence of feeding difficulties in a nationwide recruited cohort of pediatric patients with DREA in Sweden. Secondly, to determine if feeding difficulties among children with DREA differ from children with PA, and the third aim was to identify risk factors of feeding difficulties in children with DREA.\u003c/p\u003e"},{"header":"Material and Methods","content":"\u003cp\u003eThis manuscript is part of a nationwide research project in Sweden that explores postoperative morbidity, HRQOL and schooling experiences in children treated with DREA(\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e, \u003cspan citationid=\"CR27\" class=\"CitationRef\"\u003e27\u003c/span\u003e, \u003cspan citationid=\"CR28\" class=\"CitationRef\"\u003e28\u003c/span\u003e). Ethical approval was given by the Swedish Ethical Committee in 2019(2019-04,930) and 2020(2020-04,310). Written informed consent for study participation was obtained from parents for all child ages and from children at 15 years of age and older.\u003c/p\u003e \u003cdiv id=\"Sec3\" class=\"Section2\"\u003e \u003ch2\u003eAssessment of digestive, respiratory and feeding difficulties\u003c/h2\u003e \u003cp\u003eA standardized survey developed by the authors collected information of the child\u0026rsquo;s presence of symptoms in the last four weeks and included digestive symptoms and respiratory symptoms. Nine questions regarding the presence of feeding difficulties in children born with EA the past four weeks from the parents\u0026rsquo; perspectives were developed from a review of the literature in EA and discussed in a multidisciplinary team. The questions were shown to be feasible as applied in earlier studies published by our group(\u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e, \u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e) and enabled comparison of outcomes of children with PA. Additionally, in the nationwide study of children with DREA, six parent-reported questions about swallowing difficulties and food aversion were added (Supplemental material 2 and 3).\u003c/p\u003e \u003c/div\u003e\n\u003ch3\u003eChildren with DREA\u003c/h3\u003e\n\u003cp\u003eDREA was defined as when a PA was unattainable during the first surgery, either due to LGEA or frailty because of the infant\u0026rsquo;s low weight, prematurity and/or other co-morbidities. This meant that all these children\u0026rsquo;s feeding started with enteral feeding and a(prolonged) delayed establishment of oral feeding. Nationwide recruitment of children with DREA was undertaken through collaboration with all four pediatric surgical centers in Sweden. As detailed in our earlier study(\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e), children with DREA were considered eligible for recruitment if the child was between 2\u0026ndash;18 years of age at the time of the study and had a family that was proficient in written and spoken Swedish. Altogether, 45 children born between 2001 and 2018 who underwent DREA were identified through hospital records at the Karolinska University Hospital, Stockholm(n\u0026thinsp;=\u0026thinsp;15), the Uppsala University Hospital, Uppsala(n\u0026thinsp;=\u0026thinsp;14), Sahlgrenska University Hospital, Gothenburg(n\u0026thinsp;=\u0026thinsp;13) and Sk\u0026aring;ne University Hospital, Lund(n\u0026thinsp;=\u0026thinsp;3). Out of the 45 families, one child had died, and three children were lost to follow-up. Two more families were excluded from the study, five declined study participation and four families did not return written informed consent. At the end, 30(67%) families could be included in the study as they gave written informed consent, completed and returned the requested questionnaires.\u003c/p\u003e \u003cp\u003eThe surgical methods for DREA employed in the study sample were divided into the following; delayed primary anastomosis (DPA)(n\u0026thinsp;=\u0026thinsp;14), (\u003cspan citationid=\"CR31\" class=\"CitationRef\"\u003e31\u003c/span\u003e) where esophageal continuity is achieved in a single surgical session much like the one for primary anastomosis(PA). Esophageal replacement(ER) included gastric pull-up(n\u0026thinsp;=\u0026thinsp;3), (\u003cspan citationid=\"CR32\" class=\"CitationRef\"\u003e32\u003c/span\u003e, \u003cspan citationid=\"CR33\" class=\"CitationRef\"\u003e33\u003c/span\u003e), partial gastric pull-up( n\u0026thinsp;=\u0026thinsp;3)(\u003cspan citationid=\"CR34\" class=\"CitationRef\"\u003e34\u003c/span\u003e),gastric tube esophagoplasty( n\u0026thinsp;=\u0026thinsp;8)(\u003cspan citationid=\"CR35\" class=\"CitationRef\"\u003e35\u003c/span\u003e) and colon interposition (n\u0026thinsp;=\u0026thinsp;2) (\u003cspan citationid=\"CR36\" class=\"CitationRef\"\u003e36\u003c/span\u003e). In four cases, the reconstruction was delayed due to prematurity/low birth weight only, with a delay of median 137 days(range 34-1221 days). For the 26 patients whose gap length was the reason for DREA, the median time to surgery was 103 days(range 36\u0026ndash;207 days).\u003c/p\u003e \u003cp\u003e\u003cb\u003eReference population for comparison\u003c/b\u003eOne hundred-five children with EA(2\u0026ndash;18 years, Gross type C) that underwent PA were included as a reference for comparison from Sahlgrenska University Hospital, Gothenburg. They had participated in earlier studies of generic HRQOL(\u003cspan citationid=\"CR37\" class=\"CitationRef\"\u003e37\u003c/span\u003e), field tests of a condition-specific HRQOL-instrument(\u003cspan citationid=\"CR38\" class=\"CitationRef\"\u003e38\u003c/span\u003e, \u003cspan citationid=\"CR39\" class=\"CitationRef\"\u003e39\u003c/span\u003e) and studies about feeding difficulties(\u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e, \u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e).\u003c/p\u003e\n\u003ch3\u003eClinical characteristics of children with DREA and PA\u003c/h3\u003e\n\u003cp\u003eThe clinical characteristics of children with DREA and the reference population has been described for children aged 2\u0026ndash;7 and 8\u0026ndash;17 years old earlier(\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e). The median age of the children at the time of the study(DREA, n\u0026thinsp;=\u0026thinsp;30; median 11 years(range 3\u0026ndash;17) vs PA, n\u0026thinsp;=\u0026thinsp;105; median 8 years, (range 2\u0026ndash;17), (p\u0026thinsp;=\u0026thinsp;0.12), did not differ significantly between the groups. Table\u0026nbsp;\u003cspan refid=\"Tab1\" class=\"InternalRef\"\u003e1\u003c/span\u003e presents the sample of children treated with DREA and PA in the total group of children aged 2\u0026ndash;18 years. As shown, children with DREA were found to be more commonly born prematurely, with birth weight\u0026thinsp;\u0026lt;\u0026thinsp;2500 grams and more commonly with anastomotic leakage or sepsis following reconstructive surgery for EA. The prevalence of digestive- and airway symptoms during the last four weeks among children with DREA was reported in a previous study(\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab1\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 1\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eDescriptive data of children treated with delayed reconstruction of esophageal atreseia (n\u0026thinsp;=\u0026thinsp;30) and the reference population of children treated with primary anastomosis (n\u0026thinsp;=\u0026thinsp;105)(\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e)\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"6\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"char\" char=\".\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"char\" char=\".\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e \u003cdiv align=\"char\" char=\".\" class=\"colspec\" colname=\"c6\" colnum=\"6\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003en\u003csub\u003etot\u003c/sub\u003e\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eChildren with delayed reconstruction of esophageal atresia ; n(%)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003en\u003csub\u003etot\u003c/sub\u003e\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eChildren with primary anastomosis of esophageal atresia ; n(%)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c6\"\u003e \u003cp\u003ep-value following significance test using Fisher\u0026rsquo;s exact test\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNeonatal/Birth characteristics\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMale sex\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e15(50)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e63(60)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c6\"\u003e \u003cp\u003e0.40\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eGross subtype\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eA\u0026thinsp;=\u0026thinsp;12(\u003cspan citationid=\"CR40\" class=\"CitationRef\"\u003e40\u003c/span\u003e) B\u0026thinsp;=\u0026thinsp;8(\u003cspan citationid=\"CR27\" class=\"CitationRef\"\u003e27\u003c/span\u003e) C\u0026thinsp;=\u0026thinsp;10(\u003cspan citationid=\"CR33\" class=\"CitationRef\"\u003e33\u003c/span\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eC\u0026thinsp;=\u0026thinsp;105(100)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBirth weight in grams, median(range)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2133(525\u0026ndash;3225)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e101\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e2688(1070\u0026ndash;4260)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eLow birth weight, \u0026lt;\u0026thinsp;2500 g\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e20(67)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e101\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e39(\u003cspan citationid=\"CR39\" class=\"CitationRef\"\u003e39\u003c/span\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c6\"\u003e \u003cp\u003e0.011\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eGestational age in weeks, median(range)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e34(\u003cspan additionalcitationids=\"CR25 CR26 CR27 CR28 CR29 CR30 CR31 CR32 CR33 CR34 CR35 CR36 CR37 CR38 CR39\" citationid=\"CR24\" class=\"CitationRef\"\u003e24\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR40\" class=\"CitationRef\"\u003e40\u003c/span\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e103\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e38(\u003cspan additionalcitationids=\"CR29 CR30 CR31 CR32 CR33 CR34 CR35 CR36 CR37 CR38 CR39 CR40 CR41 CR42\" citationid=\"CR28\" class=\"CitationRef\"\u003e28\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR43\" class=\"CitationRef\"\u003e43\u003c/span\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePrematurely born\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e18(60)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e103\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e36(\u003cspan citationid=\"CR35\" class=\"CitationRef\"\u003e35\u003c/span\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c6\"\u003e \u003cp\u003e0.020\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAssociated anomalies\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e20(66.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e62(59)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c6\"\u003e \u003cp\u003e0.53\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCardio-vascular\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e10(33.3)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e30(28.6)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAnorectal\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e7(23.3)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e10(9.5)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOther gastro-intestinal\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e3(\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e104\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e9(8.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eUro-genital\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e11(36.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e12(11.4)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eLimb\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2(6.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e8(7.6)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eVertebrae\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e5(16.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e23(21.9)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eChoanal atresia\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1(3.3)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1(1.0)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eEye\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2(6.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e6(5.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eEar\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1(3.3)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e3(2.9)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCentral nervous system\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2(6.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e10(9.5)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eRespiratory\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2(6.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e4(3.8)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOther\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2(6.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1(1.0)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eVACTERL*\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e6(20.0)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e17(16.1)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNumber of associated anomalies, median (range)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1(0\u0026ndash;6)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e104\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1(0\u0026ndash;7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eVerified genetic deviation\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e5(16.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e10(9.5)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cb\u003eSurgical data\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMajor revisional surgery\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e5(\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e105\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e10(9.5)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c6\"\u003e \u003cp\u003e0.32\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAnastomosis leakage\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e8(\u003cspan citationid=\"CR27\" class=\"CitationRef\"\u003e27\u003c/span\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e103\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e10(9.7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c6\"\u003e \u003cp\u003e0.014\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSepsis verified by blood culture\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c4\"\u003e \u003cp\u003e103\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e14(13.6)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c6\"\u003e \u003cp\u003e0.028\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eDays to reconstruction median(range)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e184(34-1221)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eDays to discharge median(range)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e241(62-1235)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eFundoplicatio\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e9(\u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePPI-treatment\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e27\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e19(70.4)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003ctfoot\u003e \u003ctr\u003e\u003ctd colspan=\"6\"\u003e*VACTERL-association, vertebral defects, anal atresia, cardiac defects, esophageal atresia, tracheo-esophageal fistula, renal anomalies, and limb abnormalities\u003c/td\u003e\u003c/tr\u003e \u003c/tfoot\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cdiv id=\"Sec6\" class=\"Section2\"\u003e \u003ch2\u003eData analysis\u003c/h2\u003e \u003cp\u003eStatistical data were analysed using IBM SPSS Statistics for Windows(version 29.0, Armonk, NY, USA: IBM Corp) and SAS software version 9.4(SAS Institute Inc., Cary, NC, USA). Descriptive statistics were used to present the study population. For categorical variables, frequencies and percentages were used, whereas for continuous variables, median and range were analysed. Where feeding difficulties had been evaluated in both children with DREA and PA, the differences in prevalence were investigated using Fisher\u0026rsquo;s exact test for binary variables and Pearson Chi Square was used if there were more than two categories. Mann\u0026ndash;Whitney U-test were used to analyse differences between two groups, when the dependent variable was continuous. To synchronize with our earlier investigation, we regarded the number of feeding difficulties in a child with DREA as a main outcome of investigation(\u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e, \u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e). We used negative binomial regression for number of feeding difficulties to investigate its relative risk in relation to neonatal/birth data(gestational age, birth weight, associated anomalies), surgical complications after reconstructive surgery(anastomotic leakage, major revisional surgery, sepsis verified by blood culture) as well as characteristics of the child at follow up(age, number of respiratory or digestive symptoms respectively). From the negative binomial regression, the relative risk as presented with 95% confidence interval(CI). Due to the low sample size in children with DREA, a multivariable regression analysis and adjustment for possible confounders could not be executed. All tests were two-tailed. Significant level was considered as p\u0026thinsp;\u0026lt;\u0026thinsp;0.05.\u003c/p\u003e \u003c/div\u003e"},{"header":"Results","content":"\u003cdiv id=\"Sec8\" class=\"Section2\"\u003e\n \u003ch2\u003ePrevalence of fifteen feeding difficulties in children with DREA\u003c/h2\u003e\n \u003cp\u003eOut of the fifteen investigated aspects of feeding difficulties listed in \u003cem\u003ediagram 1\u003c/em\u003e, 25 children with DREA were reported to have at least one feeding difficulty during the past month. Most commonly, children with DREA reported longer mealtimes than peers and to experience cough or choking during meals. Among the least reported feeding difficulties were the experience of painful mealtimes, texture modified mealtimes and food through infusion pump.\u003c/p\u003e\n\u003c/div\u003e\n\u003ch3\u003eComparison of nine feeding difficulties between children treated with DREA and treated with PA\u003c/h3\u003e\n\u003cp\u003eIn children with DREA 19/30(63%) had at least one of the nine feeding difficulties that were compared to children with PA. Two families of children that were fed with infusion pump at the time for the study did not fully complete the feeding difficulties questionnaire. Missing data is displayed in Supplemental material, Table \u003cspan class=\"InternalRef\"\u003e1\u003c/span\u003e. The median number of feeding difficulties in children with DREA was 2(range 0\u0026ndash;8) and did not differ compared to children with PA(median 1(0\u0026ndash;9, p\u0026thinsp;=\u0026thinsp;0.25). A significantly higher rate of children with DREA were reported to avoid specific food types that were difficult to eat or swallow compared to children with PA(p\u0026thinsp;=\u0026thinsp;0.049), as was to have food trough an infusion pump(p\u0026thinsp;=\u0026thinsp;0.043) and to need assistance by an adult at mealtimes(p\u0026thinsp;\u0026lt;\u0026thinsp;0.001). \u003cem\u003ediagram 2.\u003c/em\u003e Significant results of the comparison of surgical subgroups of DREA and PA are displayed in Table \u003cspan class=\"InternalRef\"\u003e2\u003c/span\u003e, wheatears complete results are presented in Supplemental material Table \u003cspan class=\"InternalRef\"\u003e1\u003c/span\u003e.\u0026nbsp;\u003c/p\u003e\n\u003cdiv class=\"gridtable\"\u003e\n \u003ctable id=\"Tab2\" border=\"1\"\u003e\n \u003ccaption\u003e\n \u003cdiv class=\"CaptionNumber\"\u003eTable 2\u003c/div\u003e\n \u003cdiv class=\"CaptionContent\"\u003e\n \u003cp\u003eSignificant results when comparing the frequency of nine feeding difficulties in surgical treatment groups among children with delayed reconstruction of esophageal atresia(n\u0026thinsp;=\u0026thinsp;30) and primary anastomosis(n\u0026thinsp;=\u0026thinsp;105)*\u003c/p\u003e\n \u003c/div\u003e\n \u003c/caption\u003e\n \u003cthead\u003e\n \u003ctr\u003e\n \u003cth colspan=\"2\" align=\"left\"\u003e\n \u003cp\u003eFrequency in treatment groups n(%)\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003ep-value\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003c/thead\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd colspan=\"2\" align=\"left\"\u003e\n \u003cp\u003e\u0026ldquo;Eats with extra support by an adult (ex. parent, teacher, assistant\u0026rdquo;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eprimary anastomosis\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003edelayed primary anastomosis\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e16(15.2)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e7(50.0)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.006\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eprimary anastomosis\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eesophageal replacement\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e16(15.2)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e7(43.8)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.013\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd colspan=\"2\" align=\"left\"\u003e\n \u003cp\u003e\u0026ldquo;Eats small portions to facilitate eating \u0026rdquo;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eprimary anastomosis\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003edelayed primary anastomosis\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e18(17.1)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e4(30.8)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.031\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd colspan=\"2\" align=\"left\"\u003e\n \u003cp\u003e\u0026ldquo;Has nutritional intake through gastrostomy\u0026rdquo;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eprimary anastomosis\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eesophageal replacement\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e9(8.6)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e5(31.3)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.021\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd colspan=\"2\" align=\"left\"\u003e\n \u003cp\u003e\u0026ldquo;Has nutritional intake through food infusion pump\u0026rdquo;\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eprimary anastomosis\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eesophageal replacement\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e5(4.8)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e4(25.0)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.018\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n \u003c/table\u003e\n\u003c/div\u003e\n\u003cp\u003e*complete results in \u003cem\u003esupplemental material;\u003c/em\u003e Table \u003cspan class=\"InternalRef\"\u003e1\u003c/span\u003e\u003c/p\u003e\n\u003ch3\u003eRelative risk for nine feeding difficulties in children with delayed reconstruction of EA\u003c/h3\u003e\n\u003cp\u003eTable\u0026nbsp;\u003cspan class=\"InternalRef\"\u003e3\u003c/span\u003e presents the findings after negative binomial regression of feeding difficulties in children with DREA. A higher number of associated anomalies was associated to increased relative risk for feeding difficulties(p\u0026thinsp;=\u0026thinsp;0.040), as was lower child age(p\u0026thinsp;=\u0026thinsp;0.016). Children with DREA(n\u003csub\u003etot\u003c/sub\u003e=28) had a median of 2(0\u0026ndash;5) respiratory symptoms and 0(range 0\u0026ndash;3) digestive symptoms. A higher number of respiratory symptoms(p\u0026thinsp;=\u0026thinsp;0.002) and digestive symptoms(p\u0026thinsp;=\u0026thinsp;0.005) significantly increased the relative risk of having feeding difficulties.\u0026nbsp;\u003c/p\u003e\n\u003cdiv class=\"gridtable\"\u003e\n \u003ctable id=\"Tab3\" border=\"1\"\u003e\n \u003ccaption\u003e\n \u003cdiv class=\"CaptionNumber\"\u003eTable 3\u003c/div\u003e\n \u003cdiv class=\"CaptionContent\"\u003e\n \u003cp\u003eThe relationship of neonatal/birth characteristics, complications to reconstructive surgery and respiratory/digestive symptoms at follow up to the number of feeding difficulties* at follow up in children with delayed reconstruction of esophageal atresia when using Negative Binomial Regression.\u003c/p\u003e\n \u003c/div\u003e\n \u003c/caption\u003e\n \u003cthead\u003e\n \u003ctr\u003e\n \u003cth align=\"left\"\u003e\u0026nbsp;\u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eRelative risk\u003c/p\u003e\n \u003cp\u003e(95% CI)\u003c/p\u003e\n \u003c/th\u003e\n \u003cth align=\"left\"\u003e\n \u003cp\u003eP-value\u003c/p\u003e\n \u003c/th\u003e\n \u003c/tr\u003e\n \u003c/thead\u003e\n \u003ctbody\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eNEONATAL/BIRTH CHARACTERISTICS\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eBirth weight\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eBirth weight\u0026thinsp;\u0026lt;\u0026thinsp;2500g\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e1.73 (0.64\u0026ndash;4.70)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.28\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eBirth weight per 500g increase\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.90 (0.65\u0026ndash;1.23)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.50\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eGestational age\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003ePrematurely born(\u0026lt;\u0026thinsp;37 gestational weeks at birth)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.91 (0.34\u0026ndash;2.43)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.86\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eGestational Week, per one week increase\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.98 (0.87\u0026ndash;1.10)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.71\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eAssociated anomalies\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003ePresence of associated anomaly\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e1.60 (0.59\u0026ndash;4.37)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.35\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eNumber of associated anomalies, per each additional anomaly\u003csup\u003e1\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e1.28 (1.01\u0026ndash;1.63)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.040\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eRECONSTRUCTIVE SURGERY FOR EA\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eMajor revisional surgery after reconstruction of EA\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.59 (0.16\u0026ndash;2.17)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.43\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eAnastomotic leakage, conservative treatment or revisional surgery\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.97 (0.35\u0026ndash;2.70)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.95\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eSepsis verified by blood culture\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e1.17 (0.42\u0026ndash;3.24)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.76\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eFOLLOW-UP\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003ctd align=\"left\"\u003e\u0026nbsp;\u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eChild age, per one year increase\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.87 (0.78\u0026ndash;0.97)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.016\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eRespiratory symptoms, per each additional symptom\u003csup\u003e2\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e1.42 (1.14\u0026ndash;1.79)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.002\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003eDigestive symptoms, per each additional symptom\u003csup\u003e3\u003c/sup\u003e\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e1.80 (1.20\u0026ndash;2.72)\u003c/p\u003e\n \u003c/td\u003e\n \u003ctd align=\"left\"\u003e\n \u003cp\u003e0.005\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003ctr\u003e\n \u003ctd colspan=\"3\" align=\"left\"\u003e\n \u003cp\u003e*nine feeding difficulties, as stated in diagram 2\u003c/p\u003e\n \u003cp\u003e\u003csup\u003e1\u003c/sup\u003e included are cardio-vascular, anorectal, other gastro-intestinal, uro-genital, skeletal-vertebrae anomalies and those of the central nervous system, ear, eye, respiratory or others\u003c/p\u003e\n \u003cp\u003e\u003csup\u003e2\u003c/sup\u003e cough, wheezing, respiratory infection, chest tightness, dyspnea the past four weeks\u003c/p\u003e\n \u003cp\u003e\u003csup\u003e3\u003c/sup\u003e heartburn, vomiting, swallowing difficulties the past four weeks\u003c/p\u003e\n \u003c/td\u003e\n \u003c/tr\u003e\n \u003c/tbody\u003e\n \u003c/table\u003e\n\u003c/div\u003e"},{"header":"Discussion","content":"\u003cp\u003eTo our knowledge this is the first nationwide study of feeding difficulties in children with DREA. We found a high prevalence(83%) of children with feeding difficulties among children with DREA in Sweden. This was despite that these children had taken part in a standardized follow-up care program to monitor health and treatments. Compared to PA, children with DREA reported significantly more often to need assistance at mealtimes, to avoid food that is difficult to eat and to use a food infusion pump. The prevalence of feeding difficulties increased with younger child age, number of associated anomalies and with aerodigestive symptoms.\u003c/p\u003e \u003cp\u003eAlready in the early nineties Puntis et al. set the tone for how feeding difficulties can be investigated with questionnaires in children with EA and showed that \u0026ldquo;long mealtimes\u0026rdquo; was a major issue(\u003cspan citationid=\"CR40\" class=\"CitationRef\"\u003e40\u003c/span\u003e). In this study, \u0026ldquo;long mealtimes\u0026rdquo; was considered by two different questions. \u0026ldquo;Main mealtimes taking longer than 30 minutes\u0026rdquo; had a prevalence in accordance with current literature(\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e), whereas having \u0026ldquo;longer mealtimes than peers\u0026rdquo; had a surprisingly high prevalence(almost three-quarters) in children with DREA. A possible explanation is that it is easier for parents to recognize that their child finishes late at the table, than quantifying time duration for a mealtime. As having a slower pace when eating has been associated with worse social- and school functioning, negative emotional symptoms(\u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e) and condition-specific HRQOL(\u003cspan citationid=\"CR38\" class=\"CitationRef\"\u003e38\u003c/span\u003e), it\u0026rsquo;s warrants special attention in clinical practice. The wide range of how prevalent different feeding difficulties are in EA populations suggests a lack of cohesive terminology and common understanding of what condition-specific eating related morbidity looks like for this patient group(\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e). It complicates matters when studying subgroups in the population, such as DREA. Recently, validated feeding scales like the Montreal Children\u0026rsquo;s Hospital Feeding Scale have been used(\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e, \u003cspan citationid=\"CR41\" class=\"CitationRef\"\u003e41\u003c/span\u003e), where children with severe EA were found to have more feeding problems. Yet, generic instruments can miss items that forms crucial components of eating related condition-specific HRQOL(\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e, \u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e, \u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e) and morbidity(\u003cspan citationid=\"CR40\" class=\"CitationRef\"\u003e40\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eHalf of the group of children with DREA reported \u0026ldquo;difficulties eating the same food as peers\u0026rdquo;, but very few of them needed a modified food texture. A larger proportion of children with DREA than with PA avoided food that was difficult to eat or swallow, which earlier has been defined as a disengagement coping strategy(\u003cspan citationid=\"CR42\" class=\"CitationRef\"\u003e42\u003c/span\u003e) and as a symptom of esophageal dysmotility, stenosis and/or strictures(\u003cspan citationid=\"CR43\" class=\"CitationRef\"\u003e43\u003c/span\u003e). In agreement with these results, children with DREA are at risk of esophageal strictures(\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e, \u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e) and in our study population, the total number of esophageal dilatations at follow-up for children with DREA was significantly higher than in children with P (\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e). Esophageal stricture is also a risk factor for \u0026rdquo;coughing and choking during mealtimes\u0026rdquo;(\u003cspan citationid=\"CR43\" class=\"CitationRef\"\u003e43\u003c/span\u003e). Accordingly, coughing and choking during meals was one of the most frequently reported feeding difficulties in this study, and remarkably almost double the estimated pooled prevalence of patients with EA in general(\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e). Choking, or the risk of choking, is for parents the most anxiety provoking event when feeding a child with DREA(\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e, \u003cspan citationid=\"CR23\" class=\"CitationRef\"\u003e23\u003c/span\u003e). Yet, the need to increase fluid intake to facilitate swallowing was at the same levels as of the PA-group and EA-populations generally(\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e, \u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e). This was unexpected since it is a strategy recommended by caregivers and international support-organizations for EA-families to avoid choking(\u003cspan citationid=\"CR44\" class=\"CitationRef\"\u003e44\u003c/span\u003e, \u003cspan citationid=\"CR45\" class=\"CitationRef\"\u003e45\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eThe greater need for extra adult support at mealtimes in the DREA group compared to PA, may reflect a requirement of supervision at mealtimes for safety concerns or the need for gastrostomy-feeding. This was observed in another study, where the majority of children with LGEA needed support with nutritional intake in school(\u003cspan citationid=\"CR27\" class=\"CitationRef\"\u003e27\u003c/span\u003e). The higher frequency of tube-feeding in the DREA compared to the PA-group could hypothetically explain why more differences between the groups were not found. This was since two families of solely tube-fed children could not answer most feeding difficulty questions. Tube feeding at follow-up was also more common in children with ER vs with PA, which is important to know as it is associated with worse generic HRQOL(\u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eSwallowing difficulties, heartburn and vomiting as well as respiratory symptoms were associated to increased relative risk of concurrent feeding difficulties, which is not surprising. The relative risk of having feeding difficulties also decreased with higher child age. This all agrees with previous research where respiratory morbidity is found to be associated with feeding difficulties, esophageal dysfunction and dysphagia for children with EA(\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e, \u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e) and that feeding difficulties are more common in young children with EA(\u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e, \u003cspan citationid=\"CR40\" class=\"CitationRef\"\u003e40\u003c/span\u003e). Associated anomalies have been regarded as a possible marker of disease severity of EA(\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e), and in our study more associated anomalies associated with an increased relative risk of feeding difficulties for children with DREA. Yet, the mere presence of associated anomalies was not related to feeding difficulties for children with neither DREA nor EA(\u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e). These results agree with previous research, revealing links between VACTERL association and nutritional intake difficulties in children with EA(\u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e). Interestingly, children with VACTREL displays feeding problems regardless of the presence of EA (\u003cspan citationid=\"CR46\" class=\"CitationRef\"\u003e46\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e As the events that increases the relative risk of feeding difficulties are aligned with previous knowledge, we hypothesise that better knowledge of feeding difficulties in this population can be an accurate tool to determine disease burden. Our results underlines the importance of a multidisciplinary follow up, including a dietician, speech and language therapist and a clinical nurse specialist together with a pediatric surgeon, a gastroenterologist and a pulmonologist.\u003c/p\u003e \u003cdiv id=\"Sec12\" class=\"Section2\"\u003e \u003ch2\u003eStudy limitations and strengths\u003c/h2\u003e \u003cp\u003eThe concept DREA encompasses a group where surgical reconstruction was delayed meanwhile the child was fed enterally(\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e). Due to retrospective nature of the medical record review, time to oral feeds and how active the oral stimulation was while the child was waiting for reconstruction of the esophagus, could not be identified. Although the term LGEA is more widespread, it has many different definitions(\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e, \u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e). Patients with DREA represent a heterogenous group but enabled us to enlarge the study sample size. Yet, our sample size made it unfeasible to take cofounders or child age into consideration in the statistical analysis and neither could we compare feeding difficulties between the different surgical methods used in the cohort of children with DREA. Yet, with a high inclusion rate and a comparison to children that underwent PA, (\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e) our national cohort study is to our knowledge the largest and first of its kind(\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e). A drawback is the lack of a validated questionnaire about feeding difficulties in the Swedish language. Some of the feeding difficulties that had very high prevalence among children with DREA were unfortunately not included in the comparative analysis of children with PA.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec13\" class=\"Section2\"\u003e \u003ch2\u003eImplications for practice and future research\u003c/h2\u003e \u003cp\u003eFeeding difficulties among children with EA illuminates a cartesian conundrum of congenital disease; patients and their families only report illness when asked condition specific questions, and caregivers are trained to ask about general symptoms of disease(\u003cspan citationid=\"CR47\" class=\"CitationRef\"\u003e47\u003c/span\u003e). In future research, child reported qualitative studies are paramount to properly capture the perception of feeding difficulties. Our study underlines the value of developing a validated questionnaire for children with EA, which encompasses patients\u0026rsquo; and their family\u0026rsquo;s views on what they find troublesome. Furthermore, feeding difficulties should be investigated in relation to anthropometric measures at follow-up as with DREA should receive care at an aerodigestive multidisciplinary clinic. Recognizing its complexity, targeted interventions should be considered to start early in life for children with EA and their families, particularly for children with associated anomalies and persistent aerodigestive morbidity. As an example, pre-operative sham-feed stimulates early oral feeding and may have a positive long-term effect on feeding difficulties(\u003cspan citationid=\"CR48\" class=\"CitationRef\"\u003e48\u003c/span\u003e).\u003c/p\u003e \u003c/div\u003e"},{"header":"Declarations","content":"\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\u003cp\u003eMDB is the principal investigator of the study. S\u0026Ouml;A, E\u0026Ouml;, HEL, EO, VG, JFS, AMK, LJ, AT, PS, KA, and MDB contributed to the design and drafting of the study. E\u0026Ouml;, HEL, JFS, AMK, AT, PS, and S\u0026Ouml;A were responsible for contacting patients, obtaining informed consent, collecting background data from hospital records, and gathering study materials from patients at their respective surgical centers. S\u0026Ouml;A coordinated the national data acquisition process. S\u0026Ouml;A and MDB analyzed the data together with a senior statistician. All authors interpreted the data analysis. S\u0026Ouml;A drafted the manuscript, which was first critically reviewed for intellectual content by MDB, then by VG, KA, LJ, E\u0026Ouml;, HEL, EO, JFS, AMK, AT, and PS. All authors approved the final manuscript for publication and agree to be accountable for all aspects of the work, ensuring that any questions regarding the accuracy or integrity of the content are thoroughly investigated and resolved.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003ePedersen RN, Calzolari E, Husby S, Garne E (2012) Oesophageal atresia: prevalence, prenatal diagnosis and associated anomalies in 23 European regions. Arch Dis Child 97(3):227\u0026ndash;232. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1136/archdischild-2011-300597\u003c/span\u003e\u003cspan address=\"10.1136/archdischild-2011-300597\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMortality from gastrointestinal (2021) congenital anomalies at 264 hospitals in 74 low-income, middle-income, and high-income countries: a multicentre, international, prospective cohort study. Lancet 398(10297):325\u0026ndash;339. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/s0140-6736(21)00767-4\u003c/span\u003e\u003cspan address=\"10.1016/s0140-6736(21)00767-4\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDingemann C, Eaton S, Aksnes G et al (2021) ERNICA Consensus Conference on the Management of Patients with Long-Gap Esophageal Atresia: Perioperative, Surgical, and Long-Term Management. \u003cem\u003eEur J Pediatr Surg, 31\u003c/em\u003e(3), 214\u0026ndash;225. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1055/s-0040-1713932\u003c/span\u003e\u003cspan address=\"10.1055/s-0040-1713932\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBaird R, Lal DR, Ricca RL et al (2019) Management of long gap esophageal atresia: A systematic review and evidence-based guidelines from the APSA Outcomes and Evidence Based Practice Committee. J Pediatr Surg 54(4):675\u0026ndash;687. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.jpedsurg.2018.12.019\u003c/span\u003e\u003cspan address=\"10.1016/j.jpedsurg.2018.12.019\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eStadil T, Koivusalo A, Pakarinen M et al (2019) Surgical repair of long-gap esophageal atresia: A retrospective study comparing the management of long-gap esophageal atresia in the Nordic countries. J Pediatr Surg 54(3):423\u0026ndash;428. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.jpedsurg.2018.07.023\u003c/span\u003e\u003cspan address=\"10.1016/j.jpedsurg.2018.07.023\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eIzadi S, Smithers J, Shieh HF et al (2024) The History and Legacy of the Foker Process for the Treatment of Long Gap Esophageal Atresia. J Pediatr Surg 59(6):1222\u0026ndash;1227. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.jpedsurg.2023.12.020\u003c/span\u003e\u003cspan address=\"10.1016/j.jpedsurg.2023.12.020\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBorselle D, Davidson J, Loukogeorgakis S, De Coppi P, Patkowski D (2024) Thoracoscopic Stage Internal Traction Repair Reduces Time to Achieve Esophageal Continuity in Long Gap Esophageal Atresia. Eur J Pediatr Surg 34(1):36\u0026ndash;43. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1055/a-2235-8766\u003c/span\u003e\u003cspan address=\"10.1055/a-2235-8766\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003ePenikis AB, Sescleifer AM, Kunisaki SM (2024) Management of long-gap esophageal atresia. Transl Pediatr 13(2):329\u0026ndash;342. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.21037/tp-23-453\u003c/span\u003e\u003cspan address=\"10.21037/tp-23-453\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eKrishnan U, Mousa H, Dall'Oglio L et al (2016) ESPGHAN-NASPGHAN Guidelines for the Evaluation and Treatment of Gastrointestinal and Nutritional Complications in Children With Esophageal Atresia-Tracheoesophageal Fistula. J Pediatr Gastroenterol Nutr 63(5):550\u0026ndash;570. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1097/mpg.0000000000001401\u003c/span\u003e\u003cspan address=\"10.1097/mpg.0000000000001401\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eChiang J, Yap T-L, Arif S et al (2023) Gastro-esophageal and respiratory morbidity in children after esophageal atresia repair: a 23-year review from a single tertiary institution in Asia. Annals Pediatr Surg 19(1):19. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1186/s43159-023-00251-1\u003c/span\u003e\u003cspan address=\"10.1186/s43159-023-00251-1\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eRayyan M, Embrechts M, Van Veer H et al (2019) Neonatal factors predictive for respiratory and gastro-intestinal morbidity after esophageal atresia repair. Pediatr Neonatol 60(3):261\u0026ndash;269. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.pedneo.2018.07.003\u003c/span\u003e\u003cspan address=\"10.1016/j.pedneo.2018.07.003\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eShah R, Varjavandi V, Krishnan U (2015) Predictive factors for complications in children with esophageal atresia and tracheoesophageal fistula. Dis Esophagus 28(3):216\u0026ndash;223. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1111/dote.12177\u003c/span\u003e\u003cspan address=\"10.1111/dote.12177\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLu YH, Yen TA, Chen CY et al (2021) Risk factors for digestive morbidities after esophageal atresia repair. Eur J Pediatr 180(1):187\u0026ndash;194. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1007/s00431-020-03733-1\u003c/span\u003e\u003cspan address=\"10.1007/s00431-020-03733-1\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBaird R, Levesque D, Birnbaum R, Ramsay M (2015) A pilot investigation of feeding problems in children with esophageal atresia. Dis Esophagus 28(3):224\u0026ndash;228. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1111/dote.12178\u003c/span\u003e\u003cspan address=\"10.1111/dote.12178\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBourg A, Gottrand F, Parmentier B et al (2023) Outcome of long gap esophageal atresia at 6 years: A prospective case control cohort study. J Pediatr Surg 58(4):747\u0026ndash;755. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.jpedsurg.2022.07.023\u003c/span\u003e\u003cspan address=\"10.1016/j.jpedsurg.2022.07.023\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eStewart A, Govender R, Eaton S et al (2024) The characteristics of eating, drinking and oro-pharyngeal swallowing difficulties associated with repaired oesophageal atresia/tracheo-oesophageal fistula: a systematic review and meta-proportional analysis. Orphanet J Rare Dis 19(1):253. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1186/s13023-024-03259-x\u003c/span\u003e\u003cspan address=\"10.1186/s13023-024-03259-x\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003ePham A, Ecochard-Dugelay E, Bonnard A et al (2022) Feeding disorders in children with oesophageal atresia: a cross-sectional study. Arch Dis Child 107(1):52\u0026ndash;58. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1136/archdischild-2020-320609\u003c/span\u003e\u003cspan address=\"10.1136/archdischild-2020-320609\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eTraini I, Menzies J, Hughes J, Leach ST, Krishnan U (2020) Oesophageal atresia: The growth gap. World J Gastroenterol 26(12):1262\u0026ndash;1272. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.3748/wjg.v26.i12.1262\u003c/span\u003e\u003cspan address=\"10.3748/wjg.v26.i12.1262\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eThe International Ea-, Qol G (2024) An International Collaborative Initiative to Establish a Quality-of-Life Questionnaire for Children and Adolescents with Repair of Esophageal Atresia in 14 Countries. Child (Basel) 11(3). \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.3390/children11030286\u003c/span\u003e\u003cspan address=\"10.3390/children11030286\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eEstablishment of a (2023) condition-specific quality-of-life questionnaire for children born with esophageal atresia aged 2\u0026ndash;7 across 14 countries. Front Pediatr 11:1253892. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.3389/fped.2023.1253892\u003c/span\u003e\u003cspan address=\"10.3389/fped.2023.1253892\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDellenmark-Blom M, Chaplin JE, Gatzinsky V et al (2016) Health-related quality of life experiences among children and adolescents born with esophageal atresia: Development of a condition-specific questionnaire for pediatric patients. J Pediatr Surg 51(4):563\u0026ndash;569. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.jpedsurg.2015.09.023\u003c/span\u003e\u003cspan address=\"10.1016/j.jpedsurg.2015.09.023\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMenzies J, Hughes J (2020) Parental feeding concerns of infants and young children with oesophageal atresia. J Paediatr Child Health 56(11):1791\u0026ndash;1794. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1111/jpc.14840\u003c/span\u003e\u003cspan address=\"10.1111/jpc.14840\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBevilacqua F, Ragni B, Conforti A et al (2020) Fixed the gap, solved the problem? Eating skills in esophageal atresia patients at 3 years. Dis Esophagus 33(1). \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1093/dote/doz102\u003c/span\u003e\u003cspan address=\"10.1093/dote/doz102\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eWallace V, Honkalampi K, Korhonen M (2022) Fear, isolation and the importance of support: A qualitative study of parents' experiences of feeding a child born with esophageal atresia. J Pediatr Nurs 67:e9\u0026ndash;e15. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.pedn.2022.10.011\u003c/span\u003e\u003cspan address=\"10.1016/j.pedn.2022.10.011\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eWallace V, Honkalampi K, Sheils E (2021) Anxiety and Depression in Parents of Children Born with Esophageal Atresia: An International Online Survey Study. J Pediatr Nurs 60:77\u0026ndash;82. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.pedn.2021.02.016\u003c/span\u003e\u003cspan address=\"10.1016/j.pedn.2021.02.016\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDellenmark-Blom M, \u0026Ouml;rn\u0026ouml; Ax S, \u0026Ouml;st E et al (2022) Postoperative morbidity and health-related quality of life in children with delayed reconstruction of esophageal atresia: a nationwide Swedish study. Orphanet J Rare Dis 17(1):239. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1186/s13023-022-02381-y\u003c/span\u003e\u003cspan address=\"10.1186/s13023-022-02381-y\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDellenmark-Blom M, Reilly C, \u0026Ouml;st E et al (2023) Schooling experiences in children with long-gap esophageal atresia compared with children with esophageal atresia and primary anastomosis: a Swedish study. Orphanet J Rare Dis 18(1):233. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1186/s13023-023-02846-8\u003c/span\u003e\u003cspan address=\"10.1186/s13023-023-02846-8\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDellenmark-Blom M, Ax S, Lilja HE et al (2023) Prevalence of Mental Health Problems, Associated Factors, and Health-Related Quality of Life in Children with Long-Gap Esophageal Atresia in Sweden. J Pediatr Surg 58(9):1646\u0026ndash;1655. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.jpedsurg.2022.12.004\u003c/span\u003e\u003cspan address=\"10.1016/j.jpedsurg.2022.12.004\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eAx S, Abrahamsson K, Gatzinsky V, J\u0026ouml;nsson L, Dellenmark-Blom M (2021) Parent-Reported Feeding Difficulties among Children Born with Esophageal Atresia: Prevalence and Early Risk Factors. Eur J Pediatr Surg 31(1):69\u0026ndash;75. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1055/s-0040-1716880\u003c/span\u003e\u003cspan address=\"10.1055/s-0040-1716880\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003e\u0026Ouml;rn\u0026ouml; Ax S, Dellenmark-Blom M, Abrahamsson K, J\u0026ouml;nsson L, Gatzinsky V (2023) The association of feeding difficulties and generic health-related quality of life among children born with esophageal atresia. Orphanet J Rare Dis 18(1):237. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1186/s13023-023-02836-w\u003c/span\u003e\u003cspan address=\"10.1186/s13023-023-02836-w\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eShieh HF, Jennings RW (2017) Long-gap esophageal atresia. Semin Pediatr Surg 26(2):72\u0026ndash;77. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/https://doi.org/10.1053/j.sempedsurg.2017.02.009\u003c/span\u003e\u003cspan address=\"10.1053/j.sempedsurg.2017.02.009\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSpitz L, Kiely E, Pierro A (2004) Gastric transposition in children\u0026ndash;a 21-year experience. \u003cem\u003eJ Pediatr Surg, 39\u003c/em\u003e(3), 276\u0026ndash;281; discussion 276\u0026ndash;281. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.jpedsurg.2003.11.032\u003c/span\u003e\u003cspan address=\"10.1016/j.jpedsurg.2003.11.032\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eRubio M, Boglione M, R\u0026uuml;hrnschopf CG et al (2023) In a Setting of Esophageal Replacement, Total Gastric Pull-Up has Fewer Complications than Partial Gastric Pull-Up. J Pediatr Surg 58(9):1625\u0026ndash;1630. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.jpedsurg.2022.11.002\u003c/span\u003e\u003cspan address=\"10.1016/j.jpedsurg.2022.11.002\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSch\u0026auml;rli AF (1996) Esophageal reconstruction by elongation of the lesser gastric curvature. Pediatr Surg Int 11(4):214\u0026ndash;217. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1007/bf00178419\u003c/span\u003e\u003cspan address=\"10.1007/bf00178419\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eZarfati A, Tambucci R, Bagolan P, Conforti A (2023) Isoperistaltic gastric tube for long gap esophageal atresia (LGEA) in newborn, infants, and toddlers: a case-control study from a tertiary center. Front Pediatr 11:1194928. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.3389/fped.2023.1194928\u003c/span\u003e\u003cspan address=\"10.3389/fped.2023.1194928\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBakshi A, Sugarbaker DJ, Burt BM (2017) Alternative conduits for esophageal replacement. Ann Cardiothorac Surg 6(2):137\u0026ndash;143. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.21037/acs.2017.03.07\u003c/span\u003e\u003cspan address=\"10.21037/acs.2017.03.07\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eFlieder S, Dellenmark-Blom M, Witt S et al (2019) Generic Health-Related Quality of Life after Repair of Esophageal Atresia and Its Determinants within a German-Swedish Cohort. Eur J Pediatr Surg 29(1):75\u0026ndash;84. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1055/s-0038-1672144\u003c/span\u003e\u003cspan address=\"10.1055/s-0038-1672144\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDellenmark-Blom M, Abrahamsson K, Quitmann JH et al (2017) Development and pilot-testing of a condition-specific instrument to assess the quality-of-life in children and adolescents born with esophageal atresia. Dis Esophagus 30(7):1\u0026ndash;9. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1093/dote/dox017\u003c/span\u003e\u003cspan address=\"10.1093/dote/dox017\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDellenmark-Blom M, Dingemann J, Witt S et al (2018) The Esophageal-Atresia-Quality-of-life Questionnaires: Feasibility, Validity and Reliability in Sweden and Germany. J Pediatr Gastroenterol Nutr 67(4):469\u0026ndash;477. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1097/mpg.0000000000002019\u003c/span\u003e\u003cspan address=\"10.1097/mpg.0000000000002019\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003ePuntis JW, Ritson DG, Holden CE, Buick RG (1990) Growth and feeding problems after repair of oesophageal atresia. Arch Dis Child 65(1):84\u0026ndash;88. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1136/adc.65.1.84\u003c/span\u003e\u003cspan address=\"10.1136/adc.65.1.84\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBirketvedt K, Mikkelsen A, Hanssen R et al (2024) Feeding difficulties, food intake, and growth in children with esophageal atresia. JPGN Rep 5:462\u0026ndash;469. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1002/jpr3.12136\u003c/span\u003e\u003cspan address=\"10.1002/jpr3.12136\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDellenmark-Blom M, Chaplin JE, Quitmann JH et al (2019) The prevalence and role of coping strategies in the nutritional intake of children born with esophageal atresia: a condition-specific approach. Dis Esophagus 32(7). \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1093/dote/doz004\u003c/span\u003e\u003cspan address=\"10.1093/dote/doz004\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003evan Lennep M, Singendonk MMJ, Dall'Oglio L et al (2019) Oesophageal atresia. Nat Rev Dis Primers 5(1):26. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1038/s41572-019-0077-0\u003c/span\u003e\u003cspan address=\"10.1038/s41572-019-0077-0\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eFederation of Esophageal Atresia and Tracheo-Esophageal Fistula. (n.d.). Long-term esophageal complications. We Are EAT. Retrieved March 18 (2025) from \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://www.we-are-eat.org/tell-me-about-esophageal-atresia/long-term-esophageal-complications/\u003c/span\u003e\u003cspan address=\"https://www.we-are-eat.org/tell-me-about-esophageal-atresia/long-term-esophageal-complications/\" targettype=\"URL\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eEA/TEF Family Support Connection. (n.d.). A guide for teachers and caregivers. Esophageal Atresia and Tracheoesophageal Fistula Family Support. Retrieved March 18 (2025) from \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://www.eatef.org/node/4\u003c/span\u003e\u003cspan address=\"https://www.eatef.org/node/4\" targettype=\"URL\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eKassa AM, Dahl M, Strinnholm M, Engstrand Lilja H (2020) Attention difficulties and physical dysfunction common in children with complex congenital malformations: a study of preschool children with VACTERL association. Acta Paediatr 109(4):783\u0026ndash;789. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1111/apa.14566\u003c/span\u003e\u003cspan address=\"10.1111/apa.14566\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eHofmann B (2002) On the triad disease, illness and sickness. J Med Philos 27(6):651\u0026ndash;673. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1076/jmep.27.6.651.13793\u003c/span\u003e\u003cspan address=\"10.1076/jmep.27.6.651.13793\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eTollne A, \u0026Ouml;st E, Nilsson T, Almstr\u0026ouml;m M, Svensson JF (2024) Parents caring and sham-feeding their child born with Esophageal atresia at home while waiting for reconstructive surgery. Pediatr Surg Int 40(1):257. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1007/s00383-024-05839-1\u003c/span\u003e\u003cspan address=\"10.1007/s00383-024-05839-1\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"pediatric-surgery-international","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"pesi","sideBox":"Learn more about [Pediatric Surgery International](http://link.springer.com/journal/383)","snPcode":"383","submissionUrl":"https://submission.nature.com/new-submission/383/3","title":"Pediatric Surgery International","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false},"keywords":"Esophageal atresia, morbidity, feeding, surgical outcomes","lastPublishedDoi":"10.21203/rs.3.rs-6256172/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-6256172/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003ePurpose\u003c/strong\u003e: Children with delayed reconstruction of esophageal atresia (DREA) have a high risk of morbidity. The aim of this study was to determine the prevalence of feeding difficulties in children with DREA, differences to children with primary anastomosis (PA) and identify risk factors for feeding difficulties in children with DREA.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eMethods\u003c/strong\u003e: Parents of 30 children with DREA, born between 2001-2018, were recruited nationwide in Sweden and answered an author-developed screening survey about feeding difficulties. Outcomes were statistically compared to that of 105 children with PA, hypothesized as a milder affected group. Relative risk factors for feeding difficulties were investigated using negative binominal regression. Clinical data was retrieved from medical records. Level of significance p\u0026lt;0.05.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eResults\u003c/strong\u003e: A majority of children with DREA (83%) had feeding difficulties, most commonly “longer mealtimes than peers”(73%) and “cough/choking during meals”(72%). Food infusion pump(p=0.043), need to avoid specific foods(p=0.049) and need to eat with extra support by an adult(p\u0026lt;0.0001) were more frequent than in PA. The relative risk of feeding difficulties increased with younger child age(p=0.016), number of associated anomalies(p=0.04) number of respiratory(p=0.002) and digestive symptoms(p=0.005).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConclusion\u003c/strong\u003e: Feeding difficulties in children with DREA are persistent, underlining the need for multidisciplinary and targeted care early in life.\u003c/p\u003e","manuscriptTitle":"Prevalence and risk factors of feeding difficulties in children with delayed reconstruction of esophageal atresia: a Swedish nationwide study","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-04-21 09:17:19","doi":"10.21203/rs.3.rs-6256172/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2025-04-14T00:08:17+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-04-03T19:56:34+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-04-02T10:30:17+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"35752050907039197323901345389420099003","date":"2025-03-31T23:40:30+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"324760950378806630619102109221492524710","date":"2025-03-30T16:04:41+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2025-03-29T23:17:01+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2025-03-20T14:13:59+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2025-03-20T06:59:41+00:00","index":"","fulltext":""},{"type":"submitted","content":"Pediatric Surgery International","date":"2025-03-18T20:49:39+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"pediatric-surgery-international","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"pesi","sideBox":"Learn more about [Pediatric Surgery International](http://link.springer.com/journal/383)","snPcode":"383","submissionUrl":"https://submission.nature.com/new-submission/383/3","title":"Pediatric Surgery International","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false}}],"origin":"","ownerIdentity":"febcd0e1-cf7e-4287-b3a9-4194fc116bbb","owner":[],"postedDate":"April 21st, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"published-in-journal","subjectAreas":[],"tags":[],"updatedAt":"2025-06-16T16:02:20+00:00","versionOfRecord":{"articleIdentity":"rs-6256172","link":"https://doi.org/10.1007/s00383-025-06052-4","journal":{"identity":"pediatric-surgery-international","isVorOnly":false,"title":"Pediatric Surgery International"},"publishedOn":"2025-06-11 15:58:00","publishedOnDateReadable":"June 11th, 2025"},"versionCreatedAt":"2025-04-21 09:17:19","video":"","vorDoi":"10.1007/s00383-025-06052-4","vorDoiUrl":"https://doi.org/10.1007/s00383-025-06052-4","workflowStages":[]},"version":"v1","identity":"rs-6256172","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-6256172","identity":"rs-6256172","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}