European Journal of Cardiovascular Medicine

Tracheoesophageal fistula (TEF) is the most common type of airway fistula. It is caused by an abnormal link between the oesophagus and the trachea. Symptoms of tracheoesophageal fistula (TEF) include trouble eating, choking, frothing after eating, severe coughing, and lung infections that are hard to treat. 

About 1 out of every 3,500 babies who are born alive have TEF. There are five subtypes of TEF based on where the atresia is and how the airway and oesophagus are connected. Most people also have other problems with their spine, intestines, heart, limbs, or kidneys. Tracheoesophageal fistula is a multifactorial complex disease involving several genetic and environmental factors.  In 6%-10% of the patients there is a clear underlying genetic defect which has been diagnosed, however the remaining 90% of the patients the etiology is often unknown.  Certain studies have shown that maternal and in utero exposure to diethyl stilbesterol could also be a risk factor for TEF.

Acquired TOF is further divided into malignant and benign categories. Each entity makes up approximately half of the acquired cases.  The most common cancer associated with malignant TOF is oesophageal cancer, with >10% of patients developing the condition during its clinical course. The most common presentations of TOF are respiratory distress, dysphagia and recurrent lung infections; the magnitude of symptoms depend largely on its size and location.

The management of TEF is surgical repair which consists of separation of the esophagus from the trachea and closure of the defect in the tracheal wall. The presence of long gap between the atretic segments of the esophagus makes the esophageal repair more complex and challenging.  Multiple reports recommended delayed primary repair with staged approach in cases of long gap esophageal atresia or in premature infants with respiratory distress or associated congenital heart disease.  Surgical repair can be performed via thoracotomy or thoracoscopically. Thoracoscopic approach requires additional surgical experience, but its advantages include less surgical trauma and faster postoperative recovery.  Other procedures such as endoscopic fibrin occlusion, sclerotherapy, and laser coagulation exist; however they were associated with high recurrence rates.

Morbidity and mortality rates are dependent on multiple factors related to the patient’s condition, associated anomalies, surgical technique, and long-term complications. They are also highly variable between centres. Spitz classification  is the most common classification used to predict survival rates and is used as a preoperative predictor to aid in parental counselling and comparison of outcomes among pediatric centres. It is based on the presence of low birth weight <1500 grams and/or major congenital cardiac anomalies. It divided cases into 3 subgroups: group I consists of newborns with birth weight above 1500 grams without a major cardiac anomaly, group II consists of newborns with birth weight below 1500 grams or a major cardiac anomaly, and group III consists of newborns with birth weight below 1500 grams and a major cardiac anomaly. Survival rates of patients with TEF were initially reported by Spitz et al. at 97%, 59%, and 22% in groups I, II, and III, respectively. The survival rates have considerably improved due to advances in anaesthesia, surgical, and neonatal intensive care. They currently stand at 98%, 82%, and 50% for group I, group II, and group III, respectively.

Early diagnosis, prompt support in the neonatal intensive care unit, and advancement in surgical techniques positively affect the prognosis and decrease morbidity postoperatively.  Chronic cough, tracheomalacia, recurrent pulmonary infections, bronchiolitis, dyspnea, and asthma are more often reported in the infantile period; long-term bronchiectasis and scoliosis may also develop.  Concomitant tracheomalacia, swallowing dysfunction, and recanalization of fistula may lead to chronic cough and recurrent lung infections.  Other associated anomalies such as laryngomalacia, laryngeal cleft, and vocal cord dysfunction (VCD) may increase the risk of pharyngoesophageal aspiration. ,  The present study is conducted to study the diagnosis, prognosis and outcome of  tracheoesophageal fistula (TEF) disease study at tertiary care centre.

A retrospective and Prospective Study was conducted at Department of General Surgery for two years SAMPLE SIZE: Where, p = Guess of population P = 0.40 q = 1-P = 0.60 Z1 = 1.96 at ἁ =10% level of significance d = absolute precision = ± 0.12 Descriptive statistical method: Prevalence for sample size estimation was taken as 0.40 based on previously published Indian tertiary-centre studies on tracheoesophageal fistula, where approximately 38-42% of patients fell within specific high- risk categories Minimum sample size for the study was 46 METHODOLOGY: This was a retrospective and prospective study which was carried out in the Department of Surgery for a period of five years between 2020 and 2024. All the patients who were admitted in our tertiary care hospital with a diagnosis of TEF during the study period was taken up for the study. All patients who were diagnose with TEF in early neonatal period whether they were out born or whether they were inborn were included in the study; all the children were stabilised preoperatively and diagnosis was confirmed by putting red rubber catheter and X-ray chest and abdomen ( AP - Lateral ) views were taken (Fig 9). FIGURE 9: X-ray chest and abdomen (AP/Lateral) demonstrating red rubber catheter in situ showing presence of obstruction at upper oesophageal pouch. We do not do preoperative dye studies because it was not recommended; patient were kept on preoperative suction so that pulmonary optimisation could be done because recurrent aspiration is the main problem in the babies even in preop period and then they were subjected for surgery; so mode of surgery was either open surgery or thoracoscopic TEF repair and then primary anastomosis was tried, in those pts where primary anastomosis was not possible or patients with primary TEA, where there is no fistula, those patient underwent primary oesophagostomy and gastrostomy. 2D echo in postoperative patients done for cardiac evaluation.Contrast esophageal- swallow :  3 months after study  One year after surgery INCLUSION CRITERIA:  All patients diagnosed with trachea-oesophageal fistula.  All survived patients after surgery of trachea-esophageal fistula. EXCLUSION CRITERIA:  Patients those who expired immediately (within three days) after surgery they are not consider for long-term follow up. DATA COLLECTION: Data was elicited from the hospital records. Data pertaining to the demographic characteristics (age and sex), clinical presentation, antenatal period, and the type of TEF was recorded. Data pertaining to the outcome of surgery and hospitalization was also recorded. Particulars related to requirement of ventilator support was also documented. Statistical Analysis 1. The data was coded and entered into Microsoft Excel spreadsheet. Analysis was done using IBM SPSS (SPSS Inc., IBM Corporation, NY, USA) Statistics Version 25 for Windows software program. 2. Presentation of data – • Frequency distribution tables (Percentage) • Graphical presentation 3. Descriptive statistics – Mean, Standard deviation 4. The statistical tests will be conducted at 5% level of significance.

Age at presentation ranged from 1 to 4 days, with a mean of 1.91 days, suggesting early diagnosis. Birth weight varied from 1.4 to 2.6 kg (mean: 2.04 kg), reflecting that most were low birth weight. Post-operative event days ranged widely (0–36 days), with a mean of ~18 days, indicating variability in post-op complications or outcomes. Slight female predominance was noted (54.3% female vs. 45.7% male), suggesting near-equal gender distribution. 63% were term and 37% preterm, indicating that the majority were full-term neonates, though prematurity remains a considerable fraction. 80.4% were born via normal vaginal delivery (NVD) and only 19.6% via LSCS, suggesting no strong delivery method preference in TEF diagnosis. TYPE A was found 6.5% and TYPE C was found 93.5%. 23.9% of cases had cyanotic congenital heart disease (CHD), an important co-morbidity that may influence prognosis. Found in 28.3%, highlighting the syndromic or multisystem involvement often associated with TEF. A majority (58.7%) required ventilator support post-operatively, indicating high dependency on respiratory support following surgery.

Table 1: Cause of death (surgical/nonsurgical) wise distribution of the study

52.2% mortality rate observed. Leading causes: VAP (21.7%) and sepsis (19.6%), followed by ARDS, anastomotic leak, and cardiac issues, indicating infection as a dominant fatal complication.

Nearly half (47.8%) were discharged; the rest expired (52.2%), indicating a post- operative management needs lot of improvement.

Majority of neonates (73.9%) weighed between 1.5–2.5 kg, and 15.2% were <1.5 kg, suggesting that low birth weight is common in TEF cases.

Tracheoesophageal fistula (TEF) is a rare but serious congenital anomaly involving an abnormal connection between the trachea and esophagus. Its incidence ranges from 1 in 2,500 to 1 in 4,500 live births globally.  TEF is often associated with other anomalies such as cardiac, spinal, and genitourinary malformations. Despite advances in neonatal care and surgical techniques, the morbidity and mortality associated with TEF remain significant, especially in resource-constrained settings.

This study evaluates the demographic profile, associated anomalies, postoperative care, and survival outcomes in neonates diagnosed with TEF at a tertiary care center. The findings provide insight into prognostic indicators that may guide clinical decision-making.

2. Age at Presentation and Early Diagnosis

The mean age at diagnosis was 1.91 days, with all cases identified within the first four days of life. Early presentation reflects good clinical vigilance and possibly higher awareness of TEF symptoms such as frothing, choking, or failure to feed.  Early diagnosis is essential as delayed intervention can lead to complications such as aspiration pneumonia, electrolyte imbalance, and increased mortality.  In a study conducted with 105 patients in Italy, the mean age of onset of symptoms was 1.5 years, and the age of admission to the advanced center was 7.5 years, which was attributed to the lack of knowledge about multidisciplinary approach and long-term complications after surgery.

3. Birth Weight and Gestational Age

Low birth weight was common in this cohort, with a mean of 2.04 kg. Most neonates (73.9%) weighed between 1.5–2.5 kg, while 15.2% were <1.5 kg. Birth weight has been consistently linked with postoperative outcomes in TEF. Spitz et al. categorized risk groups by birth weight and cardiac anomalies, demonstrating higher mortality in infants weighing <1.5 kg.

Prematurity was present in 37% of the neonates, aligning with global reports that show 20–40% of TEF cases occur in preterm infants. Prematurity often exacerbates the risk of postoperative complications due to immature organ systems and poor nutritional reserves.

In Seo JH et al study,  the average birth weight was 2.5 kg, and more males than females were diagnosed. The preterm infants was 32%. Vaginal and caesarean delivery were found 47% and 52% respectively.

In Bakal U et al study,  the study population consisted of 36 boys and 21 girls with a 95 boy/girl ratio of 1.71. At birth, they were both term (59.6 %) and preterm (40.4 %) newborn infants with a 97 mean birth weight of 2255.26 ± 600.27 g (1150–3600 g).

4. Gender and Mode of Delivery

The gender distribution was nearly equal, with a slight female predominance (54.3%). This differs from several studies reporting a slight male predominance.  Regarding the mode of delivery, 80.4% were born via normal vaginal delivery (NVD), with only 19.6% delivered via lower segment cesarean section (LSCS). Delivery mode does not significantly influence TEF outcomes, though cesarean sections may be preferred in cases with prenatal diagnosis or suspected complications.

Al-Naimi A et al was contained 24 males and 11 females (male to female ratio of 2.2:1). Around half of the patients (49%) were full terms.  Tandon et al., we also found male preponder-ance in these late presenters.  We agree with them that it is due to more preference to boys than girls in our society.

5. Associated Anomalies: CHD and Spinal Malformations

Among the most critical prognostic factors are associated congenital anomalies. In this cohort, 23.9% had cyanotic congenital heart disease (CHD), while 28.3% had spinal anomalies. These findings are consistent with the VACTERL association (vertebral, anal, cardiac, tracheoesophageal, renal, and limb anomalies), commonly observed in TEF cases.

Mortality was highest among infants with both low birth weight and associated CHD. For instance, among <1.5 kg neonates, 28.6% had CHD and all expired. This correlates with earlier studies suggesting cardiac anomalies are the strongest predictors of poor outcomes. Similarly, spinal anomalies also demonstrated a higher incidence in the lower birth weight group, further aggravating prognosis.

The most commonly occurring congenital heart disease as reported by Choi et al,  in order of frequency, was ventricular septal defect, patent ductus arteriosus, atrial septal defect, aortic stenosis, tetralogy of Fallot, truncus, and transposition of great vessels.

Greenwood RD and Rosenthal A have shown that hospital mortality in infants with both trachea esophageal fistula and cardiovascular anomalies was as high as 79% in contrast to 23% among the patients who did not have cardiovascular anomalies.

Different rates of cardiovascular 151 system anomalies were reported by Spitz,  Mortell and 152 Azizkhan,  and Okaemoto et al,  which were 29 %, 153 30.5 %, and 30.5 %; respectively

6. Need for Postoperative Ventilator Support

A substantial proportion of patients (58.7%) required postoperative ventilator support, indicating significant perioperative respiratory distress or surgical complications. Prolonged ventilatory requirement often reflects underlying lung hypoplasia, aspiration, or sepsis.  Ventilator dependence is also linked with nosocomial infections such as ventilator-associated pneumonia (VAP), which was notably the most common cause of death in this cohort. In Prabkaran S et al study  about 58% of the participants required ventilation in both pre-operative and post-operative period of which 36.4% required pre-operative ventilation.

Immediate postoperative complications were encountered in eleven patients (33%). [Al-Naimi A et al]

7. Mortality and Causes of Death

The overall mortality in this cohort was 52.2%, which is higher than in high-resource centers (typically <20%).  The most frequent causes of death were VAP (21.7%) and sepsis (19.6%), followed by ARDS and anastomotic leaks. This suggests a need for stringent infection control measures and better postoperative surveillance. Early surgical intervention is critical, but must be coupled with robust neonatal ICU support.  In-hospital mortality was 9% and it revealed lower survival in patients with associated acute respiratory distress syndrome, ventricular septal defect (VSD), birth weight BW < 1500 g, time of operation within 24 hours of admission, coexisting renal anomaly, imperforate anus, African American race, and lowest economic status.  Therefore, mortality was measured as 22% in Al-Naimi A et al study. This is comparable with the mortality rates in other developing countries in the region;  however it is still higher than other international centers in the North America and Western Europe.

In Seo JH et al study, the overall mortality rate (20%) and morbidity rate (67%) of infants born with TEF/EA are high due to the associated anomalies and gastroesophageal and respiratory complications during infancy.

Notably, only one case each of death due to cardiac cause and anastomotic leak were reported, indicating that surgical technique may be less of a contributor than systemic or respiratory complications.

8. Outcome Correlation with Birth Weight and Comorbidities

A strong inverse relationship was observed between birth weight and mortality. In neonates <1.5 kg, survival was zero. In the 1.5–2.5 kg group, survival was around 50%, while in those >2.5 kg, survival improved significantly. These findings underscore the importance of birth weight as a standalone prognostic factor.

When combined with comorbidities such as CHD or spinal anomalies, mortality further increased. Among neonates with both low birth weight and anomalies, no survivors were reported. This reinforces the established Spitz classification, wherein the combination of low birth weight and cardiac anomalies predicts the poorest outcomes.

TRACHEOESOPHAGEAL FISTULA TYPE (TEF)

Tracheoesophageal fistula (TEF) is a congenital anomaly with various anatomical subtypes, among which Type C is most commonly reported. In the present study, Type C TEF was observed in 93.5% of the cases, while Type A TEF was found in only 6.5%. These findings are consistent with established literature, where Type C, characterized by esophageal atresia with a distal tracheoesophageal fistula, accounts for approximately 85–90% of TEF cases globally. ,

Type A TEF, or isolated esophageal atresia without any fistulous connection, is relatively rare and poses a greater diagnostic and therapeutic challenge due to the absence of air in the stomach, making early radiographic diagnosis more difficult.  The higher frequency of Type C in our study reinforces the need for a high index of suspicion and prompt surgical intervention, particularly in neonates presenting with respiratory distress and frothy secretions.

Implications and challenges in resource-limited settings

This study highlights several challenges unique to low- and middle-income countries. While surgical correction techniques may be standardized globally, postoperative ICU care, access to advanced ventilators, and infection control practices vary greatly. High rates of sepsis and VAP-related mortality point to gaps in post-op care, rather than operative failure.

Moreover, pre-operative stabilization, nutritional support, and timely identification of comorbidities are essential components that impact survival but are often suboptimal in such settings.

This study reaffirms the prognostic relevance of birth weight, gestational maturity, and associated anomalies in determining outcomes of TEF repair. Early diagnosis impact early recognition of TEF through antenatal ultrasound and prompt postnatal evaluation significantly improves surgical readiness and reduces perioperative complications. While early diagnosis and surgical correction are achievable, survival depends heavily on neonatal intensive care support as well. Measures to reduce VAP and sepsis, including strict asepsis, early extubation protocols, and use of prophylactic antibiotics, may improve outcomes. Surgical Outcomes in High-Risk Groups Birth weight <1.5 kg, presence of major congenital heart disease, and delayed presentation remain strong predictors of poor survival, consistent with Spitz and Okamoto prognostic classifications. Importance of Multidisciplinary Care Coordinated management involving neonatology, pediatric surgery, cardiology, and anesthesia teams enhances perioperative stability and postoperative recovery. Role of Intensive Postoperative Monitoring Complications such as anastomotic leak, recurrent fistula, and respiratory distress can be minimized with vigilant ICU care, targeted ventilation strategies, and early detection protocols. Need for Long-Term Follow-Up Survivors often face feeding difficulties, GERD, tracheomalacia, or recurrent respiratory infections, highlighting the importance of structured follow-up into childhood. Mortality in low resource environments can be reduced by improving surgical training, equipment availability and referral system for neonates with suspected TEF. Future directions include routine prenatal screening for TEF and associated anomalies, improved neonatal transport systems, and standardized postoperative care bundles to minimize infection risks. A multicentre database would also help consolidate data and guide evidence-based care protocols.

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