Home Print this page Email this page Small font sizeDefault font sizeIncrease font size
Users Online: 908
 
About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts Advertise Login 
     


 
 Table of Contents  
ORIGINAL ARTICLE
Year : 2019  |  Volume : 8  |  Issue : 2  |  Page : 71-74

Experience of tracheo-esophageal fistula in neonates in a Tertiary Care Center - Case series


1 Department of Neonatology, Dr. Bidari's Ashwini Hospital, Vijayapura, Karnataka, India
2 Department of Pediatrics Surgery, Dr. Bidari's Ashwini Hospital, Vijayapura, Karnataka, India
3 Department of Pediatrics, Dr. Bidari's Ashwini Hospital, Vijayapura, Karnataka, India

Date of Web Publication25-Apr-2019

Correspondence Address:
Dr. Siddu Charki
Department of Neonatology, Dr. Bidari's Ashwini Hospital, Vijayapura, Karnataka
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcn.JCN_69_18

Rights and Permissions
  Abstract 


Introduction: Tracheoesophageal fistula (TEF) is one of the most common neonatal emergencies. The most common presentation being polyhydramnios detected antenatally, excessive salivation and vomiting, respiratory distress after birth, recurrent pneumonia later in life. The incidence is 1 in 3000 to 1 in 4500 live births. Clinical Profile: Of 1206 admissions in 2017 to the neonatal intensive care unit (NICU), 51 required surgery. Out of which 11 babies were diagnosed with TEF and were subjected to surgery. Antenatal scans revealed polyhydramnios in four babies. Nine babies were born at term with an average weight of 2–2.5 kg and two babies were born preterm at 30 weeks (1.3 kg) and 32 weeks (1.8 kg). Eight babies presented on day 1–2 of life and three babies on day 2–3. The most common clinical presentation was excessive frothing from the mouth noticed since birth and respiratory distress. Nine babies (82%) presented with chest infection of varying severity. Only two babies (18%) had a clear chest. Babies were stabilized in NICU and connected to Replogle tube with continuous negative suction. All babies were subjected to surgery within 24 h of admission. Type C was the most common. Babies were subjected to contrast study to rule out anastomotic leak. Feeding initiated after 72 h of life and was gradually started on trophic feeds and reached full feeds. Outcome: Out of 11 babies, 9 babies recovered and were discharged. One baby was discharged against medical advice and one baby died due to sepsis. Growth is satisfactory and development has been normal at 1 year of age in all discharged babies at follow-up. Discussion: Success in the survival of neonate with TEF is attributed to improved neonatal intensive care with surgical advances and postoperative care. Early recognition, prompt and efficient management of the cases was possible due to multidisciplinary approach by neonatologist, intensivist, and the surgeon. A precise surgical technique with proper mobilization of upper pouch and good anastomosis is key events.

Keywords: Anastomosis, polyhydramnios, tracheoesophageal fistula


How to cite this article:
Charki S, Priyadarashini M K, Hadalgi L, Agarwal S, Kulkarni T, Loni R, Bidari LH. Experience of tracheo-esophageal fistula in neonates in a Tertiary Care Center - Case series. J Clin Neonatol 2019;8:71-4

How to cite this URL:
Charki S, Priyadarashini M K, Hadalgi L, Agarwal S, Kulkarni T, Loni R, Bidari LH. Experience of tracheo-esophageal fistula in neonates in a Tertiary Care Center - Case series. J Clin Neonatol [serial online] 2019 [cited 2019 Oct 22];8:71-4. Available from: http://www.jcnonweb.com/text.asp?2019/8/2/71/257141




  Introduction Top


The incidence of tracheoesophageal fistula (TEF) is 1:3000–4500 of live births which is a congenital anomaly of esophagus and trachea that manifests within the first few hours to days of life. The most common defect is esophageal atresia (EA) with distal TEF (type C/IIIB as described by Gross and Vogt). It is considered a surgically correctable anomaly of the gastrointestinal and respiratory system and continues to be a major challenge in neonatal surgery. However, with surgical repair, the rate of survival exceeds 90%, even in infants with low birth weight. The factors of major concern are prematurity, low birth weight, difficult airway of neonate, associated respiratory distress because of repeated aspirations or lung pathologies, congenital heart diseases (CHD), and large defects.[1],[2] Evaluation with X-ray chest after passing nasogastric (NG) tube, electrocardiograph, ECHO, computed tomography scan, abdominal and lumbar ultrasound for vertebral anomaly, and rigid bronchoscopy for evaluation of the anatomy of airway is considered necessary for anesthetic and postoperative intensive care planning. Corrective measures for low-birth weight, anemia, medical optimization of aspiration pneumonia, or congestive cardiac failure are necessary; thus, the management of the TEF needs multidisciplinary approach involving neonatologist, intensivist, pediatric surgeon as well as anesthesiologist for successful surgical outcome.[1],[2] We report a case series of TEF in neonates, 1-year experience in a tertiary care center with resource-limited settings.


  Case Series Profile Top


Out of 1206 admissions to neonatal intensive care unit (NICU) in 2017, 51 required surgery. Out of which 11 babies were diagnosed with TEF. Antenatal scans had revealed polyhydramnios in seven babies. Nine babies were born at term with an average weight of 2–2.5 kg and two babies were born preterm at 30 weeks (1.3 kg) and 32 weeks (1.8 kg) as shown in [Figure 1]. Eight babies presented on day 1–2 of life and three babies on day 2–3. The most common clinical presentation was excessive frothing from the mouth noticed since birth and respiratory distress. Nine babies (82%) presented with chest infection of varying severity. Only two babies (18%) had chest which was clear. Basic characteristics of neonates have been enlisted in [Table 1].
Figure 1: Distribution of weight in babies

Click here to view
Table 1: Baseline characteristics of neonates

Click here to view


Anatomical type of EA with TEF: 9 infants (81%) had EA with TEF in which there was EA with distal fistula (Type-C); one was Type B and one of a type as shown in [Figure 2]. Associated anomalies: three babies had ventricular septal defect (VSD), four babies had patent ductus arteriosus (PDA), and two had anorectal malformation.
Figure 2: Distribution of babies as per Gross's classification

Click here to view



  Management Top


Babies were stabilized in NICU and connected to Replogle tube with continuous negative suction. Echo screening was done in all neonates which revealed three babies had VSD and four had PDA. Babies were screened to rule out VACTERL association. Of 11 babies, three were ventilated due to respiratory distress with significant Downe's score and pulmonary hypertension. Babies with hemodynamically significant PDA with persistent pulmonary hypertension (PPHN) were started on intravenous (IV) paracetamol for PDA closure. All babies were operated on within 48–72 h of life after initial stabilization.

Intraoperatively, 82% of babies underwent standard primary anastomosis and 18% had difficult anastomosis. Surgery: repair of defect with anastomosis. Postoperative ventilator support was required after TEF surgery in neonates for 24–48 h with adequate sedation and pain management. Antibiotics were given as per unit protocol, intermittent NG aspiration, and postoperative epidural analgesia provided for 48 h. Postoperatively, babies were kept nil per orally for 72 h of life and was gradually started on trophic feeds and reached full feeds. Four babies had features of feed intolerance, and one baby developed necrotizing enterocolitis (NEC) which was treated as per standard protocol. Babies were subjected to contrast study to rule out anastomotic leak on day 3 postoperation. Early recognition, prompt and efficient management of the cases were possible due to the multidisciplinary approach by neonatologist, intensivist, and the surgeon.


  Results of Operation Top


Out of 11 babies, nine babies recovered and were hemodynamically stable on full feeds with adequate weight gain at discharge. One baby succumbed to sepsis. One baby went against medical advice as shown in [Figure 3]. Growth is satisfactory and development has been normal at 1 year of age in all discharged babies at follow-up.
Figure 3: Distribution of babies in terms of outcome

Click here to view



  Discussion Top


The worldwide incidence of EA with TEF is 1 in 3000 births with no particular preponderance of sex or race.[3] In our study, we found that 65% of the cases were males. The anatomical classification describes five types – I, II, IIIA, IIIB, IIIC depending on the esophageal and tracheal defects, but the most common is type III that is, EA with distal TEF. In this case series, nine babies were born at term with weight >2 kg and two babies were born preterm at 30 weeks (1.3 kg) and 32 weeks (1.8 kg) and 65% were male. Most of the cases were born by institutional delivery 80% and rest were home deliveries by trained attendants.

The mean age at presentation was 2.12 ± 1.87 days. The most common clinical presentation was excessive frothing from the mouth noticed since birth and respiratory distress.

Patients of EA with TEF are prone to have other associated congenital anomalies - the presence of which adversely affects the outcome. TEF may be associated with some abnormal gene-derived syndromes and anomalies such as VATER or VACTERL with frequent cardiac and anorectal defects.[3] We observed incidences of associated 40% VSD, 30% PDA, and 20% anorectal malformation.

Broemling and Campbell.[4] in a review observed success rate in various studies by Waterson et al., Spitz et al., Okamota and found >95% of survival in babies weighing >2 kg, 59%–72% if associated with cyanotic heart diseases or major CHD and 68% when associated with lobar pneumonia, atrial septal defect/PDA or other congenital limb anomalies, or cleft palate. Babies with severe pneumonia or severe congenital cardiac anomalies when weight is <1.5 kg had survival rates of 6%.[4] Millano et al.[5] studied different predictors of mortality in a series of 31 cases of TEF and observed high mortality in 26 (31) cases due to sepsis in 20 (26), low platelet count in 19 (20), low albumin levels in 11 (11). A low birth weight, anastomotic leak, respiratory failure, and postoperative sepsis were independent risk factors for poor outcomes.[6]

In this case series, three babies were sepsis screen positive and culture positive, were started on IV antibiotics as per unit protocol. Two babies received platelet transfusion due to low platelet count, and one preterm baby received PRBC transfusion due to low hemoglobin as per transfusion guidelines. Four were diagnosed with aspiration pneumonia. The survival rate was >80%. Out of 11 babies, nine babies recovered and were hemodynamically stable on full feeds with adequate weight gain at discharge. One baby which succumbed had sepsis with aspiration pneumonia with significant PPHN. One baby went against medical advice and was lost to follow-up.

Although low birth weight, the presence of other associated congenital anomalies, and presence of pneumonia are all risk factors in their individual right, relating each of them alone with mortality may not be a good idea as they are often co-existent. Hence, we made an attempt to employ Waterston's criteria to classify risk factors and evaluate the prognosis and therapeutic problems depicted in [Table 2].
Table 2: Preoperative risk classification (Waterston's criteria)

Click here to view


Waterston's classification is still relevant in developing countries where it is a good predictor of survival as evident in our study. In this regard perhaps, the new preoperative risk classification proposed by Yagyu et al.[7] suggested a better outcome [Table 3] based on the severity of grading in which risk factors described by Spitz et al.[2] (birth weight, associated cardiac anomaly) were supplemented with respiratory distress syndrome and pneumonia. In our study, we found that overall 9 (82%) cases could be treated successfully and discharged in a healthy condition. Gross[8]-classified EA with or without fistula into five types from A-E. In our study, nine infants (81%) had EA with TEF in which there was EA with distal fistula (Type-C); one was Type B and one of A Type.
Table 3: Preoperative risk classification (Yagyu's criteria)

Click here to view



  Conclusion Top


Success in the survival of a neonate with TEF is attributed to improved neonatal intensive care with surgical advances and postoperative care. Males are overwhelmingly more among the cases of EA with TEF than females. Waterston's preoperative risk factor classification still holds good in hospitals of developing countries to reflect the prognosis and therapeutic problems of EA, but preoperative risk classification proposed by Yagu et al. is preferred. Overall survival rate was 82%, and with associated congenital anomalies, survival was 50%. Hence, screening and management of associated congenital anomalies earlier improve the survival. A precise surgical technique with proper mobilization of upper pouch and good anastomosis is key events for better surgical outcome.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Uluba M, Dayi S. Tracheoesophageal fistula in a newborn. N Engl J Med 2014;371:e23.  Back to cited text no. 1
    
2.
Spitz L, Kiely EM, Morecroft JA, Drake DP. Oesophageal atresia: At-risk groups for the 1990s. J Pediatr Surg 1994;29:723-5.  Back to cited text no. 2
    
3.
Morray JP, Krane EJ, Geiduschek JM, O'Rourke PP. Aaesthesia for thoracic surgery. In: Gregory GA, editor. Pediatric Anesthesia. 3rd ed. New York: Churchill Livingstone; 1994. p. 421-64.  Back to cited text no. 3
    
4.
Broemling N, Campbell F. Anesthetic management of congenital tracheoesophageal fistula. Paediatr Anaesth 2011;21:1092-9.  Back to cited text no. 4
    
5.
Millano L, Agustriani N, Rochadi R. Predictors of mortality in newborns with esophageal atresia: A 6-year study in a single institution. Paediatr Indones 2015;55:131-5.  Back to cited text no. 5
    
6.
Kinottenbelt G, Skinner A, Seefelder C. Tracheo-oesophageal fistula (TOF) and oesophageal atresia (OA). Best Pract Res Clin Anaesthesiol 2010;24:387-401.  Back to cited text no. 6
    
7.
Yagyu M, Gitter H, Richter B, Booss D. Esophageal atresia in Bremen, Germany – Evaluation of preoperative risk classification in esophageal atresia. J Pediatr Surg 2000;35:584-7.  Back to cited text no. 7
    
8.
Gross RE. The Surgery of Infancy and Childhood. Philadelphia: W.B. Saunders; 1953. p. 76.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1], [Table 2], [Table 3]



 

Top
 
 
  Search
 
Similar in PUBMED
  Search Pubmed for
  Search in Google Scholar for
Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Series Profile
Management
Results of Operation
Discussion
Conclusion
References
Article Figures
Article Tables

 Article Access Statistics
    Viewed1110    
    Printed45    
    Emailed0    
    PDF Downloaded206    
    Comments [Add]    

Recommend this journal