Postoperative Stridor following Repair of Tracheoesophageal Fistula: A Case Report

Postoperative Stridor following Repair of Tracheoesophageal Fistula: A Case Report ABSTRACT We report a case of prolonged post-operative stridor in a full-term neonate who was operated for tracheoesophageal fistula. Initial evaluation including an endoscopy and contrast-enhanced computed tomography scan was normal. Repeat endoscopic evaluation under anesthesia revealed tight aryepiglottic folds. Aryepiglottic split was performed and stridor improved dramatically. Tight aryepiglottic folds should be kept in differential diagnosis in a case of postoperative stridor in an infant. postoperative, stridor, tracheoesophageal fistula, anesthesia CASE HISTORY This full-term, male infant with no significant antenatal and birth history develops severe respiratory distress on Day 2 of life. Infant was diagnosed to have H-type tracheoesophageal fistula (TEF) and was operated for the same on Day 4 of life. Infant was extubated on Day 20 of life and was put on heated humidified high-flow nasal cannula. Soon after extubation, infant developed severe respiratory distress and stridor. Infant was put back on ventilatory support. Flexible laryngoscopy and bronchoscopy were performed under light sedation. Except for mild subglottic edema, no abnormality was detected. Size 3.5 endotracheal (ET) tube was replaced with a Size 3 ET tube and a short course of dexamethasone (0.2 mg/kg/day × 5 days) was administered to decrease the edema. After a 10 day period, infant was weaned to continuous positive airway pressure (CPAP). However, it was not possible to take the infant off the CPAP thereafter. Contrast-enhanced computed tomography (CECT) was performed, and no significant abnormality was detected. Parents were counseled for a tracheostomy but they refused. After 1 month period, when there was no improvement in clinical condition, a repeat laryngoscopy with bronchoscopy was performed under anesthesia. Tight aryepiglottic folds were detected and aryepiglottic split was performed. Infant responded dramatically to treatment and was weaned to room air within 3 days of surgery. DISCUSSION To the best of our knowledge, this is the first case report of a postoperative stridor owing to tight aryepiglottic folds in a neonate. Stridor is an abnormal, high-pitched sound produced by turbulent airflow through a partially obstructed airway at the level of the supraglottis, glottis, subglottis or trachea [1]. Apart from other causes, ET intubation and surgeries in the vicinity or involving respiratory tract itself are not uncommon causes of stridor in infants and children [2, 3]. Our patient was operated for tracheoesophageal fistula and required an intubation of 20 days postoperatively; hence, both the possibilities were kept in mind during evaluation. Endotracheal intubation, although vital to facilitate mechanical ventilation in the intensive care unit and operating room, is associated with the potential development of glottic or subglottic edema, resulting in stridor on extubation [2, 3]. Such extra-thoracic airway obstruction following ET intubation may occur in up to 37% of critically ill pediatric patients [4]. The common causes of stridor include (1) isolated intubation laryngotracheitis; (2) intubation laryngotracheitis with neuromuscular dysfunction and (3) specific airway problems such as laryngomalacia, subglottic stenosis or vocal cord paralysis [5]. The postoperative complications following the repair of TEF, which may lead to stridor include intraoperative damage of recurrent laryngeal nerve, edema owing to surgical manipulation, tracheomalacia or tracheal obstruction. Airway hyperreactivity has also been suggested as a possible explanation for stridor in these procedures and this selected population but little information is available [6, 7]. Recurrent laryngeal nerves (unilateral or bilateral) are vulnerable to damage during operative dissection, as they lie in the groove between the trachea and the esophagus, and are closely related to the fistula. Tracheomalacia causes either a generalized or localized weakness of the trachea and allows the anterior and posterior wall to come together causing stridor when severe [7]. In our patient, for evaluation we performed flexible laryngoscopy with bronchoscopy. Except for a mild subglottic edema, no significant abnormality was present. We performed a CECT, and that was also unremarkable. Infant remained respiratory support (CPAP) dependent for a period of 30 days without any significant improvement. Finally, we decided to repeat laryngoscopy and bronchoscopy under anesthesia, and tight aryepiglottic folds were detected. Aryepiglottic fold split was performed and the infant improved dramatically. Tight aryepiglottic fold is a subtype of laryngomalacia (Type 1) [8]. Cause is unknown. Differentials include congenital stridor, hypocalcemia, subglottic stenosis and respiratory papillomatosis. Laryngomalacia, although the commonest cause of chronic stridor in infants, [9] is not a common cause of postintubation or post-TEF repair surgery stridor [5–7]. Even after an extensive literature search, we could not locate a case report describing tight aryepiglottic folds as a cause of postintubation or postoperative stridor. The diagnostic modality of choice for diagnosing laryngomalacia is fibrooptic laryngoscopy. However, when performed in an awake patient with incomplete sedation, there are chances of both false-positive and false-negative results. The anesthesia technique has been found to be superior to awake technique with a sensitivity, specificity, positive predictive value and negative predictive value of 100% each as compared with 93%, 92%, 97% and 79%, respectively, for awake technique [10]. Most probably we missed the diagnoses in the first place, as we did not perform the laryngoscopy under anesthesia or sufficient sedation. It is worth mentioning that laryngoscopy along with bronchoscopy and esophagoscopy was performed under anesthesia during the initial evaluation of TEF before surgery. This makes us strongly believe that the tight aryepiglottic folds were a complication of TEF repair surgery or prolonged intubation rather than a congenital one. CONCLUSION Tight aryepiglottic fold should be kept in deferential diagnosis in postoperative and postintubation cases of stridor in infants. Patient should be examined under anesthesia or deep sedation if initial evaluation is negative and clinical suspicion is high. References 1 Cotton RT, Reilly JS, Strider and airway obstruction. In: Blue-stone C, Stool S, Kanna M (eds). Pediatric Otolaryngology . 3rd edn. Philadelphia: WB Saunders Co 1995, 1275– 86. 2 Koka BV, Jeon IS, Andre JM, et al.   Postintubation croup in children. Anesth Anal  1977; 56: 501– 5. Google Scholar CrossRef Search ADS   3 Thompson AE, Pediatric airway management. In: Fuhrman BP, Zimmerman JJ (eds). Pediatric critical care . St. Louis: Mosby Year Book 1992, 120– 1. 4 Lin CD, Cheng YK, Chang JS, et al.   Endoscopic survey of post-extubation stridor in children. Acta Peadiatr Taiwan  2002; 43: 91– 5. 5 Beasley SW, Tracheo-oesophageal fistula. In: Atwell JD (ed.). Paediatric surgery . New York, NY: Arnold, 1998, 187– 96. 6 Filler RM, Messineo A, Vinograd I. Severe tracheomalacia associated with esophageal atresia, results of surgical treatment. J Pediatr Surg  1992; 27: 1136– 40. Google Scholar CrossRef Search ADS PubMed  7 Benjamin B. Tracheomalacia in infants and children. Ann Otol Rhinol Laryngol  1984; 93: 438– 42. Google Scholar CrossRef Search ADS PubMed  8 Kay DJ, Goldsmith AJ. Laryngomalcia: a classification system and surgical treatment strategy. Ear Nose Throat J  2006; 85: 328– 31. Google Scholar PubMed  9 Alexander KCL, Helen C. Diagnosis of strider in children. Am Fam Physician  1999; 60: 2289– 96. Google Scholar PubMed  10 Sivan Y, Ben-Ari J, Soferman R, DeRowe A. Diagnosis of laryngomalacia by fiberoptic endoscopy: awake compared with anesthesia-aided technique. Chest  2006; 130: 1412– 8. Google Scholar CrossRef Search ADS PubMed  © The Author [2017]. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Tropical Pediatrics Oxford University Press

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Abstract

ABSTRACT We report a case of prolonged post-operative stridor in a full-term neonate who was operated for tracheoesophageal fistula. Initial evaluation including an endoscopy and contrast-enhanced computed tomography scan was normal. Repeat endoscopic evaluation under anesthesia revealed tight aryepiglottic folds. Aryepiglottic split was performed and stridor improved dramatically. Tight aryepiglottic folds should be kept in differential diagnosis in a case of postoperative stridor in an infant. postoperative, stridor, tracheoesophageal fistula, anesthesia CASE HISTORY This full-term, male infant with no significant antenatal and birth history develops severe respiratory distress on Day 2 of life. Infant was diagnosed to have H-type tracheoesophageal fistula (TEF) and was operated for the same on Day 4 of life. Infant was extubated on Day 20 of life and was put on heated humidified high-flow nasal cannula. Soon after extubation, infant developed severe respiratory distress and stridor. Infant was put back on ventilatory support. Flexible laryngoscopy and bronchoscopy were performed under light sedation. Except for mild subglottic edema, no abnormality was detected. Size 3.5 endotracheal (ET) tube was replaced with a Size 3 ET tube and a short course of dexamethasone (0.2 mg/kg/day × 5 days) was administered to decrease the edema. After a 10 day period, infant was weaned to continuous positive airway pressure (CPAP). However, it was not possible to take the infant off the CPAP thereafter. Contrast-enhanced computed tomography (CECT) was performed, and no significant abnormality was detected. Parents were counseled for a tracheostomy but they refused. After 1 month period, when there was no improvement in clinical condition, a repeat laryngoscopy with bronchoscopy was performed under anesthesia. Tight aryepiglottic folds were detected and aryepiglottic split was performed. Infant responded dramatically to treatment and was weaned to room air within 3 days of surgery. DISCUSSION To the best of our knowledge, this is the first case report of a postoperative stridor owing to tight aryepiglottic folds in a neonate. Stridor is an abnormal, high-pitched sound produced by turbulent airflow through a partially obstructed airway at the level of the supraglottis, glottis, subglottis or trachea [1]. Apart from other causes, ET intubation and surgeries in the vicinity or involving respiratory tract itself are not uncommon causes of stridor in infants and children [2, 3]. Our patient was operated for tracheoesophageal fistula and required an intubation of 20 days postoperatively; hence, both the possibilities were kept in mind during evaluation. Endotracheal intubation, although vital to facilitate mechanical ventilation in the intensive care unit and operating room, is associated with the potential development of glottic or subglottic edema, resulting in stridor on extubation [2, 3]. Such extra-thoracic airway obstruction following ET intubation may occur in up to 37% of critically ill pediatric patients [4]. The common causes of stridor include (1) isolated intubation laryngotracheitis; (2) intubation laryngotracheitis with neuromuscular dysfunction and (3) specific airway problems such as laryngomalacia, subglottic stenosis or vocal cord paralysis [5]. The postoperative complications following the repair of TEF, which may lead to stridor include intraoperative damage of recurrent laryngeal nerve, edema owing to surgical manipulation, tracheomalacia or tracheal obstruction. Airway hyperreactivity has also been suggested as a possible explanation for stridor in these procedures and this selected population but little information is available [6, 7]. Recurrent laryngeal nerves (unilateral or bilateral) are vulnerable to damage during operative dissection, as they lie in the groove between the trachea and the esophagus, and are closely related to the fistula. Tracheomalacia causes either a generalized or localized weakness of the trachea and allows the anterior and posterior wall to come together causing stridor when severe [7]. In our patient, for evaluation we performed flexible laryngoscopy with bronchoscopy. Except for a mild subglottic edema, no significant abnormality was present. We performed a CECT, and that was also unremarkable. Infant remained respiratory support (CPAP) dependent for a period of 30 days without any significant improvement. Finally, we decided to repeat laryngoscopy and bronchoscopy under anesthesia, and tight aryepiglottic folds were detected. Aryepiglottic fold split was performed and the infant improved dramatically. Tight aryepiglottic fold is a subtype of laryngomalacia (Type 1) [8]. Cause is unknown. Differentials include congenital stridor, hypocalcemia, subglottic stenosis and respiratory papillomatosis. Laryngomalacia, although the commonest cause of chronic stridor in infants, [9] is not a common cause of postintubation or post-TEF repair surgery stridor [5–7]. Even after an extensive literature search, we could not locate a case report describing tight aryepiglottic folds as a cause of postintubation or postoperative stridor. The diagnostic modality of choice for diagnosing laryngomalacia is fibrooptic laryngoscopy. However, when performed in an awake patient with incomplete sedation, there are chances of both false-positive and false-negative results. The anesthesia technique has been found to be superior to awake technique with a sensitivity, specificity, positive predictive value and negative predictive value of 100% each as compared with 93%, 92%, 97% and 79%, respectively, for awake technique [10]. Most probably we missed the diagnoses in the first place, as we did not perform the laryngoscopy under anesthesia or sufficient sedation. It is worth mentioning that laryngoscopy along with bronchoscopy and esophagoscopy was performed under anesthesia during the initial evaluation of TEF before surgery. This makes us strongly believe that the tight aryepiglottic folds were a complication of TEF repair surgery or prolonged intubation rather than a congenital one. CONCLUSION Tight aryepiglottic fold should be kept in deferential diagnosis in postoperative and postintubation cases of stridor in infants. Patient should be examined under anesthesia or deep sedation if initial evaluation is negative and clinical suspicion is high. References 1 Cotton RT, Reilly JS, Strider and airway obstruction. In: Blue-stone C, Stool S, Kanna M (eds). Pediatric Otolaryngology . 3rd edn. Philadelphia: WB Saunders Co 1995, 1275– 86. 2 Koka BV, Jeon IS, Andre JM, et al.   Postintubation croup in children. Anesth Anal  1977; 56: 501– 5. Google Scholar CrossRef Search ADS   3 Thompson AE, Pediatric airway management. In: Fuhrman BP, Zimmerman JJ (eds). Pediatric critical care . St. Louis: Mosby Year Book 1992, 120– 1. 4 Lin CD, Cheng YK, Chang JS, et al.   Endoscopic survey of post-extubation stridor in children. Acta Peadiatr Taiwan  2002; 43: 91– 5. 5 Beasley SW, Tracheo-oesophageal fistula. In: Atwell JD (ed.). Paediatric surgery . New York, NY: Arnold, 1998, 187– 96. 6 Filler RM, Messineo A, Vinograd I. Severe tracheomalacia associated with esophageal atresia, results of surgical treatment. J Pediatr Surg  1992; 27: 1136– 40. Google Scholar CrossRef Search ADS PubMed  7 Benjamin B. Tracheomalacia in infants and children. Ann Otol Rhinol Laryngol  1984; 93: 438– 42. Google Scholar CrossRef Search ADS PubMed  8 Kay DJ, Goldsmith AJ. Laryngomalcia: a classification system and surgical treatment strategy. Ear Nose Throat J  2006; 85: 328– 31. Google Scholar PubMed  9 Alexander KCL, Helen C. Diagnosis of strider in children. Am Fam Physician  1999; 60: 2289– 96. Google Scholar PubMed  10 Sivan Y, Ben-Ari J, Soferman R, DeRowe A. Diagnosis of laryngomalacia by fiberoptic endoscopy: awake compared with anesthesia-aided technique. Chest  2006; 130: 1412– 8. Google Scholar CrossRef Search ADS PubMed  © The Author [2017]. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com

Journal

Journal of Tropical PediatricsOxford University Press

Published: Feb 1, 2018

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