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Abstract
Introduction
Case Reports
Discussion
Conclusion
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  Table of Contents 
CASE REPORT
Year : 2020  |  Volume : 21  |  Issue : 1  |  Page : 70-73
 

Pediatric airway management in undiagnosed congenital subglottic stenosis patients


1 Department of Anaesthesia and Critical Care, Command Hospital (Southern Command), Armed Forces Medical College, Pune, Maharashtra, India
2 Department of ENT, Command Hospital, Armed Forces Medical College, Pune, Maharashtra, India

Date of Submission02-Sep-2019
Date of Acceptance15-Sep-2019
Date of Web Publication13-Feb-2020

Correspondence Address:
Dr. Deepak Dwivedi
Department of Anaesthesia and Critical Care, Command Hospital (Southern Command), Armed Forces Medical College, Pune - 411 040, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/TheIAForum.TheIAForum_63_19

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  Abstract 


Pediatric unanticipated difficult airway in cases of subglottic stenosis (SGS) is always a challenge. We present two cases of SGS which were managed according to the pediatric difficult intubation protocols where supraglottic device was used as secondary intubation plan successfully. Airway management in these cases depends on the urgency of the surgery; if the surgery is nonemergent, the case can be postponed and both surgical and nonsurgical modalities of treatment can be offered; however, in wake of emergency surgery, surgical airway is indicated. Strategies of the pediatric airway management have evolved over the years, but the risk and morbidity associated with the adverse airway events loom larger.


Keywords: Airway management, bronchoscopy, neonate, subglottic stenosis


How to cite this article:
Dwivedi D, Dwivedi G, Gupta V, Kate S. Pediatric airway management in undiagnosed congenital subglottic stenosis patients. Indian Anaesth Forum 2020;21:70-3

How to cite this URL:
Dwivedi D, Dwivedi G, Gupta V, Kate S. Pediatric airway management in undiagnosed congenital subglottic stenosis patients. Indian Anaesth Forum [serial online] 2020 [cited 2020 Apr 7];21:70-3. Available from: http://www.theiaforum.org/text.asp?2020/21/1/70/278186





  Introduction Top


Pediatric airway management can pose a challenge to an anesthesiologist. The incidence of unpredicted difficult intubation in the pediatric population can vary between 0.05–0.57% in infants and 0.07%–0.7% in older children.[1],[2] The incidence of unpredicted difficult intubation is although rare since most children with difficult airway can be identified during preoperative evaluation. However, unpredictable difficulties at intubation or ventilation may become a cause for high mortality and morbidity.[3],[4] Subglottic stenosis (SGS) has an incidence between 0.63% and 2.0% in the neonatal period, and in majority of the cases, it is acquired in nature following prolonged intubation during the immediate postnatal period.[5] Congenital SGS occurs as a part of congenital genetic syndromes and considered to be the third most common cause of stridor in this period of life after laryngomalacia and vocal cord palsy.[6] We present two cases of undiagnosed congenital SGS, leading to unanticipated difficult intubation scenarios in pediatric surgical patients.


  Case Reports Top


Case 1

A 4-year-old female child weighing 15 kg a known case of the congenital radial club hand was planned for centralization of the limb under general anesthesia (GA) with endotracheal intubation. Preoperative airway examination revealed no abnormalities. Mouth opening was adequate for her age. The patient had no symptoms suggestive of respiratory distress, stridor, and prolonged intubation. Systemic examination and all relevant investigations were within normal limits. The patient was accepted in the American Society of Anesthesiologist Physical Status Class I and written informed consent was obtained from her parents. On the day of surgery, syrup midazolam 0.5 mg/kg was given orally in the preoperative room and the patency of the intravenous (IV) access was ascertained. After preparing the operation theater (OT) and completing the preliminary check of anesthesia machine, the child was wheeled in under parental presence inside the OT. The patient was coinduced with injection fentanyl 1.5 μg/Kg IV and injection propofol 2 mg/kg. The adequacy of the mask ventilation was established following which injection atracurium 0.5 mg/kg was administered. Direct laryngoscopy revealed Cormack–Lehane Grade II view of the glottic opening. Two attempts to intubate with cuffed endotracheal tube size 4.0 and 3.5 were unsuccessful. Each attempt was done after preoxygenating the child with 100% oxygen; hence, on no occasion, the oxygen saturation went below 99%. Supraglottic airway, i-gel®, size 2 was inserted as a rescue device. Ventilation with i-gel® was confirmed followed by fiberoptic bronchoscopy with Ambuscope (Ambu® aScope™ 3 Slim 3.8/1.2) which delineated the stenosis in the subglottic region [Figure 1]a. In wake of prolonged duration of surgery, multiple attempts of intubation, and freshly diagnosed SGS, decision for the postponement of the surgery was taken. On return of the spontaneous respiration, the patient was reversed and i-gel® was removed successfully. After extubation, the patient developed stridor with subcostal retractions. Nebulization with adrenaline (1:1000) 0.5 mg/kg was initiated and continued for 15 min till the retractions improved and the child settled with normal breathing. The patient was shifted to the pediatric intensive care unit (PICU) for the monitoring.
Figure 1: (a) Grade II and (b) Grade III congenital subglottic stenosis

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Case 2

An 18-month-old child weighing 8 kg, a known case of pure esophageal atresia with duodenal atresia, was operated within 24 h after the birth. The neonate was full term and weighed 2.8 kg at the time of birth. He underwent cervical esophagostomy with gastrostomy and duodenoduodenostomy for duodenal atresia under GA with a history of difficult intubation and the passage of 2.0-mm size uncuffed endotracheal tube. Postoperatively, the neonate was ventilated for 12 h and was successfully extubated. Remaining postoperative period was uneventful. The child was slated to undergo gastric pull-up surgery and the closure of the cervical esophagostomy. Preoperative investigations were within normal limits. Parents were counseled for the assessment of the airway by ENT surgeon and in case of any adverse airway event; consent was obtained for postprocedure ventilation, tracheostomy in view of history of difficult intubation. Difficult airway cart was kept ready with straight as well as curved laryngoscope blades, second-generation supraglottic airways, including i-gel®, Ambu® AuraGain™ laryngeal mask airway (LMA) and pediatric fiberoptic bronchoscope (Pentax, model FI 9 RBS [OD 3.1 mm, ID 2.9 mm], New Jersey USA). Standard monitoring ensued, and the child was induced by the inhalational technique with sevoflurane (5%–6%) while maintaining spontaneous respiration. Fentanyl 1.5 μg/Kg IV was given after confirming the ability to ventilate with the mask. A gentle laryngoscopy was done which revealed a pinhole-like opening in the subglottic region [Figure 1]b. An i-gel® was inserted to facilitate the fiberoptic bronchoscopy without compromising the ventilation; however, the seal was improper and the ventilation was impeded. Ambu® AuraGain™ LMA size 1.5 was successfully inserted and the egress of air from the cervical esophagostomy site in the neck was prevented by applying a gauze over it. Fiberoptic bronchoscopy staged it as a Grade 3 SGS. LMA was removed once fiberoptic bronchoscopy was completed and the spontaneous breathing commenced. On recovery, the child developed stridor with suprasternal and subcostal retractions; immediately, the child was propped up and nebulization with adrenaline was started as in the first case with 100% oxygen delivered through the nasal prongs. The child stabilized after an hour and was then shifted to PICU for observation. Both the cases described above underwent balloon dilatations before being planned for the elective surgeries.


  Discussion Top


Normal diameter of new-born trachea is 5 mm; lumen of 4 mm in full-term new-born and 3 mm in the premature infant represents SGS.[6] Proposed mechanism is incomplete recanalization during embryogenesis. Congenital SGS may vary in severity from small glottic webs, mild lateral narrowing, or to complete laryngeal atresia. Congenital and acquired SGS both are evaluated and staged with Myer–Cotton system with the aid of microlaryngoscopy and bronchoscopy. Meyer–Cotton classifies luminal narrowing as Grade I with <50%, Grade II with 51%–70%, Grade III with 71%–90%, and Grade IV with 100% obstruction.[7] Our first case was Grade II and the second case when presented during the neonatal period was Grade II to begin with which later progressed to Grade III.

Congenital SCS can be an unexpected observation during intubation since diagnosing it antenatally is difficult with the normal airway above the glottis.[6] Diagnosis eludes in such cases till the neonate present for surgery in few hours after birth.[8],[9] Inflammation in the form of upper respiratory tract infection (URTI) manifests as stridor with or without suprasternal and subcostal retractions in cases presenting in infancy and later.[6] Positive history of stridor during bouts of URTI was elucidated after repeatedly probing the parents in our second case, and both patients developed stridor with chest retractions in the immediate postoperative period following the repeated attempts of intubation and fiberoptic bronchoscopy.

In both cases, unanticipated pediatric difficult intubation guidelines (2016) as proposed by All India Difficult Airway Association was followed and step 2 of secondary intubation plan with second-generation supraglottic device (SGD) was used as a rescue measure successfully.[2] It is important to note that due to the variation in the anatomy of the airway in pediatrics, one should have varied SGD (i-gel®, LMA Supreme, Ambu® AuraGain™, and AuraOnce™) available in the armamentarium as one type of SGD may not form seal in all the airways as was evident in our case.

Pediatric adverse airway events are more common than adults resulting in hypoxemia, cardiac arrest, and hypoxic brain damage.[10] Pediatric difficult intubation registry as well as Jagannathan et al. advocated measures to enhance the patient safety and limit the morbidity by minimizing the direct laryngoscopy attempts, early transition to indirect laryngoscopy (videolaryngoscope), and maintenance of oxygenation through either SGD or nasal cannula.[10],[11]

Literature shows the association of genetic as well as congenital anomalies such as Down syndrome, duodenal atresia, tracheoesophageal fistula, tetralogy of Fallot, and Fraser syndrome with SGS raising suspicion of unanticipated difficult airway.[2],[8] Urgency of surgery will decide further airway management. The elective surgery can be postponed and the treatment modality offered is sequential balloon dilatation with fairly good results in lesser grade with short-segment SGS which was offered to both of our patients.[2],[12],[13] Surgical modalities include anterior cricoid split recommended for children <6 months and laryngotracheoplasty resection with the use of laser for children in age groups more than 6 months.[14] Emergency neonatal surgeries in cases with higher grade SGS will require tracheostomy to keep the airway patent.[8]


  Conclusion Top


Unanticipated pediatric difficult intubation can pose a challenge and more so in patients of SGS presenting as a surprise during the surgeries in infancy. It is an indomitable task to prevent adverse airway event-related morbidity in this age group. Evidence exists in literature emphasizing the role of pediatric difficult intubation guidelines which are when followed in principle has changed the outcomes. We recommend that we should always have different types of SGD as one may not form seal in all the neonates and infants with varied airway anatomy.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the child's parents have given their consent for her images and other clinical information to be reported in the journal. The child's parents understand that her names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Weiss M, Engelhardt T. Proposal for the management of the unexpected difficult pediatric airway. Paediatr Anaesth 2010;20:454-64.  Back to cited text no. 1
    
2.
Pawar DK, Doctor JR, Raveendra US, Ramesh S, Shetty SR, Divatia JV, et al. All India difficult airway association 2016 guidelines for the management of unanticipated difficult tracheal intubation in paediatrics. Indian J Anaesth 2016;60:906-14.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Heinrich S, Birkholz T, Ihmsen H, Irouschek A, Ackermann A, Schmidt J. Incidence and predictors of difficult laryngoscopy in 11,219 pediatric anesthesia procedures. Paediatr Anaesth 2012;22:729-36.  Back to cited text no. 3
    
4.
Burjek NE, Nishisaki A, Fiadjoe JE, Adams HD, Peeples KN, Raman VT, et al. Videolaryngoscopy versus fiber-optic intubation through a supraglottic airway in children with a difficult airway: An analysis from the multicenter pediatric difficult intubation registry. Anesthesiology 2017;127:432-40.  Back to cited text no. 4
    
5.
Pelser A, de Bowles PF, Loock JW. Conservative management of subglottic stenosis with home based tracheostomy care: A retrospective review of 28 patients. Int J Pediatr Otorhinolaryngol 2019;116:196-8.  Back to cited text no. 5
    
6.
Uzun S, Arun O, Ocal T. Unexpected subglottic stenosis in a Newborn in anesthesia induction. Anesteziol Derg 2014;22:223-5.  Back to cited text no. 6
    
7.
Myer CM 3rd, O'Connor DM, Cotton RT. Proposed grading system for subglottic stenosis based on endotracheal tube sizes. Ann Otol Rhinol Laryngol 1994;103:319-23.  Back to cited text no. 7
    
8.
Kerai S, Gupta A, Kohli JK, Sharma J, Sood R. Complicated airway management in a neonate of congenital trachea-oesophageal fistula with subglottic stenosis. Indian J Anaesth 2016;60:142-3.  Back to cited text no. 8
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9.
Obara S, O'Leary JD. Congenital tracheal stenosis: Unanticipated and anticipated difficult airway management in a neonate. A A Case Rep 2014;3:120-2.  Back to cited text no. 9
    
10.
Fiadjoe JE, Nishisaki A, Jagannathan N, Hunyady AI, Greenberg RS, Reynolds PI, et al. Airway management complications in children with difficult tracheal intubation from the Pediatric Difficult Intubation (PeDI) registry: A prospective cohort analysis. Lancet Respir Med 2016;4:37-48.  Back to cited text no. 10
    
11.
Jagannathan N, Sohn L, Fiadjoe JE. Paediatric difficult airway management: What every anaesthetist should know! Br J Anaesth 2016;117 Suppl 1:i3-5.  Back to cited text no. 11
    
12.
Hautefort C, Teissier N, Viala P, Van Den Abbeele T. Balloon dilation laryngoplasty for subglottic stenosis in children: Eight years' experience. Arch Otolaryngol Head Neck Surg 2012;138:235-40.  Back to cited text no. 12
    
13.
Oh SK, Park KN, Lee SW. Long-term results of endoscopic dilatation for tracheal and subglottic stenosis. Clin Exp Otorhinolaryngol 2014;7:324-8.  Back to cited text no. 13
    
14.
Okamoto M, Nishijima E, Yokoi A, Nakao M, Bitoh Y, Arai H. Strategy for surgical treatment of congenital subglottic stenosis in children. Pediatr Surg Int 2012;28:1115-8.  Back to cited text no. 14
    


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