Abstract
Subglottic stenosis is narrowing of the area in between the vocal cords and the cricoid cartilage. It can cause respiratory problems which can be mild to severe. Study the management of paediatric airway in post-intubation subglottic stenosis associated with Japanese encephalitis. Case Setting and Design: Retrospective study of 18 patients presenting with encephalitis out of which 6 cases were of post-intubation subglottic stenosis associated with Japanese encephalitis between the study period of one year (September 2017–September 2018) in the, Department of Otolaryngology and Head and Neck Surgery, North Eastern Indira Gandhi Regional Institute Of Health And Medical Sciences, Shillong, Meghalaya, India. Fifteen patients were treated in the study period. Different approaches were used. Post-intubation subglottic stenosis depends on the duration of intubation and the quality of life post-tracheostomy depends on the type of tube used. Bronchoscopic assisted procedures during the tracheostomy or after an interval helps to decrease the morbidity. Management of paediatric airway in post-intubation subglottic stenosis associated with Japanese encephalitis needs proper strategy and dedicated and intensive post-operative care. Bivona® silicone tracheostomy tube is a better option.
Keywords: Paediatric airway, Airway management paediatric, Japanese encephalitis
Introduction
The subglottis is a part of the larynx that extends from the bottom of the true vocal cords downwards to the inferior border of the cricoid cartilage. Subglottic stenosis is narrowing of the area in between the vocal cords and the cricoid cartilage. It can cause respiratory problems which can be mild to severe. Cause can be congenital or acquired (trauma, infections, non-infectious, neoplastic, and iatrogenic) [1]. The most common cause of tracheal stenosis is endotracheal intubation [2, 3]. Time from tracheal stenosis development to first intervention is important. 90% of patients who had intervention within 6 months of extubation had a positive outcome compared to 61% of those with a longer delay before intervention [4]. Endoscopic procedures can be considered in simple stenosis, as a bridge to definitive surgical intervention or for patients deemed at high risk for surgical intervention [1]. Although the traditional approach is to use rigid bronchoscopy for endotracheal intervention, other centers have described using flexible bronchoscopy to intubate, dilate, and stent patients with tracheal stenosis without complication [5]. Japanese encephalitis is an infectious disease, spread by mosquitoes and associated with headache, fever, vomiting, confusion, seizures etc. Treatment is generally supportive however in some of the cases intubation has to be done. These cases are mostly paediatric. The management of paediatric airway in post-intubation subglottic stenosis associated with Japanese encephalitis is challenging and needs proper strategy and dedicated and intensive post-operative care. Tracheostomy assisted with bronchoscopic dilatation of the stenosis and further reconstruction after an interval is presently the procedure of choice. This study has tried to evaluate the different challenges and presented a better way to deal with these challenges.
Aim
Study the management of paediatric airway in post-intubation subglottic stenosis associated with Japanese encephalitis.
Inclusion Criteria
Post-intubation subglottic stenosis associated with Japanese encephalitis.
Exclusion Criteria
Post-intubation subglottic stenosis due to other causes.
Materials and Methods
This is a retrospective study of 18 patients presenting with encephalitis out of which 6 cases were of post-intubation subglottic stenosis associated with Japanese encephalitis. Study period is of one year (September 2017–September 2018) done in the, Department of Otolaryngology and Head and Neck Surgery and Department of Paediatrics, North Eastern Indira Gandhi Regional Institute of Health And Medical Sciences, Shillong, Meghalaya, India. Different tracheostomy tubes were used and the dilatation of the stenosis were done with sequential France made of size 12–20 Porges (Fig. 5) under videobronchoscopic guidance. Post-operative management was initially in an Paediatric Intensive Care Unit and then in paediatric ward. The patients regained consciousness and orientation on different days. Silicone tracheostomy tube was used in one of the case.
Fig. 5.
France made bougies of size 12–20 porges
Results
Epidemiology
A total of 18 cases were intubated for encephalitis out of which 5 expired, 6 were tracheostomised, 3 left against medical advice and rest were extubated safely.
Region Wise Distibution
West Khasi Hills, Meghalaya and Papumpare, Arunachal Pradesh were the most affected regions with 4 cases each (Table 1). West Jaintia Hills, Meghalaya, East Khasi Hills, Meghalaya Upper Subansiri, Arunachal Pradesh and Hailakandi, Assam each reported one case with the lowest.
Table 1.
Region wise distribution
| Place of infection | Number of patients |
|---|---|
| West Jaintia Hills, Meghalaya | 1 |
| East Jaintia Hills, Meghalaya | 2 |
| West Khasi Hills, Meghalaya | 4 |
| East Khasi Hills, Meghalaya | 1 |
| Ri-Bhoi, Meghalaya | 2 |
| Papumpare, Arunachal Pradesh | 4 |
| Upper Subansiri, Arunachal Pradesh | 1 |
| East KamengSeppa, Arunachal Pradesh | 2 |
| Hailakandi, Assam | 1 |
Type of Stenosis
Most common type of stenosis was subglottic (Table 2).
Table 2.
Types of stenosis
| Patient number | Duration of intubation | Stenosis type | Intervention |
|---|---|---|---|
| 1 | 10 days | Subglottic | Tracheostomy and bronchoscopy assisted dilatation of stenosis |
| 2 | 10 days | Subglottic | Tracheostomy and bronchoscopy assisted dilatation of stenosis |
| 3 | 09 days | Subglottic | Tracheostomy and bronchoscopy assisted dilatation of stenosis |
| 4 | 08 days | Subglottic | Tracheostomy and bronchoscopy assisted dilatation of stenosis |
| 5 | 09 days | Subglottic | Tracheostomy and bronchoscopy assisted dilatation of stenosis |
| 6 | 12 days | Subglottic | Tracheostomy and bronchoscopy assisted dilatation of stenosis |
Table 2 shows summary of stenosis location and interventions performed during the study period. The maximum duration of intubation was 12 days. The type of stenosis was subglottic in all the patients. All the patients went for tracheostomy and bronchoscopy assisted dilatation of stenosis.
Bronchoscopy assisted dilatation of stenosis was done with France made bougies of size 12–20 porges (Fig. 5).
All the patients were safely weaned off from ventilators. Resection of stenotic segments and reconstruction was planned in case long term complications occur.
Post-intubation subglottic stenosis depends on the duration of intubation and the quality of life post-tracheostomy depends on the type of tube used. Bronchoscopic assisted procedures during the tracheostomy or after an interval helps to decrease the morbidity. Silicone tracheostomy tube was used in one of the case and it was seen that it helped the patient in a speedy recovery as compared to other types of tracheostomy tubes. It was also seen that an early tracheostomy with dilatation of the stenosis was associated with better outcome (Figs. 1, 2, 3, 4, 5 and 6). Two of the patients required anterior cricoid split and laryngotracheoplasty and their recovery was successful.
Fig. 1.
Patient is intubated in paediatric intensive care unit
Fig. 2.
Paediatric tracheostomy tube—Bivona size 3.5 cuffed
Fig. 3.
Paediatric tracheostomy tube—Bivona size 3.5 cuffed
Fig. 4.
Subglottic stenosis pre-dilatation
Fig. 6.
Subglottic stenosis post-dilatation
Bronchoscopy guided treatment proved a valid therapeutic option along with tracheostomy. We have been successful in weaning the patients from ventilators and providing a better quality of life.
Discussion
Bedside Flexible Fibreoptic Laryngoscopy, performed without sedation in the postextubation period in a Paediatric Intensive Care Unit setting, is useful, safe, and accurate for the evaluation of laryngeal lesions caused by intubation (including those in the subglottic region) [6–8]. According to Manica et al. this modality has 93.7% sensitivity and an Negative Predictive Value of 98.8% when used as a screening method for Subglottic stenosis in this setting [7]. Subglottic tracheal stenosis in childhood can be of ‘hard’ and ‘soft’ types occurring typically as primary lesions and stenosis caused by trauma (following intubation or faulty tracheostomy) as the most common secondary lesions. The subepithelial layers play a leading role in the origin of tracheal stenosis [9]. Involvement of the entire thickness of the tracheal wall results in more severe stenosis. Patients with smaller airways, especially infants and children, have a higher incidence of complications. They are more susceptible to upper airway obstruction secondary to glottic oedema and subglottic stenosis. Whited reported that intubation for 7 days or less had a low incidence of sequelae, whereas 10 days or more resulted in a high incidence of sequelae [10]. Cuffed tube usage for prolonged intubation and artificial ventilation substantially increases the rate of tracheal and laryngeal injury. Cuff pressure above 25–35 mm Hg further adds to the risk by compressing tracheal capillaries, which predispose to ischaemic mucosal damage [11]. Depending on the severity of the degeneration and subsequent fibrosis, tracheal stenosis may become symptomatic immediately after extubation, or may present at a later stage with difficulty in breathing [12]. The treatment strategy for laryngo-tracheal stenosis in children has for a long time been conservative with tracheostomy or bougination with the hope of a spontaneous resolution of stenosis with the growth of child. Repeated dilatations are done using rigid bronchoscopes (a set of paediatric 3.5–6 mm and adult 7–9 mm), with or without tracheal stenting [13].
Conclusion
Management of paediatric airway in post-intubation subglottic stenosis associated with Japanese encephalitis needs proper strategy and dedicated and intensive post-operative care. Treatment depends on the location, extent and shape of the stenosis, degree of airway lumen narrowing, etiology, and impact on patient’s functional status. Bronchoscopy guided treatment should be considered as a valid therapeutic option along with tracheostomy. Bivona® silicone tracheostomy tube is a better option. We have been successful in weaning the patients from ventilators and providing a better quality of life.
Footnotes
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
References
- 1.Raman T, Chatterjee K, Alzghoul BN, Innabi AA, Tulunay O, Bartter T, Meena NK. A bronchoscopic approach to benign subglottic stenosis. SAGE Open Med Case Rep. 2017;1(5):1–9. doi: 10.1177/2050313X17713151. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 2.Bacon JL, Patterson CM, Madden BP. Indications and interventional options for non-resectable tracheal stenosis. J Thorac Dis. 2014;6(3):258–270. doi: 10.3978/j.issn.2072-1439.2013.11.08. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 3.Jabbardarjani HR, Kiani A, Arab A, et al. Circumferential calcified tracheal stenosis. J Bronchology Interv Pulmonol. 2009;16(4):305–307. doi: 10.1097/LBR.0b013e3181bdbc61. [DOI] [PubMed] [Google Scholar]
- 4.Nouraei SA, Singh A, Patel A, et al. Early endoscopic treatment of acute inflammatory airway lesions improves the outcome of post intubation airway stenosis. Laryngoscope. 2006;116(8):1417–1421. doi: 10.1097/01.mlg.0000225377.33945.14. [DOI] [PubMed] [Google Scholar]
- 5.Li WT, Xiao YB, Liu GN, et al. Management of benign tracheal stenosis by intubation dilatation under flexible bronchoscopic guidance. Zhonghua Yi Xue Za Zhi. 2011;91:2995–2998. [PubMed] [Google Scholar]
- 6.Smith MM, Kuhl G, Carvalho PR, Marostica PJ. Flexible fiberoptic laryngoscopy in the first hours after extubation for the evaluation of laryngeal lesions due to intubation in the pediatric intensive care unit. Int J Pediatr Otorhinolaryngol. 2007;71:1423–1428. doi: 10.1016/j.ijporl.2007.05.016. [DOI] [PubMed] [Google Scholar]
- 7.Manica D, Schweiger C, Marostica PJ, Kuhl G, Carvalho PR. Association between length of intubation and subglottic stenosis in children. Laryngoscope. 2013;123:1049–1054. doi: 10.1002/lary.23771. [DOI] [PubMed] [Google Scholar]
- 8.Grundfast KM, Camilon FS, Jr, Pransky S, Barber CS, Fink R. Prospective study of subglottic stenosis in intubated neonates. Ann Otol Rhinol Laryngol. 1990;99:390–395. doi: 10.1177/000348949009900513. [DOI] [PubMed] [Google Scholar]
- 9.Minnigerode B, Richter HG. Pathophysiology of Subglottic tracheal stenosis in childhood. Prog Paediatr Surg. 1987;21:1–7. doi: 10.1007/978-3-642-71665-2_1. [DOI] [PubMed] [Google Scholar]
- 10.Whited RE. A prospective study of laryngotrachealsequelae in long-term intubation. Laryngoscope. 1984;94:367–377. doi: 10.1288/00005537-198403000-00014. [DOI] [PubMed] [Google Scholar]
- 11.Cooper JD, Grillo HC. Analysis of problems related to cuffs on intratracheal tubes. Chest. 1972;62:21S. doi: 10.1378/chest.62.2_Supplement.21S. [DOI] [PubMed] [Google Scholar]
- 12.Grillo HC. Congenital lesions, neoplasms and injuries of the trachea. In: Sabiston DC, Spencer FC, editors. Gibbon’s surgery of the chest. Philadelphia: W.B Saunders Co; 1990. pp. 335–371. [Google Scholar]
- 13.Phillips MJ. Stenting therapy for stenosis airway diseases. Respirology. 1998;3:215–219. doi: 10.1111/j.1440-1843.1998.tb00125.x. [DOI] [PubMed] [Google Scholar]