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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 11  |  Issue : 1  |  Page : 24-27

Acquired subglottic stenosis in a child: A diagnostic dilemma


Department of ENT, Head and Neck Surgery, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India

Date of Submission12-Sep-2020
Date of Acceptance10-May-2021
Date of Web Publication5-Jul-2021

Correspondence Address:
Prasanna Kumar Saravanam
Department of ENT, Head and Neck Surgery, Sri Ramchandra Institute of Higher Education and Research, Porur, Chennai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jlv.jlv_12_20

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   Abstract 


The most common benign cause of laryngotracheal stenosis is postintubation injury caused by mechanical ventilation in intensive care units. The incidence of postintubation laryngotracheal stenosis in pediatric population is found to be 11.3%. In this report, we present a case of Soft Subglottic Stenosis(SGS) following recurrent intubation in a 3 year old child. Elective tracheostomy and serial dilatation with tracheal intubation stylets was performed which resulted in significant improvement of airway and the child was successfully decannulated. This case report highlights the importance of high index of suspicion in diagnosis of postintubation SGS in a child and reviews the literature in regard to challenges that are faced during evaluation and management of children presenting with stridor.A

Keywords: Benign airway disease, decannulation, postintubation injury, subglottic stenosis


How to cite this article:
Saravanam PK, Eswaran S, Zaffrullah NS. Acquired subglottic stenosis in a child: A diagnostic dilemma. J Laryngol Voice 2021;11:24-7

How to cite this URL:
Saravanam PK, Eswaran S, Zaffrullah NS. Acquired subglottic stenosis in a child: A diagnostic dilemma. J Laryngol Voice [serial online] 2021 [cited 2021 Dec 3];11:24-7. Available from: https://www.laryngologyandvoice.org/text.asp?2021/11/1/24/320558




   Introduction Top


The most common cause of subglottic stenosis (SGS) is postintubation injury. Injury is produced by translaryngeal intubation or tracheostomy. The incidence of postintubation laryngotracheal stenosis in pediatric population is found to be 11.3%.[1] Higher reported prevalence of the disease is due to greater patient survival in recent studies. The SGS due to prolonged endotracheal intubation is characterized by narrowing of the laryngeal lumen below the level of vocal folds. It is the narrowest and least compliant region in the infant airway as it is surrounded by cartilage. The presence of endotracheal tube for prolonged duration results in edema, ulceration, and necrosis followed by stenosis of the subglottis.[1] The risk factors include the size of the endotracheal tube, length of intubation period, period of mechanical ventilation, number of intubations, and skills of those performing the intubation.[2]

The clinical manifestation of laryngotracheal stenosis depends on the severity of the stenosis. The time period between the extubation and development of stridor depends on the duration of endotracheal intubation and is more common in children who are intubated for a period of more than 72 hours. Children with Grade 1 and Grade 2 stenosis remain asymptomatic and may present with respiratory distress during episodes of upper airway infection. Children with Grade 3 and Grade 4 stenosis present with stridor 7–10 days after extubation.[1]


   Case Report Top


History

A 3-year-old female, developmentally normal child presented to our emergency with difficulty in breathing and increased work of breathing for 1 day associated with cold and cough for 1 week. On initial assessment, the child was tachypneic; saturation at room air was 80%; she was started on humidified oxygen, nebulization, and intravenous steroids; and she was shifted to pediatric intensive care unit (PICU). The child had a past history of endotracheal intubation 1 month back in view of Salmonella Typhi infection with sepsis with dyselectrolytemia for a period of 12 days and was discharged. On admission, evaluation was done in view of biphasic stridor, computed tomography neck was done which showed collapse of the upper lobe of the right lung, and a flexible bronchoscopy was done by a pulmonologist which showed a dynamic anteroposterior narrowing of the trachea. The child was started on triamcinolone. During the period of admission, the child developed worsening of respiratory symptoms and was intubated for a duration of 3 days and later extubated after lung signs regressed, and the child remained asymptomatic and was discharged after a week of observation in the hospital.

The child again presented with acute onset of respiratory distress after a period of 2 weeks and had to be reintubated in the PICU on an emergent basis. At this time, a flexible laryngotracheoscopy was done at the time of extubation and the laryngotracheal airway was found to be adequate with no edema or granulation [Figure 1] and [Figure 2]. After discharge, the child was comfortable for 2 weeks and later again presented with respiratory distress.
Figure 1: Flexible laryngotracheoscopy view – Subglottis of the child at the time of second extubation – No evidence of airway narrowing or edema

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Figure 2: Postextubation – Day 1 – Child is comfortable and doing normal activity in the pediatric intensive care unit

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The third episode of respiratory distress resulted in reintubation. Hence, a tracheostomy was done and upper airway was evaluated by a direct laryngoscopy which showed a patent suprastomal airway. However, the child was managed conservatively with tracheostomy tube and reevaluation by direct laryngoscopy was done after 3 weeks which showed a Grade 2 concentric SGS [Figure 3].
Figure 3: Two weeks posttracheostomy – Direct laryngoscopy showing Grade 2 concentric subglottic stenosis

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The child underwent sequent serial dilatation with tracheal intubation stylets thrice at 2-month interval in view of soft SGS. After 6 months, direct laryngoscopy was done and the airway was adequate and satisfactory [Figure 4], following which the child was decannulated. Postdecannulation, the child is stable and is on regular follow-up for 1 year with no further episodes of respiratory distress.
Figure 4: Prior to decannulation – After serial dilatation

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   Discussion Top


SGS is a congenital or acquired narrowing of the subglottic airway. With the advancement in neonatal critical care, acquired SGS is the most common acquired anomaly of the larynx in children. It is the most common abnormality requiring tracheostomy in children younger than 1 year. It occurs when the cricoid ring is injured by the endotracheal tube or instrument introduced through the larynx or inhaled corrosive or toxic vapors.[2] Risk factors of developing SGS in intubated patients include prolonged intubation (most common), size of endotracheal tube, increased motion of tube, repeated intubations, birth weight <1500 g, infection, gastroesophageal reflux disease, systemic illness like malnutrition, anemia, and hypoxia. Early management of postintubation injury is necessary to prevent the formation of cicatricial stenosis.

Myer et al. devised a classification scheme from I to IV for grading circumferential stenosis – Grade I: <50% luminal obstruction, Grade II: 50%–70% luminal obstruction, Grade III: 71%–99% luminal obstruction, and Grade IV: >99%.[3] The gold standard method of diagnosis of the laryngotracheal stenosis is visualization through a direct laryngoscope or fiber-optic laryngobronchoscope under general anesthesia. However, studies done by Veder et al. showed that awake flexible laryngoscopy at the time of extubation can identify laryngeal pathology and a direct laryngoscopy under general anesthesia is more reliable to diagnose the laryngeal pathology. In this case, a flexible laryngoscopy was done initially after the second time of intubation which showed an adequate airway, free of edema and granulation tissue.

The diagnostic fallacy of a flexible laryngoscopy at the time of intubation could be because endotracheal tube itself acts as a stent and shows a normal patent airway without scar or edema. A direct laryngoscopy was done 2 weeks after tracheostomy which revealed Grade 2 circumferential SGS. The SGS was a soft stenosis which manifests 2–3 weeks after extubation and was diagnosed early. This is the evidence of the fact that in some cases, edema and airway narrowing occur 2–3 weeks after an initial intubation injury and endoscopic evaluation just prior to extubation may not show the true picture. Hence, a high index of suspicion and prophylactic medical treatment should be given to avoid the stenosis.

Initially, medical management was done with triamcinolone nebulization. Triamcinolone is known to modulate wound healing to prevent the occurrence of SGS. They act on all three phases of wound healing and have a definitive role in modulation of wound healing. Studies done by Hirshoren and Eliashar summarized the role of drugs that modulate the wound healing to prevent the occurrence of SGS.[4] Studies done by Prasanna Kumar et al. in rabbits had shown that triamcinolone acetonide is a better modulator of wound healing in postintubation SGS.[5] In this case of soft SGS, initial treatment with triamcinolone nebulization showed symptomatic improvement.

Various etiological factors have been identified to cause postintubation SGS of which an oversized endotracheal tube, insufficiently anesthetized patient, presence of undiagnosed congenital airway narrowing, and faulty intubation techniques are the possible factors for a traumatic intubation. Even after a short period of intubation, the presence of mucosal tears, hematomas, and arytenoid luxation can lead to extubation failure. The endotracheal tube may exert its maximal pressure against a small cricoid ring, leading to concentric subglottic ulcerations and annular ulcerations over the posterior laryngeal commissure. The acute intubation lesions will evolve into a stenotic cicatricial laryngeal sequelae in 2 to 3 weeks, affecting the patient's quality of life.[6]

The child underwent serial dilatation of the SGS over a period of 6 months which resulted in adequate airway. The management of laryngotracheal stenosis includes various procedures which includes open surgical procedures, tracheotomy, and minimally invasive procedures like balloon dilatation. Study done by Hautefort et al on evaluation of the outcomes of balloon dilation laryngoplasty for subglottic stenosis showed that it is efficient and minimally invasive in comparison to open surgical procedure.[7] However, open surgical procedures is the preferred line of management in severe stenosis and failed cases. Study done by Filiz et al on long term outcomes of balloon dilatation for acquired stenosis showed that there was no recurrence and it was the preferred treatment for acquired subglottic stenosis.[8]

In this case, the child was decannulated after serial dilation for a period of 6 months as the child had an adequate and satisfactory airway. Thus, the management of soft SGS requires early diagnosis and minimally invasive procedure like dilatation. The child is on regular follow-up and is doing well. The authors have decided to report this case as prompt diagnosis of postintubation stenosis and minimally invasive surgical procedures will improve the airway and a tracheostomy-dependent lifestyle can be avoided.


   Conclusion Top


  • Postintubation SGS manifests 2–3 weeks after extubation
  • Immediate laryngoscopy and tracheoscopy may not give a true picture as the endotracheal tube will stent the airway and it takes time for the edema and cicatrization to develop
  • Early diagnosis with medical and minimally invasive surgical technique will help in successful management of postintubation stenosis.


Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Schweiger C, Marostica PJ, Smith MM, Manica D, Carvalho PR, Kuhl G. Incidence of post-intubation subglottic stenosis in children: Prospective study. J Laryngol Otol 2013;127:399-403.  Back to cited text no. 1
    
2.
Pashley NR. Risk factors and the prediction of outcome in acquired subglottic stenosis in children. Int J Pediatr Otorhinolaryngol 1982;4:1-6.  Back to cited text no. 2
    
3.
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. 3
    
4.
Hirshoren N, Eliashar R. Wound-healing modulation in upper airway stenosis-Myths and facts. Head Neck 2009;31:111-26.  Back to cited text no. 4
    
5.
Prasanna Kumar S, Ravikumar A, Thanka J. Role of topical medication in prevention of post-extubation subglottic stenosis. Indian J Otolaryngol Head Neck Surg 2017;69:401-8.  Back to cited text no. 5
    
6.
Adriaansen FC, Verwoerd-Verhoef HL, van der Heul RO, Verwoerd CD. Differential effects of endolaryngeal trauma upon the growth of the subglottis. Int J Pediatr Otorhinolaryngol 1988;15:163-71.  Back to cited text no. 6
    
7.
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. 7
    
8.
Filiz A, Ulualp SO. Long-term outcomes of balloon dilation for acquired subglottic stenosis in children. Case Rep Otolaryngol 2014;2014:1-4. [doi: 10.1155/2014/304593].  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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