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| CASE REPORT |
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| Year : 2021 | Volume
: 8
| Issue : 3 | Page : 157-160 |
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Successful management of acquired pinhole subglottic stenosis by multidisciplinary pediatric airway team
Praveen Khilnani1, Chandrashekhar Singha1, Ankur Ohri1, Rakhee Goyal2, Shandip Kumar Sinha3, Anup Sabharwal4
1 Department of Pediatric Intensive Care, Madhukar Rainbow Children's Hospital, New Delhi, India
2 Department of Pediatric Anesthesiology, Madhukar Rainbow Children's Hospital, New Delhi, India
3 Department of Pediatric Surgery, Madhukar Rainbow Children's Hospital, New Delhi, India
4 Department of Pediatric ENT, Madhukar Rainbow Children's Hospital, New Delhi, India
| Date of Submission | 01-Feb-2021 |
| Date of Decision | 02-Mar-2021 |
| Date of Acceptance | 22-Mar-2021 |
| Date of Web Publication | 21-May-2021 |
Correspondence Address:
Dr. Praveen Khilnani
Madhukar Rainbow Children's, Hospital, FC-29, Plot No. 5, Geetanjali, Near Malviya Nagar, Metro Station, Gate No.1, New Delhi - 110 017
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/jpcc.jpcc_12_21
The concept of neonatal and pediatric airway team is somewhat new in India. : In Western countries, though this concept has been established recently, it remains only limited to children's hospitals. We report the case of a 15-month-old girl with failure to thrive and critical life-threatening severe subglottic stenosis. She was managed in a comprehensive manner by multidisciplinary pediatric airway team at a tertiary-level children's hospital.
Keywords: Airway team, difficult airway, postintubation injury, subglottic stenosis
How to cite this article:
Khilnani P, Singha C, Ohri A, Goyal R, Sinha SK, Sabharwal A. Successful management of acquired pinhole subglottic stenosis by multidisciplinary pediatric airway team. J Pediatr Crit Care 2021;8:157-60 |
How to cite this URL:
Khilnani P, Singha C, Ohri A, Goyal R, Sinha SK, Sabharwal A. Successful management of acquired pinhole subglottic stenosis by multidisciplinary pediatric airway team. J Pediatr Crit Care [serial online] 2021 [cited 2023 Jun 2];8:157-60. Available from: http://www.jpcc.org.in/text.asp?2021/8/3/157/316590 |
| Introduction | |  |
The subglottic area is the narrowest part of the airway in children. Prolonged intubation remains the most common cause of acquired Subglotic stenosis. Clinical presentation is characterized by the onset of varying degrees of dyspnea and stridor in the post-extubation period which can progress in severity to acute life threatening event. Management of such severe subglottic stenosis is challenging and requires multidisciplinary approach. We describe a case of acquired subglottic stenosis that was recently managed successfully by multidisciplinary approach by Pediatric airway team. Existing literature is also reviewed in the discussion section.
| Case Report | | |
A 15-month-old female who had failure to thrive (admission weight of 5 kg) was referred to our hospital after the child had developed life-threatening hypoxia and bradycardia during flexible fiber-optic bronchoscopy, which was done for evaluation of biphasic stridor after fever for 2 days. Upon arrival in the emergency room, she had severe biphasic stridor, nasal flaring, and intercostal and marked subcostal retractions. She was fully conscious, heart rate was 178/min, respiratory rate was 46/min, blood pressure 101/64 mmHg, temperature 99.6°F, and SpO2 76%–77% on room air.
She was born as a low birth weight, small for gestational age baby (birth weight 1.8 kg) with no significant antenatal, intranatal, and postnatal events. She was admitted twice with complaints of fever and stridor in the past in a private hospital. The last admission was at 3 weeks back, where she has required intubation and mechanical ventilation for almost 7 days. After 2 weeks of discharge, the mother noticed slowly progressing respiratory distress and stridor. These symptoms worsened 2 days before this admission.
A arterial blood gas on 2 L/min oxygen by nasal cannula in the pediatric intensive care unit (PICU) revealed pH 7.22, pCO2 55.5 mmHg, paO2 66 mmHg, and HCO3 22.7; complete blood count (CBC) showed hemoglobin of 8 g/dl, total white cell count of 12,000/cmm, 40% polymorphs, and 40% lymphocytes; and her liver function tests and kidney function test were within normal limits. C-reactive protein (CRP) was 10 mg/L other sepsis markers. She was negative for HIV. X-ray chest with neck anterior-posterior had shown no major abnormality except ballooning of hypopharynx seen in the lateral airway radiographs suggestive of upper airway obstruction [Figure 1]. | Figure 1: (a) Anterior-posterior neck and chest radiograph. (b) Lateral neck radiograph revealing ballooning of hypopharynx
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The child was started on low-flow oxygen at 2 L/min and her SpO2 was ranging from 89% to 91%. Further, she was then placed on high-flow nasal cannula with FiO2 60% and flow rate of 10 L/min. SpO2 improved to 95%–96%. The child was started on intravenous (IV) ceftriaxone and oral azithromycin. Nebulization with adrenaline and L-salbutamol was started. She continued to have severe biphasic stridor with respiratory distress. In view of life-threatening critical airway obstruction, ear, nose, and throat (ENT) opinion was sought and an emergent tracheostomy was performed in operating room with 3.5-mm uncuffed tracheostomy tube. A detailed upper and lower airway evaluation by bronchoscopy was deferred to a later date as it was important to ensure adequate airway with tracheostomy to prevent acute life threatening episodes. She developed posttracheostomy pulmonary edema, most likely negative pressure pulmonary edema, and required mechanical ventilation for the next 24 h after which she had shown improvement in respiratory status with tracheostomy in situ. Her echocardiogram revealed no cardiac abnormality. Gradually, she was weaned off oxygen therapy over the next 3–4 days. In the PICU on day 7, the child developed new fever and tachypnea and her tracheal secretions were thick and purulent, suggestive of ventilator-associated event. Her CRP was 185 mg/L; CBC revealed a white count of 23,500/cmm with marked neutrophilia. Antibiotics were upgraded to piperacillin-tazobactam. Tracheal aspirate culture reports 48 h later showed Acinetobacter sensitive to colistin only. She was started on colistin IV. Contrast-enhanced computerized tomography (CT) neck is shown in [Figure 2] with small subglottic stenotic segment with pinhole stenosis of airway. Flexible fiber-optic bronchoscopy was done for evaluation of the upper airways as well as airway beyond the region of stenosis. Bronchoscopy (size 2.8-mm outer diameter) revealed normal nasal airway and vocal cord movements, depicting a pinhole appearance at the laryngeal inlet just below the level of vocal chords [Figure 3]. Bronchoscope was then also passed via tracheostomy and revealed evidence of tracheal inflammation, with normal anatomy of distal airways and purulent secretions suggestive of lower respiratory infection. Bronchoalveolar lavage cultures grew Acinetobacter baumannii sensitive to colistin but were negative for acid-fast bacillus and fungal cultures. She started improving colistin therapy for 10 days as well as with continued supportive management. | Figure 3: Bronchoscopy images revealing clear evidence of subglottic stenosis
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After she was stabilized with supportive treatment, a detailed discussion with family was held by the airway team (pediatric intensivist, ENT specialist, pediatric surgeon, and anesthesiologist) and the plan for further management of subglottic stenosis (SGS) was discussed. A combination of rigid and flexible bronchoscopy for upper airway evaluation was planned to be undertaken in the operating room with a plan to intervene surgically as required by ENT and pediatric surgeon. All the possible options including dilatation of the stenotic segment with or without surgical intervention were discussed and risks and benefits were informed to the parents. In view of fibrosis in the subglottic space, a decision was taken to repair it surgically with cautery or laser and mitomycin and follow it up with balloon dilatation of subglottic space. The anesthesia was administered via tracheostomy tube with adequate airway control. This procedure was followed by a check bronchoscopy using a combination of flexible fiber-optic as well as a rigid bronchoscope [Figure 4]. | Figure 4: (a) Subglottic repair immediate postoperative period (view via a rigid bronchoscope) revealing wider subglottic lumen. (b) Follow-up bronchoscopy 4 months later
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After the procedure, the child was discharged home in stable condition with tracheostomy in situ. Weight at the time of discharge was 6.1 kg. The child was in regular follow-up on an outpatient basis. Plan was to evaluate upper airway in 3–4 weeks with possible balloon dilatation of subglottic stenotic segment if necessary. There was a delay on the part of the family for regular evaluation as the child was doing well at home.
Four months later, she was readmitted with another episode of lower respiratory tract infection. Initial WBC count was 17,560/cu mm with CRP of 97 mg/L. Oxygen saturations were maintained on 2LO2 via tracheostomy. Chest radiograph on the second admission revealed infiltrates on the right side. She was started on piperacillin and tazobactam and admitted for close observation and further progression. Tracheal aspirate culture showed heavy growth of Pseudomonas aeruginosa continued to be treated with piperacillin-tazobactam as per sensitivity report. She improved with supportive management. An expert review by pediatric airway team was called in regarding further management plan for tracheostomy. A flexible fiber-optic bronchoscopy was planned to evaluate the status of subglottic area and plan the way forward to aim toward decannulation. Repeat bronchoscopy showed an improving SGS, and flexible bronchoscope was passed to the level of tracheostomy and beyond revealing a normal distal trachea [Figure 4]b.
Tracheostomy was partially occluded by tape in a gradual way starting with occlusion of one-third of lumen for 6 h followed by 50% occlusion at the proximal end of tracheostomy for 24 h followed by complete occlusion for another 12 h under close monitoring to complete occlusion over 72 h; the child was finally decannulated and she maintained her respiratory effort and saturations well on room air.
| Discussion | | |
This case demonstrates the multidisciplinary approach in a child with failure to thrive and recurrent lower respiratory tract infection requiring mechanical ventilation possibly leading to SGS progressing to critical life-threatening upper airway obstruction.
The subglottic area around the level of cricoid ring is the narrowest part of the airway that is only 4–5 mm wide when compared to the glottis which is the narrowest part in adults. SGS can be congenital (quite rare) or acquired. The most common cause of acquired SGS is prolonged intubation.[1] The presence of an endotracheal tube in this area, along with other factors, may cause edema, ulceration, and necrosis of subglottic structures and may lead to the development of stenosis. Clinical presentation is characterized by the onset of varying degrees of dyspnea and stridor in the postextubation period which can progress in severity. The incidence of SGS in children (median age: 3.1 months; range: 0.3–59.5 months) after prolonged intubation is 11.3%.[2] In a case series, 65 patients[3] with postintubation tracheal stenosis were treated with rigid bronchoscopy with laser resection or stent implantation as the management of choice. There were 53 (81 %) infants and children and 12 (19 %) adult patients. Among pediatric cases, 20 (38 %) were younger than 3 years of age and 39 (36 %) were younger than 6 years of age at the time of surgery. The authors reported that laser resection cured 66% of tracheal stenosis patients, while stents were inserted for the remaining patients. The use of stents in pediatric age group remains controversial.
For every 5 intubation days, there is a 50.3% increase in the risk of developing SGS.[4] Bronchoscopy is the mainstay of diagnosis[5] of postintubation tracheal stenosis. CT will reveal the exact location and extent of the stenosis. Chest X-ray may be performed but rarely detects stenosis due to smaller diameter of the neonatal and pediatric airways. Virtual bronchoscopy done with reconstructed CT images is available to measure the length of stenotic segment and any extraluminal obstruction, however, it tends to miss dynamic obstruction-related measurements and precise nature of intraluminal obstruction with respect to specific mucosal changes in various pathological diagnoses.
With the help of flexible fiber-optic bronchoscopy, the degree and extent of obstruction can also be evaluated and this helps in classifying the degree of stenosis. This is important as it determines the type of treatment options available and its outcome.[4],[5] In this case, in view of fibrosis in the subglottic space, a decision was taken to repair it surgically with cautery or laser and mitomycin and follow it up with balloon dilatation of subglottic space if necessary.
Mitomycin C (MMC) was used in our patient as a local application in the resected raw subglottic area to help with inhibition of fibroblast proliferation and possible reduction of further fibrosis. MMC application has been found to be beneficial in few studies published on surgery for laryngeal stenosis, but long-term effects on reduction of incidence of restenosis have not been studied.[6],[7] The efficacy and safety of balloon bronchoplasty for tracheal stenosis was evaluated in 26 patients by Freitag et al.[8] and reported a success rate of 100% when balloon dilation was used as part of a multimodal approach. Hautefort et al.[9] in their 8-year experience with balloon dilation and laryngoplasty found similar results.
Availability and organization of airway team for neonates and older children is a unique concept.[10] A group of skilled individuals with experience in dealing with critically ill neonates and children is the key to success of such a team. However, at multidisciplinary children's hospitals, such an organization of team should not be very difficult in view of availability of various pediatric-specific superspecialists such as pediatric intensivists, neonatologists, pulmonologists, pediatric anesthesiologists, pediatric cardiologists, pediatric radiologists, pediatric otolaryngologists, and pediatric surgeons (general, plastic, thoracic, and vascular surgeons) in addition to pediatric critical care nurses, trained neonatal staff, and pediatric technical staff for bronchoscopy as well as ready availability of all sizes of neonatal and pediatric airway equipment.
To our knowledge, this is the first-ever report of an organized pediatric airway team from India. We have found it useful to manage cases of difficult airways in complex patients with various syndromes and associated with multiple congenital anomalies.
Diagnosis and management of SGS is complex and requires a multidisciplinary approach. The concept of having a dedicated pediatric airway team to consult and intervene in a timely fashion in such cases can be not only lifesaving but also can be associated with favorable long-term outcome too.
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 | | |
| 1. | Todres ID, Khilnani P. Critical upper airway obstruction in Children. In: Roberts JT, editors. Clinical Management of the Airway. Philadelphia: WB Saunders; 1993. p. 383-97.  |
| 2. | Manica D, Schweiger C, Maróstica PJ, Kuhl G, Carvalho PR. Association between length of intubation and subglottic stenosis in children. Laryngoscope 2013;123:1049-54. |
| 3. | Alshammari J, Monnier P. Airway stenting with the LT-Mold™ for severe glotto-subglottic stenosis or intractable aspiration: Experience in 65 cases. Eur Arch Otorhinolaryngol 2012;269:2531-8. |
| 4. | Ho AM, Mizubuti GB, Dion JM, Beyea JA. Paediatric postintubation subglottic stenosis. Arch Dis Child 2020;105:486. |
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| 7. | Ubell ML, Ettema SL, Toohill RJ, Simpson CB, Merati AL. Mitomycin-c application in airway stenosis surgery: Analysis of safety and costs. Otolaryngol Head Neck Surg 2006;134:403-6. |
| 8. | Freitag L, Ernst A, Unger M, Kovitz K, Marquette CH. A proposed classification system of central airway stenosis. Eur Respir J 2007;30:7-12. |
| 9. | 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. |
| 10. | Dalesio NM, Diaz-Rodriguez N, Koka R, Kudchadkar S, Mark LJ, Cover R, et al. Development of a multidisciplinary pediatric airway program: An institutional experience. Hosp Pediatr 2019;9:468-75. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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