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Idiopathic subglottic stenosis describes subglottic stenosis where no inflammatory, traumatic, iatrogenic or other causative aetiology can be identified. The present study aimed to outline our institution's experience of patients diagnosed with idiopathic subglottic stenosis and describe a very rarely reported familial association.
A retrospective review was conducted of prospectively maintained medical records from 2011 to 2020. Patient clinical, radiological and intra-operative data were reviewed to assess for defined endpoints.
Ten patients with idiopathic subglottic stenosis were identified in this series. One familial pairing was identified, with two sisters presenting with the condition. Successful treatment with carbon dioxide laser and dilatation was achieved in most cases.
Idiopathic subglottic stenosis represents a rare, clinically challenging pathology. Management with endoscopic laser and balloon dilatation is an effective treatment. This paper highlights a very rare familial association, and describes our experience in treating idiopathic subglottic stenosis.
A 43-year-old woman presented with a 3-week history of globus sensation and malaise. A computed tomography scan of her neck showed a large right paratracheal abscess secondary to an infected tracheal diverticulum. The patient was admitted under the ENT surgical team, and underwent incision and drainage of the abscess. There were no post-operative complications and she was discharged home after 2 days, on oral antibiotics.
This case demonstrates that a tracheal diverticulum may become infected and present as a cervical abscess. To our knowledge, this is the fourth reported case in the international literature of abscess formation related to an infected tracheal diverticulum.
Post-tonsillectomy haemorrhage remains a significant complication despite modifications of technique and instrumentation. Intracapsular tonsillectomy spares the capsule as a protective barrier for underlying blood vessels and musculature. Its efficacy in children with sleep-disordered breathing has been established, along with lowered rates of haemorrhage and pain, but research pertaining to adults and for recurrent infections has been limited.
This retrospective study, encompassing 730 patients, compared post-operative haemorrhage rates between extracapsular (n = 379) and intracapsular tonsillectomy (n = 351) across all ages and indications using CoblationTM technology.
A significant difference in post-operative haemorrhage rate was observed between extracapsular and intracapsular tonsillectomy techniques (2.1 vs 0.3 per cent; p = 0.025). In addition, an age of 18 years or older was also found to be an independent risk factor for post-operative haemorrhage (p = 0.01).
CoblationTM intracapsular tonsillectomy was shown to be safe and effective across all ages and indications, with a low risk of bleeding and revision surgery.
Suprastomal granulation tissue is a common complication of long-term tracheostomy. It may be associated with bleeding, aphonia, airway obstruction and delayed decannulation.
This study describes the experience of a tertiary paediatric medical centre with CoblationTM-assisted suprastomal granulation tissue excision.
Thirteen children (mean age, 5.7 years) who underwent the procedure from 2013 to 2019 because of delayed decannulation or aphonia were included. Lumen obstruction ranged from 50 to 90 per cent, with a mean of 68.8 per cent. After the procedure, decannulation was successfully performed in 7 patients, and voice quality improved in 10 patients. There were no peri- or post-operative complications.
This is the largest series to date that describes Coblation used for the treatment of suprastomal granuloma. Coblation has advantages of high precision, relatively low temperature (thereby avoiding thermal injury to adjacent tissue), haemostatic resection and feasibility for use for even large granulomas. The promising results should prompt further studies in larger samples.
Recurrent respiratory papillomatosis is a potentially life-threatening condition characterised by the growth of exophytic lesions within the larynx and trachea. The principal aim of management is maintenance of an adequate airway by surgical debulking. Several adjuvant therapies have been used to varying effect to reduce the burden of this disease and increase the interval between debulking procedures. The most severe cases present in children aged under three years, who are therefore most likely to need adjuvant therapies. The current evidence base on adjuvant treatments relating to children who present aged under three years has been reviewed.
A literature review of articles in Cochrane, PubMed and Embase databases was carried out. Given the rarity of the condition in this age group, all the literature relates to case reports and case series.
Results and conclusion
The following adjuvant therapies have been used in children who presented under three years of age: quadrivalent human papilloma virus vaccine, intralesional cidofovir, pegylated interferon, alpha-interferon, cimetidine and cetuximab.
Total rhinectomy is an invasive procedure that significantly impairs the intranasal turbulence, humidification and heating of inspired air. The use of uvulopalatopharyngoplasty for the treatment of sleep-disordered breathing disorders such as primary snoring and obstructive sleep apnoea has diminished over the past years because of the emergence of less invasive procedures and alternative therapeutic options. This clinical record presents the treatment of a long-term side effect of total rhinectomy using uvulopalatopharyngoplasty.
In 1997, a 62-year-old male underwent total rhinectomy for a nasal schwannoma, followed by rehabilitation with a nasal prosthesis. Twenty-one years later, he presented with severe complaints of nasal blockage and breathing difficulties during both daytime and night-time. Clinical examination revealed no major anomalies besides significant velopharyngeal narrowing. Thus, in 2019, uvulopalatopharyngoplasty was performed to re-establish velopharyngeal patency. Hereafter, the symptoms of nasal blockage disappeared, resulting in an improved quality of life.
Uvulopalatopharyngoplasty may prove useful to treat selected patients with daytime breathing difficulties due to velopharyngeal narrowing.
The incidence of airway obstruction in patients with complex CHD other than vascular rings and absent pulmonary valve syndrome is unknown. We reviewed pre-operative CT and clinical data of children with conotruncal abnormalities to assess for airway obstruction. Airway obstruction was common (41% of patients), often moderate to severe, of diverse aetiology, and most commonly associated with a right aortic arch. Patients with airway obstruction showed a trend towards a higher mortality rate. Patients with complex conotruncal abnormalities should be assessed for airway obstruction as it may help predict the need for additional interventions and assist with prognostication.
To describe the utility of sleep nasendoscopy in determining the level of upper airway obstruction compared to microlaryngotracheobronchoscopy.
A retrospective observational study was conducted at a tertiary level paediatric hospital. Patients clinically diagnosed with upper airway obstruction warranting surgical intervention (i.e. with obstructive sleep apnoea or laryngomalacia) were included. These patients underwent sleep nasendoscopy in the anaesthetic room; microlaryngotracheobronchoscopy was subsequently performed and findings were compared.
Twenty-seven patients were included in the study. Sleep nasendoscopy was able to induce stridor or stertor, and to detect obstruction at the level of palate and pharynx, including tongue base collapse, that was not observed with microlaryngotracheobronchoscopy. Only 47 per cent of patients who had prolapse or indrawing of arytenoids on sleep nasendoscopy had similar findings on microlaryngotracheobronchoscopy. However, microlaryngotracheobronchoscopy was better in diagnosing shortened aryepiglottic folds.
This study demonstrates the utility of sleep nasendoscopy in determining the level and severity of obstruction by mimicking physiological sleep dynamics of the upper airway.
In this chapter discusses the relevant perioperative anesthetic concerns related to thyroidectomy surgery. Reviewed is Graves’ disease, electrolyte and anatomic considerations of thyroid surgery as well as the timeframe and pathophysiology in relation to surgery.
The use of three-dimensional printing has been rapidly expanding over the last several decades. Virtual surgical three-dimensional simulation and planning has been shown to increase efficiency and accuracy in various clinical scenarios.
To report the feasibility of three-dimensional printing in paediatric laryngotracheal stenosis and discuss potential applications of three-dimensional printed models in airway surgery.
Retrospective case series in a tertiary care aerodigestive centre.
Three-dimensional printing was undertaken in two cases of paediatric laryngotracheal stenosis. One patient with grade 4 subglottic stenosis with posterior glottic involvement underwent an extended partial cricotracheal reconstruction. Another patient with grade 4 tracheal stenosis underwent tracheal resection and end-to-end anastomosis. Models of both tracheas were printed using PolyJet technology from a Stratasys Connex2 printer.
It is feasible to demonstrate stenosis in three-dimensional printed models, allowing for patient-specific pre-operative surgical simulation. The models serve as an educational tool for patients’ understanding of the surgery, and for teaching residents and fellows.
To present clinical experience and surgical outcomes of end-to-end anastomosis in the management of laryngotracheal stenosis and tracheal defects following invasive thyroid malignancy resection.
A retrospective analysis was performed of 14 patients with laryngotracheal stenosis and tracheal invasive thyroid malignancy. All patients underwent tracheal or cricotracheal resection and primary end-to-end anastomosis.
Length of stenosis was 1.7–4 cm. Stenosis was classified as Myer and Cotton grade II in 4 patients, grade III in 6 and grade IV in 2. Surgical procedures included tracheotracheal end-to-end anastomosis (n = 4), cricotracheal anastomosis (n = 2) and thyrotracheal anastomosis (n = 6). Patients with invasive thyroid malignancy underwent segmental resection of the involved segment with tumour-free margins, and tracheal or cricotracheal end-to-end anastomosis. Successful decannulation was achieved in 13 patients (93 per cent). Post-operative complications were: wound infection (n = 1), subcutaneous emphysema (n = 1), temporary unilateral vocal fold palsy (n = 1), granulation tissue development (n = 1), and restenosis (n = 2).
End-to-end anastomosis can be used safely and successfully in the management of advanced laryngotracheal stenosis and wide laryngotracheal defects. Greater success can be achieved using previously described surgical rules and laryngotracheal release manoeuvres.
We present two interesting cases of isolated left subclavian artery from the pulmonary artery with symptoms of upper airway obstruction. The first patient had tetralogy of Fallot, pulmonary artery sling, bilateral superior caval veins, and left bronchial isomerism, suggesting heterotaxy syndrome. The second patient had a right aortic arch, isolated left subclavian artery, and bilateral arterial ducts. These two cases are interesting because of their rarity and uncommon presentation.
Pachyonychia congenita is a rare keratinising disorder, which typically presents during the first three years of life and usually affects the nails and palmoplantar surfaces. It can involve the larynx and potentially result in life-threatening airway obstruction.
A case report is presented and the findings of a literature review are reported. The review involved a PubMed search using the keywords ‘pachyonychia congenita’ together with ‘larynx’, ‘laryngeal involvement’, ‘laryngeal obstruction’, ‘airway obstruction’, ‘hoarseness’ and/or ‘stridor’.
A five-year-old boy, with confirmed pachyonychia congenita, presented with complications of laryngeal involvement over a four-year period. He required three intubations and a tracheostomy for acute airway obstruction. Treatment with potassium titanyl phosphate laser laryngoscopy stabilised the progression of laryngeal disease.
Patients with pachyonychia congenita and laryngeal involvement can have a varied presentation, ranging from hoarseness to acute airway obstruction. Management can be a challenge, requiring early evaluation, regular surveillance and aggressive treatment. This paper reports our experience in managing and treating the laryngeal complications of a child with pachyonychia congenita.
In developing countries with limited access to ENT services, performing emergency cricothyroidotomy in patients with upper airway obstruction may be a life-saving last resort. An established Danish–Zimbabwean collaboration of otorhinolaryngologists enrolled Zimbabwean doctors into a video-guided simulation training programme on emergency cricothyroidotomy. This paper presents the positive effect of this training, illustrated by two case reports.
A 56-year-old female presented with upper airway obstruction due to a rapidly progressing infectious swelling of the head and neck progressing to cardiac arrest. Cardiopulmonary resuscitation was initiated and a secure surgical airway was established via an emergency cricothyroidotomy, saving the patient. A 70-year-old male presented with upper airway obstruction secondary to intubation for an elective procedure. When extubated, the patient exhibited severe stridor followed by respiratory arrest. Re-intubation attempts were unsuccessful and emergency cricothyroidotomy was performed to secure the airway, preserving the life of the patient.
Emergency cricothyroidotomy training should be considered for all surgeons, anaesthetists and, eventually, emergency and recovery room personnel in developing countries. A video-guided simulation training programme on emergency cricothyroidotomy in Zimbabwe proved its value in this regard.
Takotsubo cardiomyopathy has been associated with the use of catecholamines; however, its development after the use of nebulised adrenaline for the management of acute airway obstruction has not previously been described.
A 66-year-old man with squamous cell carcinoma of the larynx, with tumour–node–metastasis staging of T3N2cM0, confirmed by biopsy and computed tomography, presented to the emergency department with acute airway obstruction. He was treated twice with nebulised adrenaline and intravenous dexamethasone. After a period of 24 hours, cardiac rhythm changes were noted on telemetry. A 12-lead electrocardiogram showed widespread T-wave inversion and QT prolongation suggestive of an acute coronary syndrome. Coronary angiography demonstrated no coronary artery disease, but left ventricular angiography showed marked apical ballooning and apical wall akinesia consistent with a diagnosis of takotsubo cardiomyopathy.
Takotsubo cardiomyopathy can mimic true ischaemic heart disease and the diagnosis requires a high index of suspicion in patients managed with nebulised adrenaline.
This study aimed to develop a functional model of subglottic stenosis by inducing direct airway irritation in transplanted mouse laryngotracheal complexes.
Laryngotracheal complexes from C57BL/6 mice were harvested and divided into three groups: uninjured, mechanically injured and chemically injured. Donor laryngotracheal complexes from each group were placed in dorsal subcutaneous pockets of recipient mice. Each week, the transplanted laryngotracheal complexes were harvested, and tissues were fixed, sectioned, and stained with haematoxylin and eosin. Representative slides were reviewed by a blinded pathologist, to determine the formation of granulation tissue, and graded as to the degree of granulation formation.
Direct airway irritation induced granulation tissue formation under the disrupted epithelium of airway mucosa; this was seen as early as two weeks after chemical injury.
Results indicate that granulation tissue formation in a murine model may be an efficient tool for investigating the development and treatment of subglottic stenosis.
This study aimed to: assess the mucosal alterations of the larynx and hypopharynx typical for mucopolysaccharidoses, in a standardised manner; compare the severity in different subtypes of mucopolysaccharidoses; and monitor the effect of an enzyme replacement therapy.
A classification for mucosal alterations of the larynx and hypopharynx was developed and utilised in 55 patients with mucopolysaccharidoses. Fifteen patients who started treatment with enzyme replacement therapy were followed longitudinally.
The most severe alterations were seen in the posterior region of the larynx and the arytenoids, and in the region of the false vocal folds. The alterations were most severe in patients with mucopolysaccharidosis II. No clear trend was observed in the patients who received enzyme replacement therapy.
Quantification of mucosal alterations of the hypopharynx and larynx in mucopolysaccharidoses patients can provide information about the disease's natural process and about the efficacy of enzyme replacement therapy.
Congenital airway obstruction is rare but potentially fatal. We developed a complex airways interventional delivery team to manage such cases. Antenatal imaging detects airway compromise at an early stage and facilitates the planning of delivery procedures (‘ex utero intrapartum treatment’ and ‘operation on placental support’) which maintain feto-placental circulation whilst an airway is secured.
A retrospective review was performed of cases in which ENT input was required at birth for airway obstruction.
Four neonates were delivered before implementation of the service: two were intubated and another two underwent tracheostomy but died in the peri-natal period. Seven neonates were delivered after implementation of the service: six were intubated and one underwent immediate tracheostomy. Five subsequently underwent tracheostomy (three have since been decannulated). One child with multiple congenital anomalies died due to respiratory failure. Airway obstruction was caused by lymphatic malformation, teratoma, costo-craniomandibular syndrome and choristoma.
In the absence of other anomalies, interventional airway delivery led to reduced mortality and improved outcomes.
This case report describes a patient who suffered an acute, severe complication of unilateral submandibular sialolithiasis, the disease process and management of these patients.
A 70-year-old woman was under investigation for a recurrent, painful right submandibular swelling and subsequently presented with an acute exacerbation. She exhibited symptoms of acute submandibular sialadenitis, and also reported breathing difficulty and a change in voice quality. Computed tomography imaging showed that this was caused by a submandibular gland duct sialolith, with inflammation extending to the ipsilateral supraglottis. She was treated medically and the stone was removed when the inflammation had stabilised.
This case highlights the need to thoroughly assess patients with neck swellings, especially when symptoms are atypical, to avoid life-threatening complications.