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Fungal laryngotracheitis (FLT) is rare, and the diagnosis can be challenging, as its presentation lacks specificity and may resemble other conditions such as granulomatous disease, gastroesophageal reflux, or malignancy. FLT can be very invasive, causing complete laryngotracheal separation, leading to a non-functioning larynx. We report a 39-year-old Indian woman with diabetes who presented to the emergency department with a sore throat, hoarseness, dysphagia, and stridor for two days. Initially treated for diabetic ketoacidosis due to acute tonsillopharyngitis, she required intubation for airway obstruction and severe metabolic acidosis. Fourteen days post-intubation, an airway assessment revealed bilateral vocal fold edema and pus in the subglottic and cervical trachea. CT imaging showed circumferential fluid around the trachea and a distorted larynx. Examination under anesthesia and neck exploration revealed pus around the thyroid gland and trachea, leading to a tracheostomy and sample collection. Histopathology indicated a fungal infection, confirmed as Candida guillermondii with Escherichia coli . The patient was treated with oral fluconazole and intravenous cefuroxime for four weeks. Despite treatment, a repeat CT indicated a non-functioning larynx, prompting a proposal for a total laryngectomy. After a multidisciplinary discussion, it was decided to continue antifungal therapy due to the patient's clinical improvement. At follow-up a month later, she was stable, tolerating oral intake with a double-lumen tracheostomy tube. This case underscores the importance of a high index of suspicion for FLT and the need for patient-specific decisions regarding total laryngectomy in a non-functioning larynx., Competing Interests: Human subjects: Consent was obtained or waived by all participants in this study. Conflicts of interest: In compliance with the ICMJE uniform disclosure form, all authors declare the following: Payment/services info: All authors have declared that no financial support was received from any organization for the submitted work. Financial relationships: All authors have declared that they have no financial relationships at present or within the previous three years with any organizations that might have an interest in the submitted work. Other relationships: All authors have declared that there are no other relationships or activities that could appear to have influenced the submitted work., (Copyright © 2024, Baharudin et al.)

Competing Interests: None

A 15-year-old girl presented with a 3-year-history of continuous outflow of saliva from a pharyngocutaneous fistula, located at 5 mm superior to her tracheal stoma. She was diagnosed with Miller-Dieker syndrome at birth. At 2 years of age, pediatric surgeons at our institution carried out laryngotracheal separation to prevent aspiration pneumonia. At the age of 12 years, she developed continuous saliva discharge from the fistula. We performed central-part laryngectomy and resection of the pharyngocutaneous fistula, which relieved her from the continuous saliva discharge. Central-part laryngectomy is less invasive and easier to perform than total laryngectomy. We hereby present a case and retrospective analysis of 12 patients, who underwent central-part laryngectomy., Competing Interests: Declaration of Competing Interest All authors declare that there are no conflicts of interest., (Copyright © 2022 The Authors. Published by Elsevier B.V. All rights reserved.)

Background: Owing to recurrent aspiration pneumonia and airway stenosis secondary to thoracic deformities, tracheostomy or laryngotracheal separation are often necessary in children with severe motor and intellectual disabilities. However, these procedures are associated with the risks of tracheal stenosis due to tracheal granulation and tracheoinnominate artery fistula formation. We report a case of a child with severe motor and intellectual disabilities treated with an anterior mediastinal tracheostomy., Case Presentation: The patient was a 15-year-old boy with severe motor and intellectual disabilities due to intractable epilepsy. Due to thoracic deformity and tracheomalacia, the patient had a flattened and narrowed trachea. Accordingly, laryngotracheal separation was performed 4 months before admission to avoid aspiration pneumonia. Due to a common cold, the patient required frequent sputum suctioning, which exacerbated the tracheal stenosis. Bronchoscopy revealed tracheal stenosis 4-5 cm caudal to the tracheostomy site, tracheal mucosal ulcers, and pulsation of the innominate artery on the anterior wall of the trachea. We performed an anterior mediastinum tracheostomy to release the tracheal stenosis and prevent tracheoinnominate artery fistula formation., Conclusions: Anterior mediastinal tracheostomy has several advantages. Including sufficient release of bony compression, release of tracheal hyperextension, and relief of tracheal and innominate artery contact ensures a cannula-free tracheostomy, and there is no need to dissect the brachiocephalic artery. It is the procedure of choice in cases of head and neck malignancies requiring extensive tracheal resection and could be a good surgical option for severe tracheal stenosis and tracheoinnominate artery fistula in children with severe motor and intellectual disabilities., (© 2023. The Author(s).)

A severe Angelman syndrome (AS) patient with a very large deletion (19.3 Mb) at 15q11.2-q14 required laryngotracheal separation, which is not a common surgery in AS. Comparative genomic hybridization-based microarrays can be useful to confirm deletion size and clinical severity., Competing Interests: The authors declare no conflict of interest., (© 2022 The Authors. Clinical Case Reports published by John Wiley & Sons Ltd.)

Animal-induced trauma can lead to severe injury and death, especially in medically isolated settings. Few reports of hyena attacks on humans have been reported in the literature. The goal of this report is to describe such an attack and the heroic efforts required to preserve life and function in a resource-limited environment. A 55-year-old female was attacked by a hyena in a rural region of Ethiopia. Despite delays in medical care, she was able to survive this attack and was successfully discharged after prolonged treatment efforts. Animal-induced trauma is a potential source of substantial and disfiguring injury, especially in resource-limited environments. Early transfer to tertiary care centers and creative solutions are needed to optimize outcomes in such environments., Competing Interests: The authors have declared that no competing interests exist., (Copyright © 2022, Macielak et al.)

Three patients with multiple system atrophy (MSA) who have been hospitalized for aspiration pneumonia underwent aspiration prevention surgery. Laryngeal closure was performed in 2 cases, and laryngotracheal separation was performed in 1 case. Two patients were able to continue oral intake. No recurrence of aspiration pneumonia was observed in all cases after the operation for about two years, and the reduction in the number of aspirations at night improved the patient's QOL and reduced the burden on the caregiver. It was considered that the appropriate time for surgery was when communication in vocal language became difficult. It was a time when the loss of vocal function was well accepted in 3 cases. Aspiration prevention surgery may be a useful treatment option because it may contribute to prolonging the prognosis of life by reducing the complications of respiratory infections.

Introduction: Methyl-CpG binding protein 2 gene (MECP2) is located on the X chromosome (Xq28) and is important for nervous and immune system functioning. Patients with MECP2 duplication syndrome (MDS) have recurrent respiratory infections (RRIs). Although RRIs often occur with MDS because some patients with MDS also have hypoimmunoglobulinemia and duplication of the interleukin-1-receptor-associated kinase-1 gene (IRAK1), which is also located on Xq28, the phenotype of IRAK1 duplication in patients with MDS remains unclear., Methods: The clinical course of three patients with MDS who underwent laryngotracheal separation (LTS) at two institutions was summarized., Results: Three patients with MDS were identified to have recurrent pneumonia characteristic of aspiration pneumonia, sometimes requiring artificial ventilation therapy; they had no other bacterial infections. After LTS, they rarely had pneumonia. In MDS, MECP2 expression increased two-fold naturally, while IRAK-1 expression showed no difference compared with a healthy subject., Conclusions: Since RRIs in MDS are thought to be caused by aspiration and not susceptibility to infection previously estimated to be major complication, the evaluation of aspiration is recommended for RRIs for better management of MDS., (Copyright © 2022 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.)

Complete laryngotracheal separation is a rare life-threatening trauma that may occur due to road traffic accidents. Unfortunately, this injury may be missed if emergency physicians were not aware of its possibility. We present a pediatric patient who had complete laryngotracheal separation and esophageal injury caused by strangulation with a headscarf while riding a four-wheeled motorcycle. The neck on external examination showed only a ligature mark without open wounds. The injury was identified while attempting tracheostomy as the patient had a decreased oxygen saturation and was aphonic. The patient underwent complete airway reconstruction and stenting as well as low tracheostomy with complete reconstruction and anastomosis of the esophagus. The patient was decannulated after a few weeks with the vocal cords in paramedian position and no aspiration. This case demonstrates that early prompt treatment can save the patient's life and result in good functional outcomes.

Background: Surgery to prevent aspiration has complications related to tracheostomy tube, such as the trachea-brachiocephalic artery fistula. Glottic closure procedure makes tracheostoma at a position higher than the first ring of the trachea and theoretically has a potential to prevent such complications owing to a longer distance between the tip of tracheostomy tube and the tracheal membrane adjacent to the brachiocephalic artery. Our aim is to evaluate the safety of glottic closure in neurologically impaired patients by comparing outcomes with laryngotracheal separation., Methods: This study is a single-center retrospective study from 2004 to 2019, using data of 15 and 12 patients who underwent glottic closure (GC) and laryngotracheal separation (LTS). The primary outcome was the incidence of postoperative complications induced by tracheostomy tube placement and adjustment of the tracheostomy tube position to prevent these complications, such as by converting to a length-adjustable tube and/or placing gauze between the skin and tube flange. Additionally, we analyzed the anatomical relationship between the tracheostomy tube tip and brachiocephalic artery and measured the distance between them using postoperative CT images., Results: No patients in either group had trachea-brachiocephalic artery fistula. Erosion or granuloma formation occurred in 1 patient (7%) and 4 patients (33%) in the GC and LTS groups, respectively. Adjustment of the tracheostomy tube was needed in 2 patients (13%) and 6 patients (50%) in the GC and LTS groups. CT revealed a higher proportion of patients with the tracheostomy tube tip superior to the brachiocephalic artery in GC than LTS group. The mean tracheostoma-brachiocephalic artery distance was 40.8 and 32.4 mm in the GC and LTS groups., Conclusions: Glottic closure reduces the risk of postoperative complications related to a tracheostomy tube. This may be due to the higher position of the tracheostoma at the level of the cricoid cartilage, increasing the distance between the tracheostoma and brachiocephalic artery., (© 2022. The Author(s).)

Aim of the Study: Laryngotracheal separation (LTS) is known to be the definitive solution for intractable aspiration pneumonia in neurologically impaired children. Postoperatively, a tracheostomy cannula is usually required. However, there are fatal cannula related complications such as a tracheo-innominate artery fistula (TIAF). We present our methods of preventing TIAF., Methods: A retrospective review in a single center from 2011 to 2019 identified 57 cases treated with LTS. We divided them into three groups: no pre-existing tracheostomy (n = 26), pre-existing tracheostomy with preservation of the pre-existing fistula (n = 20), and pre-existing tracheostomy without preservation of the pre-existing fistula (n = 11). The first group underwent traditional modified Lindeman's procedure. The second received transection of the trachea above the tracheostomy site, while the third had transection of the trachea at the tracheostomy site and creation of a distal end tracheostomy. Proper length and the angle of the cannula were selected to prevent damaging the innominate artery by the tip of the cannula. If the innominate artery compressed the trachea anteriorly, prophylactic arterial transection was considered., Results: Three patients (5.3%) died from causes unrelated to the surgical treatment. Only one patient had a postoperative TIAF followed by LTS (1.8%). Other postoperative complications were: wound infection (8.8%), intratracheal granuloma (12.3%), intratracheal minor bleeding (10.5%), wound granuloma (43.9%), leakage (1.8%). No one required revision of LTS., Conclusion: Success rates of LTS were high without major complications in all three groups and implies a safe operation and a definitive solution to intractable aspiration., (Copyright © 2021 Elsevier Inc. All rights reserved.)

It is impossible to secure the airway of a patient with "neck-only" breathing transorally or transnasally. Surgical removal of the larynx (laryngectomy) or tracheal rerouting (tracheoesophageal diversion or laryngotracheal separation) creates anatomic discontinuity. Misguided attempts at oral intubation of neck breathers may cause hypoxic brain injury or death. We present national data from the American Academy of Otolaryngology-Head and Neck Surgery, the American Head and Neck Society, and the United Kingdom's National Reporting and Learning Service. Over half of US otolaryngologist respondents reported instances of attempted oral intubations among patients with laryngectomy, with a mortality rate of 26%. UK audits similarly revealed numerous resuscitation efforts where misunderstanding of neck breather status led to harm or death. Such data underscore the critical importance of staff education, patient engagement, effective signage, and systems-based best practices to reliably clarify neck breather status and provide necessary resources for safe patient airway management.

Purpose: Tracheoesophageal diversion (TED) is an effective therapeutic procedure for intractable aspiration. In this study, we performed TED in cases of intractable aspiration and/or repetitive pneumonia, investigated the main route of nutritional uptake after the procedure, and evaluated the swallowing method using videofluoroscopy. We also evaluated the validity of TED for treating intractable aspiration., Methods: We retrospectively reviewed 44 patients (30 male and 14 female patients; median age, 55 years; range 15-85 years) who underwent TED for the treatment of intractable aspiration between January 2008 and December 2017. We examined the route of nutritional uptake before and after the operation and performed videofluoroscopy to detect the swallowing method after the operation., Results: The percentage of patients with oral intake increased from 21% (9/44) before TED to 56% (25/44) within 1 month after TED (p < 0.01); this percentage included patients with poor preoperative swallowing function. Overall, 60% patients who were able to communicate and mobilize using a wheelchair as well as 92% patients who were able to communicate and mobilize in the supine position were able to consume food orally. We subsequently performed videofluoroscopy in 24 of the 25 patients with oral intake and assessed the passage route of the contrast agent, which was found to move through the laryngeal route in 54% of these patients., Conclusion: TED may be suitable for the treatment of intractable aspiration and can improve oral intake, particularly in patients with good mobility and communication ability.

Less than one percent of trauma admission cases are categorized as pediatric neck trauma [13]. Nevertheless, due to an increasingly mobile society, there has been an increasing frequency of pediatric neck trauma with motor vehicle accidents being the most common mechanism of injury [8]. We present a case of laryngotracheal separation from a blunt, clothesline injury to the neck in a pediatric patient. We also review the literature and discuss the benefit of balloon airway dilation and its assistance in the management of laryngeal trauma and its resultant effects., (Copyright © 2016 Elsevier Ireland Ltd. All rights reserved.)

Pediatric laryngotracheal injuries from blunt neck trauma are extremely rare, but can be potentially catastrophic. Early diagnosis and skillful airway management is critical in avoiding significant morbidity and mortality associated with these cases. We present a case of a patient who suffered a complete tracheal transection and cervical spine fracture following a clothesline injury to the anterior neck. A review of the mechanisms of injury, clinical presentation, initial airway management, and anesthetic considerations in laryngotracheal injuries from blunt neck trauma in children are presented., (© 2015 John Wiley & Sons Ltd.)

Intractable aspiration is a serious, often life-threatening condition due to its potential impact on pulmonary function. Aspiration requires therapeutic measures, starting with conservative management but often necessitating surgical treatment. The basic surgical principle is to separate the alimentary and respiratory tracts through a variety of procedures which, unfortunately, nearly all result in the loss of phonation, with the exception of total laryngectomy (TL) which includes the placement of an indwelling voice prosthesis. In this study, we present a modified laryngotracheal separation (LTS) technique that, we believe, offers multiple advantages compared to standard TL. After reviewing the medical records of 35 patients with intractable aspiration who have undergone LTS, we describe the surgical technique and present the postoperative result. In a second surgical procedure about two months following LTS, we aimed to achieve voice restoration by placement of an indwelling voice prosthesis. Intractable aspiration was successfully treated in all patients. Placement of an indwelling voice prosthesis during a second operation was successful in 15 patients, representing the largest reported cohort thus far. LTS is a reliable surgical technique to treat intractable aspiration, with restoration of oral intake, thereby improving the general condition and quality of life of these unfortunate patients. Furthermore, voice restoration can be achieved in selected patients, by placement of a voice prosthesis.

There is a risk of developing a fatal trachea-innominate artery fistula following laryngotracheal separation for the prevention of intractable aspiration pneumonia. We developed a novel technique of surgical closure of the larynx to avoid this complication and provide long-term cannula-free care.

Conclusions: Tracheoesophageal diversion (TED) and laryngotracheal separation (LTS) can prevent aspiration pneumonia and improve the morbidity of patients with severe motor and intellectual disability (SMID). By improving hospitalization rates and care needs, the quality-of-life can be improved for the patients and their parents., Objectives: This study evaluated the clinical outcomes of TED and LTS in patients with intractable aspiration and SMID., Methods: This study retrospectively reviewed patients with SMID and intractable aspiration pneumonia who underwent TED or LTS at the institution between January 2008 and January 2015. It assessed the frequency of sputum suctioning, the number of pre-operative and post-operative hospitalizations, the operative time, and complications., Results: Forty patients were identified during the study period. After surgery, there were significant reductions in the frequency of secretion suctioning (from 165.0 times/day to 33.0 times/day) and the number of hospitalizations because of aspiration pneumonia (from 5.4 times/year to 0.2 times/year). A tracheocutaneous fistula occurred in one (2.5%) patient, and two (5.4%) patients developed tracheoinnominate artery fistulas. In the latter group, the innominate arteries were successfully ligated and endovascular embolization was performed.

Aim of the Study: Sialorrhoea and chronic salivary aspiration are a major problem in many neurologically impaired children causing embarrassment, skin issues and recurrent lower respiratory tract infections (LRTI). The aim of this study was to assess the efficacy of salivary gland surgery in the treatment of chronic salivary aspiration in such children., Objectives: To compare admission rates for LRTI per annum before and after surgical intervention., Methods: Retrospective review of all patients who underwent salivary management surgery for chronic aspiration under Princess Margaret Hospital's (PMH) Otolaryngology department from 2006 until 2013., Results: Twelve patients were included in this review. Their ages ranged from 3 to 21 years (mean=11.4). Their genders were equally distributed. Two patients had underlying congenital disorders; one had an acquired brain injury, while the majority (n=9, 75%) had cerebral palsy secondary to a sustained perinatal injury. Most patients (n=11, 91.7%) had bilateral submandibular gland excision and parotid duct ligation as a primary procedure. One patient had a laryngotracheal separation. Two patients went on to have a second procedure. The mean follow up time was five years. Using Wilcoxon Signed-Rank test we showed that the median rate of admission per annum for LRTI pre-operatively was 1.0. This was reduced to 0.5 post-operatively, which was statistically significant (p≤0.05)., Conclusions: We hypothesize that the combination of bilateral submandibular gland excision and bilateral parotid duct ligation is effective in reducing admissions with aspiration pneumonia in neurologically impaired children, and therefore improves the quality of life in these patients., (Crown Copyright © 2014. Published by Elsevier Ireland Ltd. All rights reserved.)

Objectives: Tracheo-innominate artery fistula (TIF) is a rare but life-threatening complication of tracheostomy. There are many reports about TIF but the mechanism of TIF formation remains poorly documented. Our objective is to investigate the TIF pathologically and suggest the possible mechanism of TIF formation., Patient and Methods: The patient was an 11-year old boy with a history of severe childhood epilepsy, cerebral palsy, and psychomotor retardation who died from TIF. We investigated the TIF histopathologically through his autopsy and reviewed bibliographical considerations., Results: Autopsy revealed massive blood aspiration from a large TIF and histopathological findings showed the following: (1) fibroepithelial polyps around the fistula; (2) squamous metaplasia of tracheal mucosa; (3) disappearance of tracheal cartilages and tracheal glands; and (4) arteritis (infiltration of neutrophils into the mesarterium and endarterium). Probably, (1) and (2) indicate chronic and repetitive irritations caused by cannula. (3) and (4) indicate intense inflammation caused by local infection. In this case, it is suggested that the chronic damage by cannula and the local infection resulted in the fistula formation and rupture of innominate artery., Conclusion: The present case suggests a pathogenesis in which chronic damage to the anterior tracheal wall by the cannula and consequent infection led to fistulation and rupture of the innominate artery., (Copyright © 2013 Elsevier Ireland Ltd. All rights reserved.)

Laryngotracheal separation (LTS) is the most immediately life-threatening airway injury. LTS is so rare that very few otolaryngologists have experience with it. LTS is one of the diagnostic and therapeutic challenges in airway diseases and its management remains to be established. We experienced a patient with complete LTS after attempted hanging. A high index of suspicion, adequate imaging, prompt airway establishment and early surgical repair are the most vital factors in managing a patient with LTS.