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Abstract 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.

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. [ABSTRACT FROM AUTHOR]

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. [ABSTRACT FROM AUTHOR]

Abstract 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.

Background: Stenosis of the permanent tracheostoma causes the patient reduced quality of life. We herein report a treatment of permanent tracheostomal stenosis with local steroid injection after laryngotracheal separation in a patient with severe motor and intellectual disabilities. Case presentation: The patient was a 22-year-old woman with congenital myopathy who underwent laryngotracheal separation at the age of 21 years. Tracheostomal enlargement was performed at the age of 22 years. Two months after the operation, permanent tracheostomal stenosis developed owing to a hard cicatricial constriction. Although the size of the tracheostoma decreased, yet the patient did not have any respiratory distress. Therefore, we started local steroid injection therapy using triamcinolone acetonide instead of revision surgery. After those 3 injections, the tracheostomal stenosis was apparently dilated. The patient received this treatment 19 times in 3 years and 4months. She has not needed this treatment in the past 1 year and 8 months. No adverse effect of the corticosteroid was seen during the 5-year period. Conclusions: On the basis of our experience, if patients have no symptoms related to stenotic airway obstruction, triamcinolone acetonide injection therapy, instead of revision surgery, is a simple, safe, and highly effective procedure for permanent tracheostomal stenosis.

Abstract 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.

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. The clinical course of three patients with MDS who underwent laryngotracheal separation (LTS) at two institutions was summarized. 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. 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. [ABSTRACT FROM AUTHOR]

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. [ABSTRACT FROM AUTHOR]

• Laryngotracheal separation is the definitive solution to intractable aspiration pneumonia in neurologically impaired children. • Tracheo-innominate artery fistula is a potentially fatal complication. • Surgical options depend on whether patients have the pre-existing tracheostomy or can transect the trachea above the tracheostomy site. • Appropriate angle and length of the cannula should be selected. 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. 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. 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. 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. [ABSTRACT FROM AUTHOR]

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.)

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. [ABSTRACT FROM AUTHOR]

Competing Interests: None

Stenosis of the permanent tracheostoma causes the patient reduced quality of life. We herein report a treatment of permanent tracheostomal stenosis with local steroid injection after laryngotracheal separation in a patient with severe motor and intellectual disabilities. The patient was a 22-year-old woman with congenital myopathy who underwent laryngotracheal separation at the age of 21 years. Tracheostomal enlargement was performed at the age of 22 years. Two months after the operation, permanent tracheostomal stenosis developed owing to a hard cicatricial constriction. Although the size of the tracheostoma decreased, yet the patient did not have any respiratory distress. Therefore, we started local steroid injection therapy using triamcinolone acetonide instead of revision surgery. After those 3 injections, the tracheostomal stenosis was apparently dilated. The patient received this treatment 19 times in 3 years and 4months. She has not needed this treatment in the past 1 year and 8 months. No adverse effect of the corticosteroid was seen during the 5-year period. On the basis of our experience, if patients have no symptoms related to stenotic airway obstruction, triamcinolone acetonide injection therapy, instead of revision surgery, is a simple, safe, and highly effective procedure for permanent tracheostomal stenosis. • Permanent tracheostomal stenosis (PTS) can cause the patient reduced quality of life. • We show a conservative treatment with local steroid injection therapy using triamcinolone acetonide (TAC). • If patients have no symptoms related to stenotic airway obstruction, TAC injection therapy, instead of revision surgery, is a simple, safe, and highly effective procedure for PTS to maintain their QOL. [ABSTRACT FROM AUTHOR]

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.

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.