Human tracheal transplantation: A systematic review of case reports.

BACKGROUND: Patients with long-segment airway stenosis not amenable to conventional surgery may benefit from tracheal transplantation. However, this procedure has been only anecdotally reported, and its indications, techniques, and outcomes have not been extensively reviewed. METHODS: We conducted a systematic Literature search to identify all original articles reporting attempts at tracheal transplantation in humans. RESULTS: Of 699 articles found by the initial search, 11 were included in the systematic review, describing 14 cases of tracheal transplantation. Patients underwent transplantation for benign stenosis in nine cases, and for malignancies in five cases. In 12 cases blood supply to the trachea was provided by wrapping the graft in a vascularized recipient's tissue, while in 2 cases the trachea was directly transplanted as a vascularized composite allograft. The transplantation procedure was aborted before orthotopic transplantation in two patients. Among the remaining 12 patients, there was 1 operative mortality, while 4 patients experienced complications. Immunosuppressants drugs were administered to the majority of patients postoperatively, and only one group of authors attempted their withdrawal, in five patients. At the end of follow-up, all 11 patients surviving the operation were alive, but 2 had a recurrent tracheal stenosis requiring an airway appliance for breathing. CONCLUSION: Human tracheal transplantation is still at an embryonic phase. Studies available in the Literature report different surgical techniques, and information on long-term outcomes is still limited. Future research is needed in order to understand the clinical value of this procedure.

Unsuspected severe tracheal stenosis and tracheomalacia after correction surgery for congenital scoliosis: A case report.

Tracheal stenosis and tracheomalacia in patients with congenital scoliosis are serious and rare conditions caused by congenital dysplasia, postintubation injury, trauma, and tracheal tumor. Anesthesia of a child with tracheal stenosis is challenging for anesthesiologists. We describe an 8-year-old female patient developed severe tracheal stenosis and tracheomalacia after growing rod implantation for congenital scoliosis. Comprehensive assessment of preoperative pulmonary function and airway morphology, which can be neglected clinically, should be performed in congenital scoliosis patients.

Anesthesia for laryngotracheal reconstruction in children: A narrative review.

Laryngotracheal stenosis, congenital or acquired, is a common cause of pediatric airway obstruction. Acquired subglottic stenosis frequently results from prolonged neonatal intubation. The clinical presentation of subglottic stenosis is variable, ranging from biphasic stridor and frequent upper respiratory infections to acute airway compromise. Optimal patient care requires clinical coordination within a multidisciplinary subspecialty team. Medical management includes optimizing respiratory status, gastroesophageal reflux, speech, feeding, nutrition therapies, and providing psychosocial support. If surgical intervention is required, the otolaryngologist, anesthesiologist, and perioperative team must collaborate closely to ensure successful operative outcomes. This narrative review of laryngotracheal stenosis will discuss the pathophysiology, clinical evaluation, medical management, and surgical interventions, and focus on the perioperative anesthetic considerations for children undergoing laryngotracheal reconstruction.

The impact of early surgical treatment of tracheal stenosis on neurorehabilitation outcome in patients with severe acquired brain injury.

INTRODUCTION: Acquired tracheal stenosis (TS) is a potentially life-threatening condition following prolonged intubation and/or tracheostomy in adult patients with severe Acquired Brain Injury (sABI), requiring a tracheal resection and reconstruction. METHODS: We included 38 sABI adult patients with TS, admitted at a post-acute Neurorehabilitation Hospital. Disability Rating Scale (DRS) and other functional assessment measures were recorded at admission (t1), before TS surgical treatment (t2), and at discharge (t3). Patients were defined as 'improved' when they changed from a more severe to a less severe disability, between time t2 and time t3, and as "not improved" when they did not show any further improvement between t2 and t3, or they already exhibited a disability improvement since time interval t1-t2. RESULTS: Time interval between the injury onset and TS surgical treatment (t2-t0) was associated with the patient's disability improvement, suggesting the t2-t0 time interval ≤ 115 days as a cutoff value for a possible functional recovery. A t2-t0 time interval ≤ 170 days is also associated to absence of persistent dysphagia. CONCLUSIONS: Early TS surgical treatment within 115 days from the injury onset contributes to the improvement of the disability level in patients with sABI, optimizing their functional outcomes and recovery potential.

The efficiency of endoscopic versus open surgical interventions in adult benign laryngotracheal stenosis: a meta-analysis.

BACKGROUND: The optimal treatments for adult benign laryngotracheal stenosis presently remains controversial. The majority of the disadvantages of endoscopic interventions with high recurrence rate and open surgical therapy accompanied by sophisticated techniques, complication and mortality, highlights the dilemma of option for treatments. PURPOSE: To compare endoscopic treatments with open surgical interventions in adult patients with benign laryngotracheal stenosis, analyze their clinical outcomes, recurrence, complication and mortality. METHODS: In the meta-analysis, the databases including PubMed, Embase, Ovid and Web of Science were searched for studies reporting adult benign laryngotracheal stenosis, and clinical outcomes were compared. The duplicate publications, reviews, comments or letters, conference abstracts, case reports were excluded. The random effect model was used for calculating the pooled effect estimates. RESULTS: Eight studies (1627 cases) referring to six retrospective and two prospective researches were ultimately included in the meta-analysis. The decreased risk estimates of recurrence rate in patients receiving open surgical interventions were detected, comparing with endoscopic interventions (P < 0.05). Subgroup analysis revealed that decreased risk estimates of restenosis rate were also observed in patients receiving open surgical interventions compared with endoscopic interventions (P < 0.05), based on prospective studies, Europe and America, < 2-year follow-up, laryngeal stenosis, stenotic length without inter-group difference or stenotic grade II alone. However, there were no statistically significant difference of recurrence rate between the two interventions (P > 0.05) based on retrospective studies, South Asia and Africa, ≥ 2-year follow-up, involving tracheal lesion, stenotic length with inter-group difference, or stenotic grades of I-IV. No notable difference in the incidence of complication or mortality were identified. CONCLUSIONS: Open surgical interventions were more suitable for most laryngotracheal stenosis without contraindications. Endoscopic interventions are increasingly being used to treat simple laryngotracheal stenosis, as well as complex airway stenosis in carefully selected cases. Multi-center prospective randomized controlled trials should be conducted to search for the standard treatments for laryngotracheal stenosis.

A refashioned foley catheter: novel cost-effective available stent in pediatric laryngotracheal reconstruction.

PURPOSE: Pediatric laryngotracheal reconstruction (LTR) for laryngotracheal stenosis (LTS) mandates stenting in certain situations. This study presents a novel commercially available and cost-effective stent, a refashioned foley catheter. METHODS: This prospective clinical study was performed on pediatric cases with LTS up to 8 years. The study was performed in a tertiary referral center. The selection of the proper foley catheter size for age was explained. The atraumatic insertion maneuver of the stent was also shown in detail in different situations of LTS. The endoscopic removal of the stent was also described. The mean follow-up was 6.45 ± 1.3 months. RESULTS: The study included 31 cases using the refashioned foley catheter stents. The study included 17 males and 14 females with a mean age of 3.45 ± 1.09. Subglottic stenosis was the most common cause of LTR in the study (74.2%) cases. The mean duration of stenting was 40.5 ± 3.7 days. Decannulation was achieved in 96.8% of cases. No stent complications were encountered like stent migration, excess granulation tissue, intractable aspiration, or pressure necrosis. CONCLUSION: The refashioned foley catheter is a novel, available, and inexpensive stent that can be utilized for LTR cases for pediatric LTS. The newly described stent is soft, pliable with atraumatic insertion and easy endoscopic removal with minimal complications.

Not Just Dyspnoea: Swallowing as a Concern for Adults with Laryngotracheal Stenosis Undergoing Airway Reconstruction.

Acquired laryngotracheal stenosis (LTS) is a rare condition causing dyspnea and stridor. Patients often require multiple surgical procedures with no guarantee of a definitive outcome. Difficulty swallowing is a recognised problem associated with LTS and the reconstructive surgeries required to manage the condition. The breathlessness patient's experience impacts on swallowing, and the vulnerable structures of the larynx are implicated during complex surgeries. This leads to dysphagia post-surgery, with some patients experiencing more chronic symptoms depending on the biomechanical impact of the surgery, or a pre-existing dysphagia. Despite this there is limited observational research about the dysphagia associated with LTS, with no exploration of the patient experience. Our aim was to investigate patient experience of living with LTS focussing on dysphagia in order to guide clinical practice. A qualitative study was completed using focus groups and semi-structured interviews with 24 patients who have had reconstructive surgery for LTS. Thematic analysis was used to identify three over-arching themes: The Physical Journey, The Emotional Journey and The Medical Journey. Key sub-themes included the importance of self-management and control, presence of symptoms, benefits of therapy, living with a life-long condition, fear and anxiety, autonomy, medicalisation of normal processes and the dichotomy between staff expertise and complacency. Swallowing was connected to all themes. The results are reviewed with consideration of the wider literature of lived experience particularly in relation to other chronic conditions and those that carry a high symptom burden such as head and neck cancer. Future clinical and research recommendations have been made. Akin to other clinical groups, adults with LTS are keen that management of their swallowing is person-centred and holistic.

Surgical management of congenital tracheal stenosis associated with complex cardiovascular anomalies.

PURPOSE: Although surgical outcomes have improved in patients with congenital tracheal stenosis (CTS), the management of such patients with complex cardiovascular anomalies (CVAs) remains a challenge. This study aimed to clarify the clinical features of this combination. METHODS: Medical records of 14 patients with complex CVAs who had undergone slide tracheoplasty for CTS between May 2016 and February 2022 were retrospectively reviewed. Complex CVAs were defined as CVAs without left pulmonary artery sling and simple cardiac shunts. Data collected included age and body weight at tracheal reconstruction, preoperative respiratory support, surgery details, and mortality. RESULTS: The median age and body weight at tracheal reconstruction were 3.5 (range, 1-17) months and 4.7 (range, 2.3-8.2) kg, respectively. Simultaneous repair of CTS and CVAs was performed in eight patients and staged repair in six patients. Patients who underwent simultaneous repair required preoperative respiratory support more frequently than those who underwent staged repair (8/8 [100%] vs 2/6 [33.3%]; P = 0.015). No mortality occurred. CONCLUSIONS: Patients with complex CVAs often require tracheal reconstruction in early infancy. Surgical management strategy for this combination was generally decided on the basis of the severity of respiratory symptoms due to CTS.

Treatment of tracheal stenosis with extracorporeal membrane oxygenation support in infants and newborns.

Tracheal stenosis in children is a challenge for the healthcare team, since it is a rare disease. Patients usually have other clinical comorbidities, mainly previous cardiac surgical interventions. This retrospective single-center study included infants with tracheal stenosis (congenital or acquired) operated between 2016 and 2020 on venoarterial extracorporeal membrane oxygenation (VA ECMO). Five patients were operated and the median age of detection of the tracheal disease was 3.7 months, and the median age at the operation was 5 months. All patients had associated cardiac anomalies. Four patients had congenital tracheal stenosis; two with associated pig bronchus. One patient had acquired subglottic stenosis with concomitant stenosis at the carina. After the operation, the patients were referred to ICU on ECMO with an open chest. Minor ECMO-related complications occurred in two patients (hemothorax and wound infection). All patients required endoscopic evaluation during the postoperative care; median of 3.2 procedures. Two patients are currently in follow-up and three have died. Slide tracheoplasty with VA-ECMO can be successfully performed in infants with prior cardiac surgery. Nevertheless, a difficult postoperative course should be anticipated, with possible prolonged ECMO use, readmissions, and higher morbidity and mortality than in children with tracheal stenosis alone.

Pseudomembranous Tracheobronchitis With Severe Tracheal Stenosis and Masked Bronchial Obstruction.

BACKGROUND: Pseudomembranous tracheobronchitis (PMTB) is a rare condition characterized by the formation of endobronchial pseudomembranes. PMTB overlaps with necrotizing tracheobronchitis or plastic bronchitis. The reported infectious etiology mainly includes invasive aspergillosis. PMTB can cause serious airway obstruction; however, urgent tracheotomy is rarely required. CASE REPORT: A 46-year-old woman was transferred to the emergency department (ED) with a 1-week history of progressive dyspnea and cough that was preceded by fever and sore throat. She was previously healthy except for a 20-year history of mild palmoplantar pustulosis. Stridor was evident. Nasolaryngoscopy performed in the ED revealed severe tracheal stenosis caused primarily by mucosal edema and secondarily by pseudomembranes. Initially, tracheitis was considered the sole cause of dyspnea. Although she underwent urgent tracheotomy to prevent asphyxia, her respiration deteriorated progressively. Bronchoscopy revealed massive pseudomembranes obstructing the bilateral bronchi, which led to the clinical diagnosis of PMTB. Subsequent toilet bronchoscopy markedly improved her ventilation. The causative pathogen was not identified despite extensive work-up, including molecular biological testing. Histopathologic examination of the pseudomembranes revealed fibrin with abundant neutrophils, which was consistent with PMTB. Associated conditions, including immunodeficiency, were not found. Her condition improved with antibiotics and repeated toilet bronchoscopy. WHY SHOULD AN EMERGENCY PHYSICIANS BE AWARE OF THIS?: PMTB is an important differential diagnosis of airway emergencies. PMTB can present with critical edematous tracheal stenosis and masked bronchial pseudomembranous obstruction. Emergency physicians should include PMTB in the differential diagnosis in adult patients with acute central airway obstruction because it requires prompt multimodal treatment.

Rigid dilatation of pediatric laryngotracheal stenosis as an adequate alternative to balloon dilatation.

INTRODUCTION: Endoscopic balloon dilation (EBD) is the mainstay of endoscopic therapy for laryngotracheal stenosis (LTS), although there is no evidence that it achieves better results than traditional rigid laryngeal dilators. Rigid bougie dilators are less expensive and easier to use, and confer the advantage of providing tactile information about the stenosis to the surgeon. We analyzed the outcome of endoscopic rigid bougie dilatation of LTS in a large series of children and compared it to the reported results of EBD in the same setting. PATIENTS AND METHODS: All cases of pediatric LTS treated by endoscopic rigid dilatation in a tertiary referral center between 2006 and 2015 were retrospectively studied. They were divided into a primary dilatation group (PDG) and a post-reconstruction dilatation group (PRG). The PDG children had no history of reconstructive airway surgery, and dilatation was the major treatment approach. The PRG children underwent dilatations after airway reconstruction surgery as part of routine postoperative management. A successful primary outcome was defined as improvement of dyspnea and achievement of a functional airway without reconstructive laryngotracheal surgery or need for a tracheostomy at final follow-up. RESULTS: Sixty-two children (68 cases, mean age 5.1 years, range 0.7-17.2) underwent 156 endoscopic rigid dilatations. Successful outcome was achieved in 48 cases (70.6%), 73.0% in the PDG and 67.7% in the PRG. There were no procedure-related adverse events. CONCLUSIONS: Endoscopic rigid dilatation is a relatively inexpensive and efficacious tool in endoscopic management of pediatric LTS. Its success rates are in the same range as those of EBD.

Pitfalls in the management of congenital tracheal stenosis: is conservative management feasible?

PURPOSE: Congenital tracheal stenosis (CTS) is rare and challenging. Complete tracheal rings cause a wide spectrum of airway-obstructing lesions and varying degrees of respiratory distress. Although surgical reconstruction is the primary option for symptomatic CTS, sometimes an appropriate management strategy may be difficult due to other anomalies. We aimed to identify pitfalls in the management of CTS. METHODS: We retrospectively reviewed the records of patients with CTS during the last 10 years in our institution. RESULTS: Sixteen pediatric patients were diagnosed with CTS. Of the 16 patients, 12 (75.0%) had cardiovascular anomalies including seven left pulmonary artery sling. Six patients with dyspnoea caused by CTS and three patients with difficult intubations due to CTS underwent tracheoplasty. Four patients underwent only cardiovascular surgery without tracheoplasty. Three asymptomatic patients were followed up without undergoing any surgical procedure. We repeatedly discussed management of four patients with especially complex pathophysiology at multidisciplinary meetings. Right ventricular outflow tract obstruction, tracheobronchial malacia, increased pulmonary blood flow, and pulmonary aspiration due to gastroesophageal reflux presumably accounted for their severe respiratory distress, and we forewent their tracheal reconstruction. CONCLUSION: The management of CTS should be individualized, and conservative management is a feasible option in selected cases.

Durability of Silicone Airway Stents in the Management of Benign Central Airway Obstruction.

PURPOSE: The literature is devoid of a comprehensive analysis of silicone airway stenting for benign central airway obstruction (BCAO). With the largest series in the literature to date, we aim to demonstrate the safety profile, pattern of re-intervention, and duration of silicone airway stents. METHODS: An institutional database was used to identify patients with BCAO who underwent rigid bronchoscopy with dilation and silicone stent placement between 2002 and 2015 at Rush University Medical Center. RESULTS: During the study period, 243 stents were utilized in 63 patients with BCAO. Pure tracheal stenosis was encountered in 71% (45/63), pure tracheomalacia in 11% (7/63), and a hybrid of both in 17% (11/63). Median freedom from re-intervention was 104 (IQR 167) days. Most common indications for re-intervention include mucus accumulation (60%; 131/220), migration (28%; 62/220), and intubation (8%; 18/220). The most common diameters of stent placed were 12 mm (94/220) and 14 mm (96/220). The most common lengths utilized were 30 mm (60/220) and 40 mm (77/220). Duration was not effected by stent size when placed for discrete stenosis. However, 14 mm stents outperformed 12 mm when tracheomalacia was present (157 vs. 37 days; p = 0.005). Patients with a hybrid stenosis fared better when longer stents were used (60 mm stents outlasted 40 mm stents 173 vs. 56 days; p = 0.05). CONCLUSION: Rigid bronchoscopy with silicone airway stenting is a safe and effective option for the management of benign central airway obstruction. Our results highlight several strategies to improve stent duration.

A rare cause of noninvasive ventilation failure: tracheal stenosis.

Noninvasive ventilation is the first line treatment of choice in acute respiratory failure in many diseases including post-extubation respiratory failure. Herein we report a case unresponsive to noninvasive ventilation due to tracheal stenosis. A 49- year -old female was admitted to intensive care unit after successful resuscitation of cardiac arrest. During the follow-up, she was extubated on 16th day and then transferred to the coronary ward. Four days later, she started to have progressive dyspnea and difficulty in breathing. Arterial blood gas evaluation showed respiratory acidosis with moderate hypercapnia. Noninvasive ventilation was initiated with the diagnosis of cardiogenic pulmonary edema however she did not respond to noninvasive ventilation therapy. Pulmonary consultation revealed that she had a new onset stridor. She had an urgent fiberoptic bronchoscopy which revealed severe tracheal stenosis. Tracheal stenosis should be considered in patients who do not respond to noninvasive ventilation after extubation like in our case.

Effects of Balloon Dilation for Idiopathic Laryngotracheal Stenosis on Voice Production.

OBJECTIVE: To evaluate the effects of balloon dilation for idiopathic laryngotracheal stenosis on voice production. METHODS: Retrospective review of 10 female patients with idiopathic laryngotracheal stenosis undergoing balloon dilation. Voice outcomes were evaluated by comparing pre- and posttreatment patient-reported, perceptual, aerodynamic, and acoustic parameters. Complete data sets were not available for all subjects; sample size for each parameter is reported with the corresponding result. RESULTS: Total Voice Handicap Index (VHI) decreased significantly (22.9 ± 13.5 to 6.8 ± 6.5; n = 8; p = .015), as did glottal function index (7.2 ± 4.9 to 1.5 ± 2.0; n = 6; p = .022). No changes were observed in the GRBAS (grade, roughness, breathiness, asthenia, strain) scale. Changes in aerodynamic parameters were not statistically significant. Percent jitter decreased (1.32 ± 1.37 to 0.60 ± 0.29; n = 7; P = .078), and fundamental frequency range was preserved (507 ± 325 to 612 ± 281; n = 7; P = .309). CONCLUSIONS: Our sample of patients with idiopathic laryngotracheal stenosis had a mild-moderate dysphonia that improved with balloon dilation. Importantly, adverse effects on voice that can occur with open procedures were not observed. Patient perception of dysphonia improved while fundamental frequency range was maintained and aerodynamic parameters remained within or moved toward the normal range. Larger prospective studies are warranted to further evaluate changes in voice production associated with balloon dilation.

A Novel Therapy for Laryngotracheal Stenosis: Treatment With Ethosomes Containing 5-Fluorouracil.

PURPOSE: The purpose of our article was to explore the effect of ethosomes containing 5-fluorouracil (5-FU) with different sizes on laryngotracheal stenosis treatment. METHODS: The physical characteristics of ethosomes containing 5-FU were investigated, including size, shape, and entrapment percentage. The effect of ethosomes containing 5-FU was evaluated on the airway stenosis rabbit model. The formation of fibrous/scar tissue was investigated by hematoxylin and eosin (HE) staining, and the permeation depth was observed under fluorescence microscope. RESULTS: The mean sizes of 5-FU ethosomes extruded by D=50 nm and D=100 nm pore were 60±10 nm and 110±13 nm, respectively. The 5-FU entrapment percentage of ethosomes was determined to be 15% (D=60±10 nm) and 32% (D=110±13 nm). After being treated by ethosomes containing 5-FU (D=60±10 nm), the fibroblast and collagenous fiber distributed sparsely in the deep scar tissue. The permeation capability of ethosomes containing 5-FU (D=60 nm) was significantly better than ethosomes (D=110 nm). Besides, the 5-FU ethosomes resulted in less stenosis than 5-FU only. CONCLUSIONS: Topical administration of 5-FU ethosomes may be a novel candidate therapy for laryngotracheal stenosis treatment.

Safety of outpatient airway dilation for adult laryngotracheal stenosis.

OBJECTIVES: To evaluate the safety of outpatient airway dilation for adult patients with subglottic or tracheal stenosis. METHODS: The records of patients treated with airway dilation between October 2003 and September 2013 were reviewed. Outcomes of patients who underwent dilation as inpatients versus outpatients were compared. Emergency room visits, readmissions, and 3 or more primary care physician visits within 30 days postoperatively were specifically evaluated. Postoperative hemorrhage, airway edema, recurrent laryngeal nerve paralysis, reintubation, tracheostomy, tracheal rupture, pneumomediastinum, pneumothorax, acute respiratory distress, or death were also reviewed. RESULTS: One hundred fourteen dilations performed in 53 patients with airway stenosis were included. Outpatient dilation was performed in 93 (82%); 21 (18%) underwent the procedure in the inpatient setting. Complications were low among both inpatient and outpatient groups (10% vs 1%, P=.09). No complications occurred during the overnight stay of the inpatient group. CONCLUSIONS: Outpatient airway dilation is a safe and feasible procedure. It can be routinely performed on an ambulatory basis.

Voice outcomes after endoscopic treatment of laryngotracheal stenosis.

OBJECTIVE: This study aimed to assess vocal disability in patients with laryngotracheal stenosis who are managed with endoscopic surgery, comparing patients with posterior glottic stenosis (PGS) to those with subglottic or tracheal stenosis (SGS/TS). METHODS: Patients who underwent endoscopic treatment for laryngotracheal stenosis with voice outcomes data from 2005 to 2013 were studied. The mean Voice Handicap Index-10 (VHI-10) was compared over the study period. Of those with SGS/TS, the distance from the vocal folds to the proximal portion of the stenosis was obtained and compared to VHI-10. RESULTS: Forty-four patients met inclusion criteria. The mean VHI-10 for all patients was 14.6. Fifty percent were dysphonic (VHI-10>11). The mean VHI-10 for PGS patients was 22.4, and for SGS/TS patients, 10.9 (P=.004). Of those with PGS, 78.6% were dysphonic compared to only 36.7% of those with SGS/TS. Voice Handicap Index-10 improved from 14.1 for those with proximal stenosis to 4 for those with stenosis more than 2 cm distal to the vocal folds. CONCLUSION: Following endoscopic management of stenosis, those with PGS have poorer voice outcomes compared to those with SGS/TS. The majority of those with SGS/TS are not dysphonic. Vocal outcomes are greatest for those with stenosis beginning at least 2 cm distal to the vocal folds.

Primary fibrosarcoma of the trachea presenting with acute airway loss.

Primary sarcomas of the trachea are rare occurrences that present with nonspecific symptoms, making timely diagnosis difficult. We report a case of primary fibrosarcoma of the trachea that presented with acute airway loss secondary to tracheal discontinuity due to tumor destruction. This unusual clinical presentation highlights the difficulties posed in the diagnosis and management of tracheal sarcomas. A discussion of the relevant literature is presented.