BACKGROUND: Pediatric laryngotracheal stenosis often requires open airway reconstruction. While these surgeries establish an airway for adequate ventilation, many patients develop subsequent dysphonia. Numerous studies have reported outcomes related to voice.
OBJECTIVE: This study aims to evaluate dysphonia in pediatric patients following open airway reconstruction, focusing on acoustic parameters, perceptual voice quality, and voice-related quality of life.
METHODS: A comprehensive search using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines across 6 databases identified articles involving pediatric patients who underwent open airway reconstruction and reported postoperative vocal acoustic parameters, perceptual voice quality, voice-related quality of life, or vocal mechanics. Articles were assessed for bias risk, and common outcomes were synthesized qualitatively and quantitatively using meta-analyses.
RESULTS: Among 4089 articles, 21 were included, involving 497 pediatric patients. Laryngotracheoplasty was the most common procedure followed by cricotracheal resection. The Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) scale was frequently used to assess voice quality, with a mean score of 55.6 [95% confidence intervals (CIs): 47.9-63.3]. Voice-related quality of life was measured using the pediatric Voice Handicap Index (pVHI) and Pediatric Voice-Related Quality of Life Survey, with mean scores of 35.6 (95% CI: 21.4-49.7) and 83.7 (95% CI: 74.1-93.2), respectively. The fundamental frequency was 210.5 (95% CI: 174.6-246.3). Other common findings included supraglottic phonation, anterior commissure blunting, posterior glottic diastasis, and abnormal vocal cord mobility.
CONCLUSIONS: Pediatric patients experiencing dysphonia after open airway reconstruction exhibited moderately decreased voice quality and reduced voice-related quality of life. However, there was inconsistency in study protocols and outcome measures used. Preserving voice quality during airway reconstruction is crucial to avoid negative impacts on quality of life.

BACKGROUND: A straight silicone stent can be used to treat proximal benign tracheal stenosis in non-surgical candidates. However, stent migration is a common complication when placed at a particular location and can lead to major complications. This case series of laryngotracheal stenosis reports a fixation method for straight silicone stents in the subglottic trachea (Stage 3 of the McCaffrey classification).
METHODS: The medical charts of these patients scheduled for straight silicone stent placement with suture fixation between 2014 and 2020 at the CHU UCL Namur Hospital (Belgium) were retrospectively reviewed. The procedure was performed using a rigid bronchoscope. Details of the procedure were obtained from medical records.
RESULTS: This case series included six patients (males: 4, females: 2). The median patient age was 59 years. Two suture fixations were placed following previous silicone stent migration episodes, whereas the others were placed proactively to avoid this risk. All fixations were performed by the device Freka® Pexact II ENFIt®, originally developed for gastropexy in endoscopic gastrostomy. The sutures were subcutaneously buried.
CONCLUSIONS: During the 6-month follow-up period, complications such as fixation issues and stent migration were reported despite the off-label use of the treatment. The straight silicone stent fixation technique used in this case series was simple and effective for securing the stent in upper benign tracheal stenosis.

The article presents an analysis of the plastic reconstructive surgery effectiveness for patients with an extended tracheal defect using an allograft based on the dura mater (DM) at the final stage of surgical treatment of laryngeal and tracheal cicatricial stenosis. The study included 20 patients with cicatricial stenosis of the larynx and trachea, who were previously performed plastic reconstructive treatment with scar tissue excision in the lumen of the respiratory tract and restoration of the supporting frame of the larynx and trachea using allografts based on costal allocartilage. The age of the patients ranged from 21 to 54 years, the duration of the disease was from 1 to 5 years. After a standard clinical and laboratory examination, with a mandatory video endoscopic examination of the larynx and trachea, multislice computed tomography of the larynx and trachea, patients underwent plastic closure of the tracheal defect using DM. Dynamic outpatient monitoring was carried out once a week for 1 month, once a month for 3 months, control examination was done 6 months after surgical treatment. The results of the study demonstrated a full-fledged social and labor rehabilitation of all 20 patients after the final stage of surgical treatment using DM, the absence of rejection reaction and migration of allo-implantation material, the preserved lumen of the larynx and trachea with a rigid supporting skeleton and the absence of anterior tracheal wall floatation. The use of DM as an additional strengthening of the anterior tracheal wall for patients with deficiency of muscular aponeurotic tissues and more than 2 cm size tracheal defect is highly effective at the final stage of surgical treatment for plastic closure of the tracheal defect.
В статье представлен анализ эффективности реконструктивно-пластического оперативного лечения пациентов с протяженным трахеальным дефектом с использованием аллотрансплантата на основе твердой мозговой оболочки (ТМО) на заключительном этапе оперативного лечения рубцового стеноза гортани и трахеи (РСГТ). В исследование включены 20 пациентов с РСГТ, получивших ранее реконструктивно-пластическое лечение с иссечением рубцовой ткани в просвете дыхательных путей и восстановлением опорного каркаса гортани и трахеи с помощью аллотрансплантатов на основе реберного аллохряща. Возраст пациентов составил от 21 года до 54 лет, длительность заболевания — от 1 года до 5 лет. После стандартного клинико-лабораторного обследования с обязательным видеоэндоскопическим исследованием гортани и трахеи, мультиспиральной компьютерной томографией (МСКТ) гортани и трахеи пациентам проведено пластическое закрытие трахеального дефекта с использованием ТМО. Динамическое амбулаторное наблюдение осуществлялось 1 раз в неделю в течение 1 мес, 1 раз в месяц в течение 3 мес, контрольный осмотр — через 6 мес после оперативного лечения. Результаты исследования демонстрировали полноценную социально-трудовую реабилитацию всех 20 пациентов после заключительного этапа оперативного лечения с использованием ТМО, отсутствие реакции отторжения и миграции аллоимплантационного материала, сохранный просвет гортани и трахеи с ригидным опорным скелетом и отсутствием флотирования передней трахеальной стенки. Использование ТМО в качестве дополнительного укрепления передней трахеальной стенки при дефиците собственных мышечно-апоневротических тканей и протяженности трахеального дефекта более 2 см высокоэффективно на заключительном этапе оперативного лечения в виде пластического закрытия трахеального дефекта.

OBJECTIVE: Treatment options for benign subglottic stenosis include endoscopic techniques or open surgery. Although endoscopic treatment is less invasive, a considerable proportion of patients develop recurrent stenosis. Endoscopic pretreatments do not exclude patients from a later surgical repair; however, the impact of previous endoscopic treatment attempts on functional outcome after open surgery is unknown.
METHODS: All patients, who received a cricotracheal resection (CTR) between January 2017 and June 2023 at the Department of Thoracic Surgery, Medical University of Vienna, were included in this retrospective study. Patient characteristics, surgical variables and postoperative outcome including a detailed functional assessment were analysed.
RESULTS: A total of 65 patients received a CTR during the study period, of which 40 were treatment naïve and 25 had a median of 2 (range 1-9) endoscopic pretreatments. Less-invasive voice-sparing CTR or standard CTR were more often possible in treatment-naïve patients. In contrary, pretreated patients regularly required extended procedures (P = 0.049). Three or more endoscopic treatments resulted in a significantly lower mean fundamental frequency (F0) after open repair (P = 0.048). In addition, a trend towards smaller mean sound pressure levels, a higher voice handicap index, higher impairments in RBH scores (roughness, breathing and hoarseness) and a higher dysphagia severity index was found in pretreated patients. The respiratory outcome after surgery was comparable between both groups.
CONCLUSIONS: Multiple endoscopic pretreatments lead to worse voice quality after CTR. The impact of prior endoscopic treatment before surgical repair should be considered when discussing treatment options with patients suffering from subglottic stenosis.

Congenital subglottic stenosis is a rare but potentially catastrophic condition. In this report, we describe the management of a term neonate who was noted to have biphasic stridor during preassessment for correction of an imperforate anus at 26 hours of life. The neonate was found to have a pinhole trachea secondary to congenital subglottic stenosis. It was impossible to pass an endotracheal tube, so the neonate underwent an emergency surgical tracheostomy with a good outcome. A high index of suspicion led to appropriate steps being taken to safely anaesthetise the neonate.

OBJECTIVE: Utilizing a novel histopathological scoring system and subglottic stenosis (SGS) rabbit model, we aimed to compare degrees of inflammation and severity of narrowing in the subglottis between two minimally invasive therapeutic modalities: endoscopic balloon dilation (EBD) alone versus EBD with placement of a bioabsorbable ultra-high ductility magnesium (UHD-Mg) alloy stent.
METHODS: SGS was induced endoscopically via microsuspension laryngoscopy in 23 New Zealand white rabbits. The control group (n = 11) underwent EBD alone, the study arm (n = 12) underwent EBD with implantation of bioabsorbable UHD-Mg alloy stents. Rabbits were euthanized at 2-, 3-, and 6-weeks after SGS induction, coinciding with wound healing stages. Using Optical Coherence Tomography (OCT), cross-sectional areas of airways were compared to calculate the mean percentage of intraluminal area at sequential time points. A novel histopathological scoring system was used to analyze frozen sections of laryngotracheal complexes. The degree of inflammation was quantified by scoring changes in inflammatory cell infiltration, epithelial ulceration/metaplasia, subepithelial edema/fibrosis, and capillary number/dilation. Univariate analysis was utilized to analyze these markers.
RESULTS: We found rabbits implanted with the bioabsorbable UHD-Mg alloy stent had statistically significantly higher scores in categories of hyperplastic change (stents vs controls: 1.48 vs 0.46 p < 0.001), squamous metaplasia (22 vs 5 p < 0.001), and neutrophils/fibrin in lumen (31 vs 8, p < 0.001). Rabbits who received EBD alone had higher scores of subepithelial edema and fibrosis (2.70 vs 3.49, p < 0.0256). The stented rabbits demonstrated significantly increased mean percent stenosis by intraluminal mean area compared to controls at 2 weeks (88.56 vs 58.98, p = 0.032), however at all other time points there was no significant difference between intraluminal subglottic stenosis by mean percent stenosis area.
CONCLUSIONS: Rabbits with SGS treated with UHD-Mg alloy stents demonstrated histopathologic findings suggestive of lower levels of tracheal fibrosis. This could indicate a reduced tendency towards the development of stenosis when compared to EBD alone. There was not a difference in luminal size between stent and non-stented rabbits at the six-week end point. Histologically, however, overall the use of bioabsorbable UHD-Mg alloy stenting elicited a greater tissue response at the level of the superficial mucosa rather than fibrosis of the lamina propria seen in the stented rabbits. This suggests more favorable healing and less of a tendency towards fibrosis and stenosis even though there may not be a benefit from a luminal size standpoint during this early healing period. Compared to known complications of currently available non-bioabsorbable metal or silicone-based stents, this proof-of-concept investigation highlights the potential use of a novel biodegradable UHD-Mg stent as a therapeutic modality for pediatric SGS.

Endoscopic posterior cricoid split and costal rib graft placement (EPCSCG) is an important tool in enlarging the glottic and subglottic airway, both of which can be disproportionally affected in the small airways of neonates and early infants. We present a series of 8 patients under the age of one who successfully underwent EPCSCG, with 7/8 patients avoiding tracheostomy entirely. Of these patients, the indication for EPCSCG was isolated bilateral vocal fold immobility (6/8), bilateral vocal fold immobility with subglottic stenosis (1/8), and isolated subglottic stenosis (1/8). EPCSCG can be safely applied to select patients less than one year of age.

OBJECTIVE: To investigate the role of periostin (POSTN) and the transforming growth factor β (TGF-β) pathway in the formation of laryngotracheal stenosis (LTS) scar fibrosis and to explore the specific signaling mechanism of POSTN-regulated TGF-β pathway in tracheal fibroblasts.
METHODS: Bioinformatics analysis was performed on scar data sets from the GEO database to preliminarily analyze the involvement of POSTN and TGF-β pathways in fibrosis diseases. Expression of POSTN and TGF-β pathway-related molecules was analyzed in LTS scar tissue at the mRNA and protein levels. The effect of POSTN on the biological behavior of tracheal fibroblasts was studied using plasmid DNA overexpression and siRNA silencing techniques to regulate POSTN expression and observe the activation of TGF-β1 and the regulation of cell proliferation and migration via the TGF-β/RHOA pathway.
RESULTS: The bioinformatics analysis revealed that POSTN and the TGF-β pathway are significantly involved in fibrosis diseases. High expression of POSTN and TGF-β/RHOA pathway-related molecules (TGFβ1, RHOA, CTGF, and COL1) was observed in LTS tissue at both mRNA and protein levels. In tracheal fibroblasts, overexpression or silencing of POSTN led to the activation of TGF-β1 and regulation of cell proliferation and migration through the TGF-β/RHOA pathway.
CONCLUSIONS: POSTN is a key molecule in scar formation in LTS, and it regulates the TGF-β/RHOA pathway to mediate the formation of cicatricial LTS by acting on TGF-β1. This study provides insights into the molecular mechanisms underlying LTS and suggests potential therapeutic targets for the treatment of this condition.
METHODS: NA Laryngoscope, 134:4078-4087, 2024.

The aim of this article is to present experiences of the Department of Otorhinolaryngology and Head and Neck Surgery, Zagreb University Hospital Center with the treatment of patients with subglottic stenosis. Subglottic stenosis is a rare congenital or acquired disorder of airway patency that is part of a wider complex of disorders known as laryngotracheal stenosis with the ultimate effect in the form of respiratory insufficiency that can be life-threatening. As an acquired condition, it is most often the result of iatrogenic damage to the larynx and trachea during invasive airway management, whether it is intubation or tracheotomy. In the case of intubation as the etiologic factor, cases of prolonged intubation were most common. Retrospective analysis of patient medical histories over a ten-year period was performed and 29 patients met the inclusion criteria. All patients were monitored for at least two years after completion of treatment. Out of a total of 29 treated patients, 20 were permanently decannulated, of which 4 have paresis of one or both vocal cords. In conclusion, there is no clear treatment protocol for patients with subglottic stenosis. The optimal modality of treatment is combined endoscopic and open surgical treatment.

A female infant born at 38 weeks and 2 days via induced vaginal delivery was admitted to the neonatal intensive care unit for respiratory distress soon after birth. Noted to have aphonia on examination, the patient underwent direct laryngoscopy and was diagnosed with an anterior glottic web and subglottic stenosis. The patient underwent a genetic workup including whole exome sequencing which resulted in a diagnosis of a FREM1-associated disorder. Congenital glottic webs and subglottic stenoses have not been previously described as clinical manifestations of FREM1-associated disorders.