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1.
Int J Pediatr Otorhinolaryngol ; 181: 111994, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38823367

ABSTRACT

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. DISCUSSION: 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.


Subject(s)
Absorbable Implants , Alloys , Disease Models, Animal , Laryngoscopy , Laryngostenosis , Magnesium , Stents , Animals , Rabbits , Laryngostenosis/pathology , Laryngostenosis/therapy , Inflammation/pathology , Dilatation/instrumentation , Severity of Illness Index
2.
BMJ Case Rep ; 17(6)2024 Jun 04.
Article in English | MEDLINE | ID: mdl-38834313

ABSTRACT

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.


Subject(s)
Laryngostenosis , Respiratory Sounds , Tracheostomy , Humans , Infant, Newborn , Respiratory Sounds/etiology , Laryngostenosis/surgery , Trachea/surgery , Trachea/abnormalities , Male , Intubation, Intratracheal/methods
3.
Eur J Cardiothorac Surg ; 65(6)2024 Jun 03.
Article in English | MEDLINE | ID: mdl-38885366

ABSTRACT

OBJECTIVES: 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.


Subject(s)
Cricoid Cartilage , Laryngostenosis , Trachea , Humans , Male , Female , Retrospective Studies , Laryngostenosis/surgery , Middle Aged , Cricoid Cartilage/surgery , Adult , Aged , Trachea/surgery , Treatment Outcome , Laryngoscopy/methods , Endoscopy/methods , Young Adult , Voice Quality/physiology
4.
Int J Pediatr Otorhinolaryngol ; 181: 111985, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38776721

ABSTRACT

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.


Subject(s)
Costal Cartilage , Cricoid Cartilage , Laryngostenosis , Humans , Cricoid Cartilage/surgery , Male , Infant , Costal Cartilage/transplantation , Female , Laryngostenosis/surgery , Infant, Newborn , Vocal Cord Paralysis/surgery , Treatment Outcome , Endoscopy/methods , Laryngoscopy/methods , Retrospective Studies , Ribs/transplantation , Ribs/surgery
5.
Eur Arch Otorhinolaryngol ; 281(7): 3701-3706, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38705896

ABSTRACT

PURPOSE: An accurate diagnosis and proper treatment plan are required to restore an adequate patent airway in fibrotic subglottic stenosis (SGS). Currently, the definitive treatment entails single-stage balloon dilatation with steroid injections. The primary aim was to evaluate successful airway restoration and general quality of life in cases with SGS in northern Sweden using robust patient reported outcomes. METHODS: All participants with need of surgical treatment due to SGS that had been referred to the department of otorhinolaryngology, University Hospital of Umeå from September 2020 to August 2023 was included. Exclusion criteria included malignant, extrathoracic or cartilaginous cause, age < 18 years, or incompetent to sign consent documents. We assessed the patient-reported outcome measures pre- as well as 3 months postoperatively. RESULTS: Of the 40 cases fulfilling the eligibility criteria's, 33 cases completed the Dyspnea index (DI) and the short form health survey (SF-36) pre- as well as 3 months post-operatively. Receiver operating characteristics showed significant improvement in DI as well as in SF 36 scores post-operatively. CONCLUSIONS: Evaluation of balloon dilatation in SGS in this cohort follow-up analysis shows clear improvement in patient quality of life using robust PROM 3 months postoperatively, ensuring the use of a safe and well-tolerated procedure.


Subject(s)
Dilatation , Dyspnea , Laryngostenosis , Patient Reported Outcome Measures , Quality of Life , Humans , Laryngostenosis/therapy , Male , Female , Dyspnea/etiology , Dyspnea/therapy , Middle Aged , Dilatation/methods , Aged , Adult , Treatment Outcome , Sweden
6.
Laryngorhinootologie ; 103(S 01): S148-S166, 2024 May.
Article in English, German | MEDLINE | ID: mdl-38697146

ABSTRACT

The laryngotracheal junction is an anatomical region with special pathophysiological features. This review presents clinical pictures and malformations that manifest pre-dilectively at this localisation in children and adolescents as well as in adults. The diagnostic procedure is discussed. The possibilities of surgical reconstruction are presented depending on the pathology and age of the patient.


Subject(s)
Larynx , Plastic Surgery Procedures , Trachea , Humans , Trachea/surgery , Trachea/abnormalities , Larynx/surgery , Larynx/abnormalities , Adolescent , Child , Plastic Surgery Procedures/methods , Adult , Laryngostenosis/surgery
7.
BMJ Case Rep ; 17(5)2024 May 06.
Article in English | MEDLINE | ID: mdl-38719265

ABSTRACT

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.


Subject(s)
Aphonia , Laryngoscopy , Laryngostenosis , Respiratory Distress Syndrome, Newborn , Humans , Infant, Newborn , Female , Aphonia/genetics , Aphonia/diagnosis , Laryngostenosis/diagnosis , Laryngostenosis/genetics , Respiratory Distress Syndrome, Newborn/etiology , Respiratory Distress Syndrome, Newborn/diagnosis , Glottis
8.
Am J Otolaryngol ; 45(4): 104323, 2024.
Article in English | MEDLINE | ID: mdl-38677144

ABSTRACT

BACKGROUND: Subglottic stenosis (SGS) is a condition leading to narrowing of the upper airway which can lead to dyspnea and life-threatening airway obstruction. Although other proposed grading systems exist, the Cotton Myer (CM) and percent stenosis systems are the most widespread in clinical practice. Despite this, the CM system has not yet been validated for visual assessment of SGS. OBJECTIVE: To determine the interrater and intrarater reliability of the CM grading system among a cohort of physicians who manage patients with SGS. METHODS: An online survey created with videos of tracheoscopies from 20 adult patients with subglotticstenosis (SGS) was sent individually to 9 expert physicians from various medical specialties, all of whom managed patients with SGS. Physicians were asked to view the 20 tracheoscopy videos and assess both the percent stenosis and Cotton Myer (CM) grade of each patient. After a period of 4 weeks, the physicians were sent the same survey of the 20 tracheoscopy videos. The interrater and intrarater reliability was calculated using the intraclass correlation coefficient (ICC), a measurement used to evaluate the reliability (the extent to which a measurement can be replicated) of two or more raters measuring the same subject. RESULTS: Overall, CM and percent stenosis systems were found to have an ICC of 0.94 and 0.90 within the domain of interrater reliability, respectively, and ICC of 0.71 and 0.81 within the domain of intrarater reliability, respectively. CONCLUSION: Our findings suggest that the CM and percent stenosis grading systems remain a valid clinical tool to measure and communicate the severity of airway obstruction in SGS.


Subject(s)
Laryngostenosis , Severity of Illness Index , Humans , Laryngostenosis/diagnosis , Reproducibility of Results , Observer Variation , Adult , Video Recording , Surveys and Questionnaires , Male , Female
9.
Eur J Cardiothorac Surg ; 65(5)2024 May 03.
Article in English | MEDLINE | ID: mdl-38637945

ABSTRACT

OBJECTIVES: Surgical treatment for airway stenosis necessitates personalized techniques based on the stenosis location and length, leading to favourable surgical outcomes. However, there is limited literature on functional outcomes following laryngotracheal surgery with an adequate number of patients. METHODS: We conducted a retrospective analysis of patients who underwent laryngotracheal surgery at the Department of Thoracic Surgery, Medical University of Vienna, from January 2017 to June 2021. The study included standardized functional assessments before and after surgery, encompassing spirometry, voice measurements, swallowing evaluation and subjective patient perception. RESULTS: The study comprised 45 patients with an average age of 51.9 ± 15.9 years, of whom 89% were female, with idiopathic being the most common aetiology (67%). Procedures included standard cricotracheal resection in 11%, cricotracheal resection with dorsal mucosal flap in 49%, cricotracheal resection with dorsal mucosal flap and lateral cricoplasty in 24% and single-stage laryngotracheal reconstruction in 16%. There were no in-hospital mortalities or restenosis cases during the mean follow-up period of 20.8 ± 13.2 months. Swallowing function remained intact in all patients. Voice evaluations showed a decrease in fundamental vocal pitch [203 (81-290) Hz vs 150 (73-364) Hz, P < 0.001] and dynamic voice range (23.5 ± 5.8 semitones vs 17.8 ± 6.7 semitones, P < 0.001). However, no differences in voice volume were observed (60.0 ± 4.1 dB vs 60.2 ± 4.8 dB, P = 0.788). The overall predicted voice profile changed from R0B0H0 to R1B0H1. CONCLUSIONS: Laryngotracheal surgery proves effective in fully restoring breathing capacity while preserving vocal function. Even in cases of high-grade and complex airway stenosis necessitating laryngotracheal reconstruction, favourable functional outcomes can be achieved.


Subject(s)
Laryngostenosis , Tracheal Stenosis , Humans , Female , Middle Aged , Male , Retrospective Studies , Laryngostenosis/surgery , Tracheal Stenosis/surgery , Adult , Treatment Outcome , Aged , Trachea/surgery , Larynx/surgery , Plastic Surgery Procedures/methods , Deglutition/physiology , Postoperative Period
10.
Eur Arch Otorhinolaryngol ; 281(6): 3083-3093, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38564008

ABSTRACT

PURPOSE: To examine the factors that affect graft healing after laryngotracheal reconstruction (LTR). METHODS: We conducted a retrospective chart review at King Abdulaziz University Hospital, Riyadh, Saudi Arabia, between January-2008 and October-2023. We included all patients who underwent LTR and required anterior and/or posterior graft placement, while those who underwent procedures without graft placement and those with incomplete information were excluded. RESULTS: Forty-nine patients were analyzed. Most patients were pediatric (65.3%), male (65.3%), had no coexisting comorbidities (55.1%), and harbored grade 3-4 stenosis (59.2%). Thirty patients (61.2%) underwent open surgery. Various graft complications occurred including infection (n = 1, 2%), dehiscence (n = 3, 6.1%), scar (n = 6, 12.2%), and granulation (n = 29, 59.2%). Only 15 patients (30.65%) achieved the composite status of "healthy" graft. Among 43 patients who had postoperative cultures, positive results for Pseudomonas aeruginosa and methicillin-resistant Staphylococcus aureus were observed in 10 and four patients, respectively. Open surgery and double-stage procedure were significantly associated with higher rates of granulation tissue formation. Pediatric-age group had significantly higher rate of complete epithelization compared to adult-age group. A significantly greater proportion of patients who had unhealthy grafts had open surgery. The rate of double-stage LTR was significantly higher in unhealthy grafts compared to healthy grafts. Prolonged stent duration was linked to various graft-related complications. Multivariate logistic regression analyses showed no statistically significant correlations between various factors and postoperative graft-related complications. CONCLUSION: Open surgery, double-stage procedure, pediatric age group, and stent duration were not significant risk factors associated with postoperative graft-related complications during LTR in multivariate analysis.


Subject(s)
Laryngostenosis , Plastic Surgery Procedures , Tracheal Stenosis , Humans , Male , Female , Retrospective Studies , Child , Adolescent , Laryngostenosis/surgery , Plastic Surgery Procedures/methods , Tracheal Stenosis/surgery , Adult , Child, Preschool , Wound Healing , Postoperative Complications/epidemiology , Middle Aged , Young Adult , Saudi Arabia/epidemiology , Trachea/surgery , Risk Factors
11.
Article in Chinese | MEDLINE | ID: mdl-38563176

ABSTRACT

Objective:To investigate the effect of acellular dermal matrix in preventing laryngeal stenosis in glottic carcinoma patients. Methods:Fifty-five patients with glottic carcinoma(T2, T3) from February 2018 to December 2022 were divided into experimental group(28 cases) and control group(27 cases) according to their wishes. Acellular dermal matrix was placed in the operation cavity in the experimental group after laryngofission, while control group 12 cases were sutured by pulling the upper and lower edges, 15 cases were repaired with sternohyoid muscle fascia flap. Results:In the experimental group, 1 case had laryngeal stenosis caused by laryngeal mucosa swelling after operation, and extubated successfully after symptomatic treatment. In the control group, 7 cases had laryngeal stenosis after operation, of which 3 cases were caused by granulation tissue hyperplasia in laryngeal cavity, and extubated after symptomatic treatment. 2 cases extubated after operation suffered from progressive dyspnea during radiotherapy, and underwent tracheotomy again, extubation successful after treatment. 2 cases caused by laryngeal mucosa swelling, after symptomatic treatment, one case was successfully extubated, and one case had long-term intubation. The laryngeal stenosis rate of the experimental group was 3.6%(1/28) , which was lower than control group 25.9%(7/27), and the therapeutic effect of the experimental group was significantly better than control group (χ²=5.526, P=0.019). Conclusion:Implanting acellular dermal matrix in the operation cavity of glottic carcinoma can reduce the occurrence of laryngeal stenosis and have satisfactory preventive effect on laryngeal stenosis.


Subject(s)
Acellular Dermis , Carcinoma , Laryngeal Edema , Laryngeal Neoplasms , Laryngostenosis , Humans , Laryngostenosis/prevention & control , Laryngeal Neoplasms/surgery , Surgical Flaps
12.
Otolaryngol Head Neck Surg ; 171(1): 247-253, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38482949

ABSTRACT

OBJECTIVES: Laryngotracheal reconstruction (LTR) has revolutionized the management of pediatric subglottic stenosis (SGS). However, postoperative stenosis remains a difficult hurdle to overcome. Our goal is to determine the clinical impact of recalcitrant stenosis after LTR and the factors contributing to postoperative stenosis. STUDY DESIGN: Retrospective review of 181 open LTR patients between 2008 and 2021. SETTING: Stand-alone tertiary children's hospital. METHODS: Recalcitrant stenosis was defined as new or worsening stenosis despite open LTR. Fisher's exact and Mann-Whitney tests were used to detect differences in categorical and continuous clinical data between patients with and without treatment-resistant stenosis. Time-to-decannulation analysis of both groups was performed using Kaplan-Meier analysis and evaluated with log-rank and Cox proportional hazards regression. Multivariate logistical regression was used to assess the validity of associations found in univariate analysis. RESULTS: As expected, the 27 patients with postoperative stenosis were less likely to be decannulated (P < .001, Fisher's Exact), more likely to require a postoperative tracheostomy (P < .001, Fisher's Exact) or revision LTR (P < .001, Fisher's Exact) and had prolonged time to decannulation (P < .001, Log-rank). Children with Grade IV SGS (P = .004, Fisher's Exact), and those with longer suprastomal stent duration (P = .03, Fisher's Exact) were more likely to suffer from recalcitrant stenosis. Stent duration longer than 4 weeks (P = .01) contributed to refractory stenosis when controlling for all aforementioned variables using multivariable logistic regression. Interposition grafts had a protective effect (P = .005). CONCLUSION: Maintaining suprastomal stents over 4 weeks after LTR increases the risk for postoperative stenosis and its sequelae.


Subject(s)
Laryngostenosis , Postoperative Complications , Humans , Retrospective Studies , Male , Female , Laryngostenosis/surgery , Laryngostenosis/etiology , Child, Preschool , Infant , Child , Tracheal Stenosis/surgery , Tracheal Stenosis/etiology , Plastic Surgery Procedures/methods , Risk Factors , Tracheostomy
13.
Otolaryngol Head Neck Surg ; 171(1): 180-187, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38482973

ABSTRACT

OBJECTIVE: To report on a series of patients with cANCA/PR3-positive, granulomatosis with polyangiitis (GPA)-associated subglottic stenosis (SGS) and evaluate response to medical maintenance therapy with rituximab versus other immunosuppressants following initial endoscopic laser excision. STUDY DESIGN: Retrospective chart review. SETTING: Tertiary academic center. METHODS: A retrospective chart review of patients with SGS and cANCA/PR3-positive GPA who received immunosuppressive maintenance therapy following endoscopic laser excision at our institution from June 1989 to March 2020 was performed. Data pertaining to patient demographics, clinical features, medications, and endoscopic laser procedures were collected. RESULTS: A total of 27 patients (15 women) with mean age (range) of 40 (19-59) years and mean (range) follow-up of 12.6 years (1.5-28.6) were identified. Sixteen patients (60%) had limited GPA. Six patients (24%) had previously received local intervention with open surgery (n = 1, 4%) or endoscopic techniques (n = 5, 20%). All patients experienced symptom improvement following initial CO2 laser excision at our institution without any procedural complications or adverse events. Following initial laser excision, 15 patients (60%) were treated with rituximab and 10 patients (40%) were treated with nonrituximab immunosuppressive agents. Patients treated with rituximab were less likely to recur (P = 0.040). Limited GPA was associated with an increased incidence of recurrence (P = 0.031). Median time (years) to recurrence (range) was 3.2 (0.3-19.3) and was not significantly associated with treatment or GPA subtype. CONCLUSION: Endoscopic CO2 laser excision is a safe and effective local intervention for GPA-associated SGS. Medical maintenance therapy with rituximab reduces risk of recurrence following initial laser excision relative to treatment with non-rituximab agents.


Subject(s)
Granulomatosis with Polyangiitis , Immunosuppressive Agents , Laryngostenosis , Laser Therapy , Rituximab , Humans , Granulomatosis with Polyangiitis/complications , Granulomatosis with Polyangiitis/drug therapy , Female , Male , Laryngostenosis/surgery , Laryngostenosis/etiology , Adult , Retrospective Studies , Middle Aged , Rituximab/therapeutic use , Laser Therapy/methods , Immunosuppressive Agents/therapeutic use , Treatment Outcome , Young Adult , Laryngoscopy , Maintenance Chemotherapy
14.
J Pediatr Surg ; 59(6): 1066-1071, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38429129

ABSTRACT

BACKGROUND: Airway anomalies, symptoms and interventions are commonly reported in children with oesophageal atresia with tracheoesophageal fistula (OA/TOF). The purpose of this study was to assess the incidence of these airway pathologies and those requiring interventions in the long-term. METHODS: A retrospective case note review of all patients admitted to the Neonatal Unit at the Royal Hospital for Children, Glasgow between January 2000 and December 2015 diagnosed with OA/TOF. Included patients had a minimum of 5 years follow-up. RESULTS: 121 patients were identified. 118 proceeded to OA/TOF repair. 115 patients had long-term follow-up data. Ninety-five (83%) children had one or more airway symptom recorded. Thirty-six (31%) neonates underwent airway endoscopy at the time of their initial OA/TOF repair. Forty-six (40%) children underwent airway endoscopy at a later date due to airway symptoms. Airway pathologies identified included airway malacia, thirty-two (28%), subglottic stenosis, eleven (10%), tracheal pouch, twenty-five (22%), laryngeal cleft, seven (6%) and recurrent fistula, five (4%). Airway interventions included endoscopic division of tracheal pouch, ten (9%), tracheostomy, seven (6%), aortopexy, six (5%), repair of recurrent fistula, five (4%), endoscopic repair of laryngeal cleft, three (3%) and four (3%) required open airway reconstruction for subglottic stenosis. One child (1%) remains tracheostomy dependent. CONCLUSIONS: Long-term airway pathologies are common in children with OA/TOF. Many of these are remediable with surgical intervention. Clinicians should be cognisant of this and refer to Airway Services appropriately.


Subject(s)
Esophageal Atresia , Tracheoesophageal Fistula , Humans , Tracheoesophageal Fistula/surgery , Tracheoesophageal Fistula/complications , Esophageal Atresia/surgery , Esophageal Atresia/complications , Retrospective Studies , Infant, Newborn , Male , Female , Follow-Up Studies , Infant , Treatment Outcome , Child, Preschool , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Laryngostenosis/surgery , Recurrence , Larynx/abnormalities , Larynx/surgery , Congenital Abnormalities
15.
Laryngoscope ; 134(7): 3260-3266, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38501339

ABSTRACT

OBJECTIVES: A small number of Idiopathic subglottic stenosis (iSGS) patients are treated at institutions across the country. Divergence in operative techniques for endoscopic dilation (ED) of iSGS has been anecdotally recognized but not formally characterized. Additionally, the relationship between procedural variation and clinical outcome has not been studied. METHODS: Secondary analysis of the NoAAC iSGS1000 cohort investigated variation in procedural techniques and treatment outcomes in patients treated with ED across high-enrolling treatment centers (enrolled >10 patients in PR-02 trial). RESULTS: Thirteen NoAAC centers each enrolled >10 patients treated with ED for a total of 281 subjects. There was significant variation in procedural details and rate of recurrence among institutions. Hierarchal cluster analysis revealed significant heterogeneity among institutions and clusters in all procedural variables. However, analysis demonstrated a transient delay in disease recurrence in cluster 2 which disappeared with longer longitudinal follow-up. Patient-reported outcome and peak expiratory flow data supported the potential benefit of the technical variation in Cluster 2. Distinct to cluster 2, however, was routine use of adjuvant triple medical therapy (proton pump inhibitor (PPI), antibacterial agent, and steroid inhaler). CONCLUSIONS: Both outcome and procedural technique vary among centers employing ED to treat iSGS. A transient delay in recurrence was observed among centers that routinely prescribed adjuvant medical therapy (antibiotic, inhaled corticosteroid, and PPI) to iSGS patients after endoscopic dilation, which was further supported by patient-reported data and peak expiratory flow data. Prospective studies are needed to understand the effects of adjuvant medical therapy on recurrence after endoscopic dilation. LEVEL OF EVIDENCE: 4 Laryngoscope, 134:3260-3266, 2024.


Subject(s)
Dilatation , Laryngostenosis , Humans , Laryngostenosis/surgery , Laryngostenosis/therapy , Dilatation/methods , Male , Female , Middle Aged , Treatment Outcome , Laryngoscopy/methods , Adult , Recurrence , Aged
16.
Laryngoscope ; 134(7): 3245-3252, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38450771

ABSTRACT

OBJECTIVES: Recent immunologic study of the adaptive immune repertoire in the subglottic airway demonstrated high-frequency T cell clones that do not overlap between individuals. However, the anatomic distribution and antigenic target of the T cell repertoire in the proximal airway mucosa remain unresolved. METHODS: Single-cell RNA sequencing of matched scar and unaffected mucosa from idiopathic subglottic stenosis patients (iSGS, n = 32) was performed and compared with airway mucosa from healthy controls (n = 10). T cell receptor (TCR) sequences were interrogated via similarity network analysis to explore antigenic targets using the published algorithm: Grouping of Lymphocyte Interactions by Paratope Hotspots (GLIPH2). RESULTS: The mucosal T cell repertoire in healthy control airways consisted of highly expressed T cell clones conserved across anatomic subsites (trachea, bronchi, bronchioles, and lung). In iSGS, high-frequency clones were equally represented in both scar and adjacent non-scar tissue. Significant differences in repertoire structure between iSGS scar and unaffected mucosa was observed, driven by unique low-frequency clones. GLIPH2 results suggest low-frequency clones share targets between multiple iSGS patients. CONCLUSION: Healthy airway mucosa has a highly conserved T cell repertoire across multiple anatomic subsites. Similarly, iSGS patients have highly expressed T cell clones present in both scar and unaffected mucosa. iSGS airway scar possesses an abundance of less highly expanded clones with predicted antigen targets shared between patients. Interrogation of these shared motifs suggests abundant adaptive immunity to viral targets in iSGS airway scar. These results provide insight into disease pathogenesis and illuminate new treatment strategies in iSGS. LEVEL OF EVIDENCE: NA Laryngoscope, 134:3245-3252, 2024.


Subject(s)
Adaptive Immunity , Laryngostenosis , Humans , Adaptive Immunity/immunology , Male , Female , Laryngostenosis/immunology , Middle Aged , T-Lymphocytes/immunology , Adult , Case-Control Studies , Receptors, Antigen, T-Cell/immunology , Receptors, Antigen, T-Cell/genetics , Respiratory Mucosa/immunology , Aged , Single-Cell Analysis
17.
Ann Otol Rhinol Laryngol ; 133(6): 618-624, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38444374

ABSTRACT

OBJECTIVES: The management of idiopathic subglottic stenosis (iSGS) poses a clinical challenge due to high recurrence rates following both endoscopic and open approaches, often leading to tracheostomy. The activation of abnormal T-cells and cytokine pathways has been linked to iSGS pathogenesis. Autologous adipose tissue centrifugation yields lipoaspirate, offering optimal anti-inflammatory effects and biocompatibility widely utilized in various medical settings. This report presents the first 3 cases employing endoscopic dilation (ED) in combination with local lipoaspirate injection to address recurrent iSGS. METHODS: A prospective observational study was conducted, involving multidisciplinary evaluation by the Tracheal Team at the University of Modena. Patients meeting specific criteria were directed to undergo ED + lipoaspirate injection. RESULTS: Three patients fulfilled the inclusion criteria. The mean number of prior endoscopic procedures performed was 8. Endoscopic examination revealed 90% stenosis in patient A, 60% stenosis in patient B, and 60% stenosis in patient C. All patients presented inflammatory tissue or incipient granulations at the stenotic site, with an average time of 6 months between previous procedures. After 15 months, none of the patients required further procedures, and endoscopic examination revealed a significant reduction or disappearance of inflammatory tissue with a stable airway lumen. CONCLUSIONS: The observed results are encouraging in terms of reducing local inflammation and halting stenosis progression, especially in cases of short-term relapsing iSGS.


Subject(s)
Adipose Tissue , Laryngostenosis , Recurrence , Humans , Laryngostenosis/surgery , Laryngostenosis/etiology , Male , Adipose Tissue/transplantation , Prospective Studies , Female , Middle Aged , Dilatation/methods , Adult , Laryngoscopy/methods , Treatment Outcome
18.
JAMA Otolaryngol Head Neck Surg ; 150(5): 368-377, 2024 May 01.
Article in English | MEDLINE | ID: mdl-38483372

ABSTRACT

Importance: Patients with induced laryngeal obstruction (ILO) present with a variety of behavioral health profiles. Identifying these profiles is crucial in that behavioral health conditions may affect treatment duration and outcomes. Objective: To characterize the prevalence of anxiety, depression, posttraumatic stress disorder (PTSD), and physical somatic symptoms in adult and pediatric patients with ILO and determine the factors associated with anxiety, depression, PTSD, and physical somatic symptoms in patients with ILO? Design, Setting, and Participants: This cross-sectional study included a nonprobability sample of 83 adult and 81 pediatric patients diagnosed with ILO at outpatient adult and pediatric otolaryngology clinics between 2021 and 2023. Exclusion criteria included a comorbid respiratory diagnosis other than asthma, head or neck cancer, or neurological impairments. Recruitment took place between September 2021 and March 2023. The analyses were run in January 2024. Main Outcome Measures: Patients were prospectively screened for anxiety, depression, PTSD, and somatic physical symptoms. In addition, any past behavioral health diagnoses were extracted from the medical record. Comorbidities, ILO symptoms triggers, and onset details were gathered from ILO evaluations. Adult patients completed the Screen for Adult Anxiety Related Disorders (SCAARED), depression (Patient Health Questionnaire [PHQ]-9), and somatic physical symptoms portions of the Patient Health Questionnaires (PHQ-15), and the PTSD Checklist for the Diagnostic and Statistical Manual of Mental Disorders (Fifth Edition) (PCL-5). Pediatric patients completed the Screen for Child Anxiety Related Disorders (SCARED), depression (PHQ-9A) and somatic physical symptoms portions of the Patient Health Questionnaires for Adolescents (PHQ-15A), and the UCLA PTSD Reaction Index brief screeners. Results: Eighty-three adult patients participated in this study (mean [SD] age, 45.8 [14.3] years; 64 female, 19 male). Eighty-one pediatric patients participated (mean [SD] age, 13.83 [2.55] years; 67 female, 14 male). Adult and pediatric patients with ILO screened positive for elevated rates of anxiety (53 adults [63%]; 49 children [60%]), depression (27 adults [32%]; 25 children [30%]), and PTSD (29 adults [34%]; 13 children [16%]). Most of the patients with anxiety and depression symptoms were formally diagnosed prior to ILO evaluation, with rates of previously diagnosed anxiety, depression, and PTSD also above published norms. Adults were twice as likely as children to present with PTSD (odds ratio, 2.1; 95% CI, 0.05-4.48). Elevated rates of physical somatic symptoms were also evident, with 38 adults (45%) and 32 children (39%) scoring in the moderate to severe range. Conclusions and Relevance: This study found high rates of adult and pediatric patients with ILO screened positive for anxiety, depression, and PTSD symptoms. Future work should investigate how behavioral health and ILO treatments can best be coordinated to maximize treatment outcomes.


Subject(s)
Anxiety , Depression , Stress Disorders, Post-Traumatic , Humans , Male , Female , Cross-Sectional Studies , Stress Disorders, Post-Traumatic/epidemiology , Adult , Depression/epidemiology , Depression/etiology , Child , Middle Aged , Anxiety/epidemiology , Anxiety/etiology , Adolescent , Prevalence , Laryngostenosis/psychology , Laryngostenosis/epidemiology , Aged
19.
Vestn Otorinolaringol ; 89(1): 10-15, 2024.
Article in Russian | MEDLINE | ID: mdl-38506019

ABSTRACT

OBJECTIVE: To study the efficacy and safety of balloon dilation as the first choice method in the treatment of children of the first year of life with acquired subglottic stenosis. MATERIAL AND METHODS: A retrospective analysis of the treatment of 25 patients aged 27 days to 11 months of life (average age 5.3±3.76 months) with subglottic stenosis caused by prolonged intubation, in whom balloon dilation was the first method of treatment. Grade III Cotton-Myer stenosis was preoperatively detected in 22 children, the remaining 3 had grade II stenosis. RESULTS: The success rate of balloon dilation was 100%; tracheotomy was not required in any case, the absence of stenosis during a follow-up examination in the catamnesis was recorded in 14 (56%) children, the remaining 11 (44%) had grade 0-I stenosis and did not cause respiratory disorders. In 1 child (1.5 years old), a subglottic cyst was removed after balloon dilation. One dilation was required in 18 (72%) children, two - in 5 (20%), three and four - respectively for 1 patient. If additional intervention was necessary, the operation was repeated 10 days - 3 months after the previous one. There were no postoperative complications. CONCLUSION: Balloon dilation is a highly effective and safe alternative to traditional surgical interventions for acquired subglottic stenosis in children of the first year of life and can be recommended as a method of first choice.


Subject(s)
Laryngostenosis , Child , Humans , Infant , Laryngostenosis/diagnosis , Laryngostenosis/etiology , Laryngostenosis/surgery , Constriction, Pathologic/complications , Constriction, Pathologic/surgery , Tracheotomy/adverse effects , Retrospective Studies , Dilatation/adverse effects , Dilatation/methods , Treatment Outcome
20.
Zentralbl Chir ; 149(3): 308-314, 2024 Jun.
Article in German | MEDLINE | ID: mdl-38359868

ABSTRACT

Idiopathic subglottic stenosis is a circular scarred narrowing of the airway at the transition from the cricoid cartilage to the trachea. The stenosis is found radiologically and endoscopically at the level of the cricoid cartilage without involvement of the cricoid or tracheal cartilage itself. The disease practically only affects women between the ages of 20 and 60. The same clinical picture occurs in granulomatosis with polyangiitis and less frequently in other autoimmune diseases, where it requires systemic treatment. The clinical picture usually begins insidiously with coughing and sputum production and leads to dyspnoea and a restricted cough. As the course is insidious and the patients are otherwise healthy, the symptoms are often misinterpreted and the diagnosis is delayed. Treatment consists of local measures, ranging from dilatation and laser surgical resection, sometimes with local application of medication to inhibit the proliferation of new scar tissue, to laryngotracheal resection of varying degrees. The disease is located in the border area between the trachea and larynx and the patients are therefore treated by ENT medicine, pneumology and thoracic surgery.


Subject(s)
Laryngostenosis , Tracheal Stenosis , Humans , Tracheal Stenosis/surgery , Tracheal Stenosis/etiology , Tracheal Stenosis/diagnosis , Laryngostenosis/etiology , Laryngostenosis/surgery , Laryngostenosis/diagnosis , Laryngostenosis/pathology , Female , Middle Aged , Adult , Diagnosis, Differential , Laser Therapy , Male , Dilatation , Laryngoscopy , Tomography, X-Ray Computed
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