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1.
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
2.
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
3.
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
4.
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
5.
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
6.
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
7.
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
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 Arch Otorhinolaryngol ; 281(8): 3977-3984, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38578502

ABSTRACT

OBJECTIVES: The primary goal of this study was to systematically identify all relevant published articles on the use of primary endoscopic balloon dilation for the management of pediatric patients with subglottic stenosis, critically assess the technique's success, and determine which patients are the best candidates for the procedure. METHODOLOGY: This was a systematic review and meta-analysis that aimed to investigate the use and success rate of EBD for treating pediatric SGS. An electronic systematic literature search of three major databases, PubMed, EBSCO, and Web of Science&MEDLINE through Clarivate, was conducted to include the eligible articles. RESULTS: A total of 14 unique studies were included in the final analysis, with 473 cases of pediatric SGS. The pooled success rate of EBD in treatment of pediatric SGS was 76% (k = 14 studies, 95% confidence interval [CI] = 0.65-0.86, P < 0.001, Q test for heterogeneity = 0.03, P < .001, I2 = 91%). CONCLUSIONS: We reported a high success rate of EBD in treating pediatric SGS. The reported complications were uncommon, although they can be serious and life threatening. The intensity of SGS may be related to the likelihood of therapy failure.


Subject(s)
Dilatation , Laryngoscopy , Laryngostenosis , Humans , Laryngostenosis/therapy , Child , Dilatation/methods , Dilatation/instrumentation , Laryngoscopy/methods , Treatment Outcome
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.
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
13.
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
14.
Otolaryngol Head Neck Surg ; 171(2): 486-493, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38591708

ABSTRACT

OBJECTIVE: Bilateral vocal fold paralysis (BVFP) and posterior glottic stenosis (PGS) are causes of bilateral vocal fold immobility (BVFI) and may cause shortness of breath, stridor, and need for surgical intervention. Although increased body mass index (BMI) is associated with restrictive breathing patterns in patients with normal upper airways, it is unclear how BMI impacts dyspnea and need for surgical intervention in BVFI patients. STUDY DESIGN: Retrospective cohort study. SETTING: Three tertiary academic centers in the United States. METHODS: Demographics, BMI, Dyspnea Index (DI), etiology, presence of tracheostomy and surgical intervention (dilation, tracheostomy, cordotomy, arytenoidectomy, open reconstruction) were collected. Primary outcomes included dyspnea measured by DI and need for surgery to improve airway. Linear regressions were performed to assess continuous outcomes. Mann-Whitney U-test was utilized to assess categorical outcomes. RESULTS: Among 121 patients, 52 presented with BVFP and 69 with PGS. Previous neck surgery was the most common cause of BVFI (40.2%). 44.3% of patients received a tracheostomy. Through multivariate linear regression, increased BMI was significantly associated with increased DI in the entire cohort (ß = .43, P = .016). Increased BMI was also associated with need for any surgical intervention (odds ratio [OR] = 1.07, 95% confidence interval [CI] = [1.01-1.13]) in the overall cohort. When stratifying our data, BMI was only significantly associated with DI in BVFP (ß = .496) and need for surgical intervention in PGS (OR = 1.11, 95% CI = [1.01-1.21]), although a positive trend was seen in all analyses. CONCLUSION: Increased BMI may correlate with worsening dyspnea symptoms and need for surgical intervention in patients with BVFI. Weight-loss-related counseling may benefit symptom management.


Subject(s)
Body Mass Index , Dyspnea , Vocal Cord Paralysis , Humans , Male , Vocal Cord Paralysis/surgery , Vocal Cord Paralysis/etiology , Female , Dyspnea/etiology , Dyspnea/surgery , Retrospective Studies , Middle Aged , Aged , Adult , Laryngostenosis/surgery , Tracheostomy
15.
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
16.
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
17.
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
18.
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
19.
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
20.
Otolaryngol Head Neck Surg ; 171(2): 471-477, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38509830

ABSTRACT

OBJECTIVE: Endoscopic management of subglottic stenosis (SGS) includes a wide range of techniques. This 17-year review compares treatment outcomes between carbon dioxide (CO2) laser and cold steel. STUDY DESIGN: Retrospective chart review. SETTING: Single tertiary care center. METHODS: A chart review was performed for all patients undergoing endoscopic treatment of SGS at Cleveland Clinic between July 12, 2000 and September 1, 2017. Data collected included demographics, stenosis etiology, stenosis severity, comorbidities, treatment modality, and airway procedure history. The primary endpoint was repeated treatment-free survival (RTFS) within 2 years using a Kaplan-Meier analysis and Cox proportional hazard model. RESULTS: A total of 139 patients (median [interquartile range] aged 48.7 [37.8, 57.0] years; 83.4% female) were included in the analysis, with etiologies including idiopathic (56.8%), granulomatosis with polyangiitis (25.2%), and intubation (16.5%). All patients underwent either cold steel (107 patients) or CO2 laser (32 patients) lysis of stenosis with concurrent dilation. RTFS within 2 years was 50.2% for CO2 laser and 31.9% for cold steel (hazard ratio [HR] and 95% confidence interval [CI]: 1.69, 0.96-2.97, P = .07). In patients with no prior airway procedures, there was no difference in RTFS between laser and cold knife (P = .41). However, in patients with prior airway procedures, RTFS was significantly greater in the laser group, even after adjusting for age, smoking history, and stenosis etiology (50.0% vs 16.8%, adjusted HR and CI: 2.82, 1.14-6.98, P = .025). CONCLUSION: Endoscopic lysis of SGS with CO2 laser should be considered in revision cases.


Subject(s)
Laryngostenosis , Lasers, Gas , Humans , Laryngostenosis/surgery , Retrospective Studies , Female , Middle Aged , Male , Lasers, Gas/therapeutic use , Adult , Treatment Outcome , Laryngoscopy/methods , Laser Therapy/methods
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