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Tracheobronchopathia osteochondroplastica (TO) is an uncommon non-malignant tracheal ailment characterized by the existence of numerous bone and/or cartilage nodules in the submucosa of the trachea and bronchi. At present, there is a lack of standardized treatment recommendations for TO. This article presents two instances of severe tracheal stenosis caused by TO, which were effectively managed through the implementation of bronchoscopic stent placement and rigid bronchoscopy curettage, respectively. Both cases exhibited favorable outcomes, providing novel insights and references for the treatment of TO. Laryngoscope, 134:3093-3095, 2024., (© 2024 The American Laryngological, Rhinological and Otological Society, Inc.)

Objectives: To characterize tracheal cartilage morphology in mouse models of fibroblast growth factor receptor (Fgfr2)-related craniosynostosis syndromes. To establish relationships between specific Fgfr2 mutations and tracheal cartilaginous sleeve (TCS) phenotypes in these mouse models., Methods: Postnatal day 0 knock-in mouse lines with disease-specific genetic variations in the Fgfr2 gene (Fgfr2 C342Y/C342Y , Fgfr2 C342Y/+ , Fgfr2 +/Y394C , Fgfr2 +/S252W , and Fgfr2 +/P253R ) as well as line-specific controls were utilized. Tracheal cartilage morphology as measured by gross analyses, microcomputed tomography (μCT), and histopathology were compared using Chi-squared and single-factor analysis of variance statistical tests., Results: A greater proportion of rings per trachea were abnormal in Fgfr2 C342Y/+ tracheas (63%) than Fgfr2 +/S252W (17%), Fgfr2 +/P253R (17%), Fgfr2 +/Y394C (12%), and controls (10%) (P < .001 for each vs. Fgfr2 C342Y/+ ). TCS segments were found only in Fgfr2 C342Y/C342Y (100%) and Fgfr2 C342Y/+ (72%) tracheas. Cricoid and first-tracheal ring fusion was noted in all Fgfr2 C342Y/C342Y and 94% of Fgfr2 C342Y/+ samples. The Fgfr2 C342Y/C342Y and Fgfr2 C342Y/+ groups were found to have greater areas and volumes of cartilage than other lines on gross analysis and μCT. Histologic analyses confirmed TCS among the Fgfr2 C342Y/C342Y and Fgfr2 C342Y/+ groups, without appreciable differences in cartilage morphology, cell size, or density; no histologic differences were observed among other Fgfr2 lines compared to controls., Conclusion: This study found TCS phenotypes only in the Fgfr2 C342Y mouse lines. These lines also had increased tracheal cartilage compared to other mutant lines and controls. These data support further study of the Fgfr2 mouse lines and the investigation of other Fgfr2 variants to better understand their role in tracheal development and TCS formation., Level of Evidence: NA Laryngoscope, 131:E1349-E1356, 2021., (© 2020 American Laryngological, Rhinological and Otological Society Inc, "The Triological Society" and American Laryngological Association (ALA).)

Objectives: Awake Flexible Tracheobronchoscopy (FTB) is an alternative to rigid bronchoscopy or sedated flexible bronchoscopy and allows an awake examination of the tracheobronchial tree. We hypothesized that the ability to perform office bronchoscopy as the need arises during a clinic visit would lead to a high rate of previously undiagnosed and clinically relevant findings. This study reports the rate and nature of such findings for this procedure at our institution., Study Design: Retrospective chart review., Methods: The records of 127 adult patients evaluated at the voice and swallowing disorders clinic between June of 2012 and January of 2015 were reviewed. New findings were defined as new pathology visualized during FTB exam that was not previously diagnosed by means of other diagnostic modalities., Results: A total of 233 scope procedures (84 transnasal bronchoscopies and 149 tracheoscopies) were reviewed, 232 of which were completed and one of which was incomplete due to severe subglottic stenosis. New, clinically relevant findings were seen in 57% of transnasal bronchoscopies (48 of 84) and 21% of tracheoscopies (32 of 149). All of these findings provided additional information directing workup or resulted in a change in patient management., Conclusion: Office-based evaluation of the tracheobronchial tree yields a high rate of new findings. In our study, office bronchoscopy had a 57% rate of new findings and was performed without complications. The utility of tracheoscopy was also apparent in its ability to quickly and safely examine the trachea, with a 21% rate of new findings., Level of Evidence: 4. Laryngoscope, 127:1376-1380, 2017., (© 2016 The American Laryngological, Rhinological and Otological Society, Inc.)

Tracheobronchopathia osteochondroplastica (TBOC) is a rare disease of the trachea and bronchi characterized by submucosal nodules of osseous and cartilaginous tissue. In this series, we present three cases highlighting the varied clinical presentations of this rare disease process, which ranged from a rough voice to a chronic cough to lobar pneumonia. The disorder may mimic other lesions. We review the clinical presentations, pathophysiology, lab tests, imaging, diagnosis, and management of TBOC patients. Laryngoscope, 126:2006-2009, 2016., (© 2015 The American Laryngological, Rhinological and Otological Society, Inc.)

Reconstruction of a tracheal defect is a challenge because it often requires invasive surgery associated with relatively high morbidity. We recently invented a less-invasive method using a modified infrahyoid myocutaneous (IHMC) flap for the reconstruction of a tracheal defect in an 83-year-old male. A tracheal defect, the right half of the cricoid cartilage plus the right three quarters of the I-IV tracheal cartilage (about 3 × 4 cm), was reconstructed with a modified IHMC flap composed of the sternohyoid and platysma muscles and a skin pedicle. Considering the age of patient, we avoided rigid reconstruction and used a soft silicone tracheal opening retainer (Koken Co., Ltd., Tokyo, Japan) as an anterior wall dilator after surgery and waited for the scarring of the flap until it become rigid enough. The postoperative course was uneventful and the trachea was reconstructed safely. Tracheal reconstruction with an IHMC flap is a useful and less-invasive alternative compared to end-to-end anastomosis or reconstruction with a forearm flap, which is currently used as a mainstay., (Copyright © 2012 The American Laryngological, Rhinological, and Otological Society, Inc.)

A tracheocele is a rarely encountered entity that may be congenital or acquired. This tracheal lesion is characterized by the presence of a single cystic pouch filled with air or a mixture of liquid and air. We recently managed a case of a large voluminous acquired tracheocele originating from the right posterolateral tracheal wall. A 39-year-old male patient presented with chronic cough and breathy voice. Rigid laryngoscopy revealed a right immobile vocal fold. Computed tomography scan revealed a tracheocele that was excised externally with recurrent laryngeal nerve preservation., (Copyright © 2011 The American Laryngological, Rhinological, and Otological Society, Inc.)

Educational Objectives: Describe the clinical presentation and management of tracheoceles., Study Design: Retrospective case review and review of the medical literature., Results: A 56 year old female patient presented with progressive dysphonia and history of benign thyroid nodules. Flexible laryngoscopy revealed a severely paretic right true vocal cord. Computed tomography revealed a right sided air filled sac in the tracheoesophageal groove suspicious for causing compression of the right recurrent laryngeal nerve. The patient underwent an elective right hemithyroidectomy and resection of the air filled sac. Post operative pathology of the air filled sax was consistent with a tracheocele. A laryngoscopy performed 6 months postoperatively demonstrated recovery of right cord function., Conclusions: Tracheoceles are an uncommon entity first described in 1846 by Rokitansky (1) with only a paucity of case reports in the literature describing the surgical management and treatment of this disease. Herein, we report a patient who presented with dysphonia from a tracheocele in the right tracheoesophageal groove who subsequently underwent surgical resection ultimately improving vocal cord function.

Objectives: To compare the clinical course of tracheal, bronchial, and pulmonary papillomatosis with clinical course of laryngeal papillomatosis in children., Study Design: The records of the 448 children with recurrent respiratory papillomatosis treated in St. Vladimir Moscow Children's Hospital between 1988 and 2003 were reviewed. In all cases, the diagnosis was confirmed histologically. Age at onset of symptoms, age at first surgery, number of surgical procedures, mean duration of surgical interval, possible causes, and age at a point of papillomatosis spread in the lower airways and course of the disease were analyzed., Setting: Academic children's hospital., Results: Papillomas extension down to lower airways was observed in 40 children (8.9%). Among 40 patients with lower airway recurrent respiratory papillomatosis, 8 (20%) demonstrated pulmonary involvement. The basic cause of papilloma extension to lower airways appeared to be tracheotomy performed in children with laryngeal papillomatosis (92.5% of cases). Incidence of satellite pharyngeal and esophageal papillomatosis is significantly higher in patients with lower airways papillomatosis, presenting evidence of lager process extension. The clinical course of lower airways papillomatosis is more aggressive as compared with laryngeal papillomatosis, and treatment efficacy in such children is lower., Conclusions: All the patients with laryngeal papillomatosis having a history of tracheotomy require a regular endoscopic control and chest radiographs or computed tomography scanning because tracheal or pulmonary papillomatosis may occur in such patients even several years after decannulation. The prognosis for the disease after development of pulmonary papillomatosis is always serious.

Objectives/hypothesis: Spontaneous rupture of the trachea or subglottis as a complication of difficult delivery has not been reported in the United States literature. There have been a few cases reported in the European literature. The present report describes a series of newborns with this complication and discusses the signs and treatment options of this difficult, life-threatening problem., Study Design: Retrospective review., Methods: Newborns born between 1996 and 2001 who were treated for spontaneous subglottic or tracheal rupture at a tertiary care children's hospital neonatal intensive care unit were reviewed., Results: Four cases of spontaneous subglottic rupture were seen at the hospital. In three of the four cases the tracheas were intubated on an emergency basis after subcutaneous air was noted in the anterior aspect of the neck. In the fourth patient the trachea was not intubated until the subglottic tear was visualized intraoperatively. Two of the four patients died. One died without securing of an airway; the other died of complications of prolonged hypoxia. Eight cases from European literature of spontaneous neonatal subglottic and tracheal tears are reviewed and are compared with the cases presented in the current report., Conclusions: Early detection of airway rupture by flexible endoscopy is essential for timely diagnosis and appropriate treatment. Standard endotracheal intubation can exacerbate the problem and should be deferred if possible until direct airway visualization can be accomplished. Signs associated with tracheal tears include subcutaneous emphysema, respiratory distress, pneumothorax, and pneumomediastinum. These should lead to emergent consultation with otolaryngologists for examination and securing of the airway.

Objectives/hypothesis: To review the clinical presentation and diagnostic evaluation of patients with symptomatic congenital vascular anomalies causing tracheobronchial compression and to establish the short- and long-term results of surgical intervention with respect to postoperative complications, persistent symptoms, and ventilator and tracheostomy dependence., Study Design: Retrospective review., Methods: Chart review and telephone follow-up., Results: Between 1987 and 1996, 35 children underwent surgical intervention to relieve symptomatic tracheobronchial compression resulting from a congenital vascular anomaly. Historically, the onset of symptoms occurs within the first months of life; however, only 12 (34%) of patients were diagnosed by 6 months of age and 13 (37%) were diagnosed at greater than 1 year of age. Excluding anomalous innominate artery, chest radiography or barium swallow was suggestive of a congenital vascular anomaly in 30 (94%) of the patients. Magnetic resonance imaging correctly delineated the anatomy of the vascular anomaly in 29 patients. Bronchoscopy was diagnostic in all three patients with anomalous innominate arteries causing tracheal compression. Postoperative follow-up was obtained in 32 (91%) of patients; 25 (78%) of these were asymptomatic at the time of their most recent examination. The remaining patients had persistent stridor, recurrent respiratory tract infections, and/or chronic cough. In all three patients who underwent postoperative bronchoscopy for persistent symptoms, tracheomalacia was demonstrated in the region of previous compression., Conclusions: Tracheobronchial compression from congenital vascular anomalies is a rare but treatable cause of respiratory symptoms. Early diagnosis requires a prompt, thorough clinical and radiologic evaluation. Surgery affords excellent long-term resolution of symptoms.

Magnetic resonance imaging (MRI) of the airway has recently emerged as a diagnostic tool for evaluating tracheobronchial obstruction in pediatric patients. Computer-assisted reconstructions of transaxial images have enabled three-dimensional reconstructions of the airway to be manipulated in any plane and visualized in relation to adjacent anatomical structures. Eight patients in whom magnetic resonance imaging was used to diagnose a variety of airway obstructive lesions are presented. A protocol for the evaluation of pediatric tracheobronchial obstruction is presented, with the role of three dimensionally reconstructed MR imaging emphasized.