Tracheobronchopathia osteochondroplastica concurrent with peripheral lung cancer: a case report and perioperative considerations.

Background: Tracheobronchopathia osteochondroplastica (TPO) is a rare, benign, chronic disorder of unknown etiology. It is characterized by submucosal nodules, often calcified, which predominantly affect the anterolateral aspects of the trachea and main bronchi, while sparing the posterior bronchial wall. The co-occurrence of TPO and lung cancer is exceedingly rare. This report presents a case of TPO association with early-stage lung cancer, which was managed through surgical intervention. No active treatment was undertaken for the TPO. Case Description: A patient presented with a nodule in the right upper lobe, which was identified during a computed tomography (CT) scan of the chest, suggestive of early-stage lung cancer. Concurrently, multiple calcifications in the cartilaginous rings of the trachea were noted. Bronchoscopy revealed distinctive "pebblestone" nodules along the anterior and lateral tracheal walls, indicative of extensive TPO. The patient underwent bronchofiberscopy, which showed patency in the bronchial lumen of the right lung's upper lobe. A biopsy was not undertaken during this procedure. Comprehensive preoperative tests, including a blood biochemical examination, tumor-marker tests, lung-function tests, head-enhanced magnetic resonance imaging, abdominal ultrasound, and whole-body bone emission CT revealed no significant abnormalities. Despite this, the patient declined a whole-body positron emission tomography (PET)-CT scan. Given the potential malignancy of nodules in the right lung's upper lobe, the lobectomy for lung cancer was carried out, a procedure that would have proceeded irrespective of the presence or absence of TPO. Preoperative planning for potential tracheal intubation difficulties involved consultation with the anesthesiologist, resulting in a smooth intraoperative process. The pathology confirmed invasive adenocarcinoma. Post-surgery, the patient developed an infection in the right lung's lower lobe, identified as pseudomonas aeruginosa and Klebsiella pneumoniae through sputum culture and bronchoscopic lavage. Treatment with meropenem for 2 weeks, as guided by drug sensitivity results and respiratory advice, led to an improvement, allowing for discharge. A follow-up lung CT four months post-operation showed inflammation absorption in the right lower lobe. Conclusions: Surgical resection in cases of TPO association with lung cancer may have an increased risk of postoperative pulmonary infection. Proactive intraoperative sputum aspiration by anesthesiologists and the postoperative reinforcement of anti-infection measures, guided by drug sensitivity results, are recommended.

Tracheopathia osteochondroplastica: a case report illustrating the importance of imaging in this rare disorder.

Background: Tracheopathia osteochondroplastica (TPO) is an uncommon and benign abnormality of the cartilaginous tracheal wall. It is characterized by multiple calcified nodules in the submucosa of the trachea and main bronchi. This disorder can present as a wide range of nonspecific symptoms; therefore, TPO is usually found as an incidental finding during bronchoscopy. However, it does pose a risk for complicated intubations and recurrent respiratory infection. The etiology of the disease is unknown, and treatments are often only for symptomatic relief, if necessary. Case Description: In this report, we present a case of a 71-year-old woman who presents with chest pain and dyspnea on exertion. She has a history of takotsubo cardiomyopathy, and the symptoms have been persistent since the diagnosis over 6 months ago. A chest computed tomography (CT) demonstrated possible endobronchial lesions and minimal fibrosis. A bronchoscopy was completed following this observation, and it revealed nodular lesions on the cartilaginous rings of the trachea. She was diagnosed with TPO. The patient also has a history of gastroesophageal reflux disease (GERD) and reports relief from chest pain and dyspnea post-esophageal dilation for dysphagia. Conclusions: This case illustrates the relationship between esophageal, cardiac, and pulmonary symptoms, and highlights the role of imaging in the diagnosis of TPO. It also demonstrates a possible treatment option for symptomatic TPO that hasn't previously been discussed.

Building and validating an artificial intelligence model to identify tracheobronchopathia osteochondroplastica by using bronchoscopic images.

BACKGROUND: Given the rarity of tracheobronchopathia osteochondroplastica (TO), many young doctors in primary hospitals are unable to identify TO based on bronchoscopy findings. OBJECTIVES: To build an artificial intelligence (AI) model for differentiating TO from other multinodular airway diseases by using bronchoscopic images. DESIGN: We designed the study by comparing the imaging data of patients undergoing bronchoscopy from January 2010 to October 2022 by using EfficientNet. Bronchoscopic images of 21 patients with TO at Anhui Chest Hospital from October 2019 to October 2022 were collected for external validation. METHODS: Bronchoscopic images of patients with multinodular airway lesions (including TO, amyloidosis, tumors, and inflammation) and without airway lesions in the First Affiliated Hospital of Guangzhou Medical University were collected. The images were randomized (4:1) into training and validation groups based on different diseases and utilized for deep learning by convolutional neural networks (CNNs). RESULTS: We enrolled 201 patients with multinodular airway disease (38, 15, 75, and 73 patients with TO, amyloidosis, tumors, and inflammation, respectively) and 213 without any airway lesions. To find multinodular lesion images for deep learning, we utilized 2183 bronchoscopic images of multinodular lesions (including TO, amyloidosis, tumor, and inflammation) and compared them with images without any airway lesions (1733). The accuracy of multinodular lesion identification was 98.9%. Further, the accuracy of TO detection based on the bronchoscopic images of multinodular lesions was 89.2%. Regarding external validation (using images from 21 patients with TO), all patients could be diagnosed with TO; the accuracy was 89.8%. CONCLUSION: We built an AI model that could differentiate TO from other multinodular airway diseases (mainly amyloidosis, tumors, and inflammation) by using bronchoscopic images. The model could help young physicians identify this rare airway disease.

Tracheopathia osteochondroplastica-A benign disorder with a daunting appearance.

Tracheopathia osteochondroplastica (TO) is a rare and benign condition. It typically manifests as multiple osteocartilaginous nodules in the submucosa of the central airway. TO-related clinical symptoms and physical signs are nonspecific. The bronchoscopic examination is helpful in establishing the diagnosis. Treatment for TO is mostly conservative and symptom-oriented. The prognosis of TO is generally good, although cases of associated airway stenosis have been reported. In this case report, we describe the clinical, imaging, and histological features, and videoed bronchoscopic findings, of a middle-aged male patient with incidentally diagnosed TO.

Bronchoscopic Intervention for Tracheobronchopathia Osteochondroplastica: A Report of Two Cases.

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.

Clinicopathological Features and Management Principles of Tracheobronchopathia Osteochondroplastica - A Scoping Review.

Tracheobronchopathia osteochondroplastica (TO) is an orphan disease of the tracheobronchial tree without any known etiological attributes. There are several case reports published on this condition, yet the available information about the TO is discrete and of little clinical value. This scoping review is the first large-scale review on TO that collates individual patient data from the published case reports and descriptively analyses the clinicopathological features of this unique condition along with its management approaches and therapeutic outcomes. The objective was to synthesize comprehensive literature review on TO that can aid clinical practice and further research. An electronic search conducted in five large databases, including PubMed, EMBASE, CINAHL, CENTRAL, and Web of Science, for the published articles of TO yielded 1072 items. After screening, the individual patient data of 371 TO cases from 228 eligible articles were included and analysed in this scoping review. Supplementary Information: The online version contains supplementary material available at 10.1007/s12070-023-03998-6.

A case of tracheobronchopathia ostochondorplastica.

Tracheobronchopathia osteochondroplastica (TO) is a disorder caused by the accumulation of calcium phosphate in the submucosa of large airways. Benign proliferation of bone and cartilage lead to the narrowing of airways. Bronchoscopy is the diagnostic test for TO. It shows characteristic smooth nodules emerging from tracheal rings that never involves the posterior membranous wall.

A rare case of rapidly progressive Tracheobronchopathia Osteochondroplastica.

Tracheobronchopathia Osteochondroplastica (TO) is a rare benign disorder that is seldom progressive. Here, we report a case diagnosed with TO in our hospital. Bronchoscopy revealed multiple cartilaginous and ossifying nodules that are diagnostic for TO. Nodules protruding into the airways were observed as widespread and extended by the repeat bronchoscopy after 2 months of the diagnosis. TO was confirmed with the histopathology of the biopsies from nodules. Then he was referred to an interventional pulmonologist for laser ablation.

Tracheobronchopathia osteochondroplastica: clinical, bronchoscopic, and comorbid features in a case series.

BACKGROUND: Tracheobronchopathia osteochondroplastica (TO) is a rare condition of unknown etiology. TO is characterized by submucosal nodules, with or without calcifications, protruding in the anterolateral walls of the trachea and proximal bronchi. The objective of this study was to describe TO features and associated comorbidities in a series of patients. METHODS: Patients suffering from TO were retrospectively included by investigators from the Groupe d'Endoscopie Thoracique et Interventionnelle Francophone (GETIF). Demographic, clinical, comorbidities, bronchoscopic, functional, and radiological characteristics, and outcomes were recorded and analyzed. RESULTS: Thirty-six patients were included (69% male with a mean of 65 ± 12 years). Chronic symptoms were described by 81% of patients including cough (74%) and dyspnea on exertion (74%). TO was associated with COPD in 19% of the cases and gastroesophageal reflux disease in 6%. A mild to severe airflow obstruction was present in 55% of the cases. CT scan showed tracheal submucosal nodules in 93% of patients and tracheal stenosis in 17%. Bronchoscopy identified TO lesions in the trachea in 65% of the cases, and 66% of them were scattered. A bronchoscopic reevaluation was performed in 7 cases, 9 ± 14 months [1-56] after initial diagnosis, and showed the stability of lesions in all cases. Three patients underwent interventional bronchoscopic treatment. CONCLUSION: The diagnosis of TO relies on typical bronchoscopic findings and can be evoked on a CT scan. Histologic diagnosis can be useful in atypical cases for differential diagnosis. Given its low consequences in terms of symptoms, lung functions, and evolution, no treatment is usually required.

Tracheobronchopathia Osteochondroplastica: A Rare, Underrecognized Entity.

Tracheobronchopathia Osteochondroplastica (TO) is an extremely rare condition characterized by the presence of nodules made of bone and cartilage within the submucosa of the tracheobronchial wall. These protuberant nodules inside the trachea and bronchi can lead to airway obstruction, resulting in patients who experience recurrent respiratory systems and infections. The exact etiology is unknown. The mean age of diagnosis is in the 5th - 6th decades of life. TO is often confused with other diagnoses, especially asthma. We report a 41-year-old female who presented with intermittent exertional dyspnea for 10 years. Workups, including pulmonary function test, CT chest, and most importantly, flexible bronchoscopy, aided in the appropriate diagnosis. The unique feature observed during bronchoscopy is the sparing of the posterior wall of the trachea and bronchi.

Tracheobronchopathia osteochondroplastica: A rare case of misdiagnosis and difficult intubation.

Tracheobronchopathia osteochondro-plastica (TPO) is a rare disorder of yet unknown etiology. The common clinical features include cough with expectoration, breathing difficulty, hemoptysis, and recurrent airway infections, which can lead to a misdiagnosis as another chronic respiratory illness in a large number of cases due to the rare nature of TPO. Here we present a 25-year-old lady who was misdiagnosed as bronchial asthma for many years, and was found to have TPO after difficulty in intubation for administering general anesthesia prior to a surgical procedure.

A rare case of hemoptysis.

A 39-year-old nonsmoker male presented with complaints of cough with recurrent mild hemoptysis. Computed tomography of thorax showed nodular lesions on the right lateral wall of trachea. Fiber-optic bronchoscopy revealed multiple nodular lesions on the right lateral wall of lower one-third of trachea. Histopathological examination of biopsy specimen from the nodules was suggestive of tracheobronchopathia osteochondroplastica which is an uncommon airway disorder.

The Rocks Valley: Tracheopathia Osteochondroplastica: A Case Report.

Tracheopathia Osteochondroplastica is a rare benign tracheobronchial condition. Its unspecific symptoms and high clinical threshold for diagnosis make the condition an incidental diagnosis. We report an incidental tracheopathia osteochondplastica in 66 years old male patient with suspected thyroid malignancy.

Multi-Nodule of Large Airway: Tracheobronchopathia Osteochondroplastica, Two Cases Report and Literature Review.

We report two subjects with tracheobronchopathia osteochondroplastica (TO), including the clinical manifestations, histological findings, and clinical treatments, which were analyzed retrospectively. One patient with TO was a 60-year-old woman, and the other was a 47-year-old man. The main clinical manifestations were cough, chest pain, and dyspnea. Computed tomography (CT) images showed that TO mainly occurred in the trachea and main bronchus. Histological analysis showed inflammatory exudation, squamous metaplasia, submucosal cartilaginous, and ossification. We present the two cases to increase physician and patient awareness of this benign disease and to improve their understanding of the disease manifestations and potential complications.

Tracheobronchopathia osteochondroplastica-stalactite of airways.

A case of tracheobronchopathia osteochondroplastica (TO) was diagnosed in a 53-year-old man, who presented with prolonged cough and recurrent respiratory tract infection. Bronchoscopy revealed exostosis over the anterolateral wall of trachea and main bronchi sparing the posterior membranous wall. The endobronchial biopsy subsequently revealed ossification of the cartilage. To date, the aetiology of this condition remains unknown, and treatment is mainly symptomatic, emphasizing on timely management of recurrent respiratory infections. Bronchoscopy or surgical intervention is usually reserved for symptomatic patients with severe airway narrowing and airflow obstruction.

Pebbled Trachea: A Case Series and Literature Review of Tracheobronchopathia Osteochondroplastica.

We report a series of three cases diagnosed with tracheobronchopathia osteochondroplastica on bronchoscopy and computed tomography (CT) chest. Most patients were diagnosed incidentally on evaluation for chronic cough. The association of this entity with chronic bacterial infections and tuberculosis is an intriguing entity that was observed in our patients. Nodular, ulcerative, and calcific lesions in the trachea are bronchoscopic findings seen in quite a few other conditions posing diagnostic challenges. However, the classical bronchoscopic appearance with CT imaging in an appropriate clinical context can lead to an accurate diagnosis of this condition. We describe this array of cases with varying clinical presentations, their associations, and deliberate the literature reviews on this rare entity.

Surgical Management in Tracheobronchopathia Osteochondroplastica: A Case Study.

Tracheobronchopathia Osteochondroplastica is a benign condition characterized by osseous and cartilaginous submucosal growths of the tracheobronchial tree. This is a case report of an individual that was to undergo elective surgery using general anesthesia with endotracheal tube intubation. However, the anesthesiologist encountered a large osseous mass of the precricoid region and could not be intubated. This case report describes the technique for removal of the obstructing lesion using a Sonopet ultrasonic aspirator. Laryngoscope, 131:E911-E913, 2021.

Multi-nodule of large airway: tracheobronchopathia osteochondroplastica.

BACKGROUND: Tracheobronchopathia osteochondroplastica (TO) is a benign rare disease characterized by multiple tracheobronchial nodules, which has not been clearly illuminated and there are no standard guidelines for TO management. The aim of this study was to clarify the clinical features and treatment outcomes of TO and provide basis for clinical diagnosis and treatment. METHODS: A total of 32,215 patients taken bronchoscopy were retrospectively reviewed to select patients diagnosed with TO. Clinical characteristics, bronchoscopic manifestations, CT images, histopathological features, treatments and clinical course were investigated. RESULTS: 13 male and 6 female were identified as TO. The average age was 47.42±12.12 years. The incidence rate of TO in our study by bronchoscopy was 0.06%. The most frequent manifestation was chronic cough. Based on bronchoscopic manifestations, patients were categorized as three groups: the largest proportion was group of Stage II (10/19), followed by Stage III (5/19) and Stage I (4/19). The positive rate of CT in stage I, stage II and stage III was 0%, 50%, 100%, respectively. Histopathological analysis presented inflammatory exudation, squamous metaplasia, submucosal cartilaginous and osseous deposition. Bronchoscopic intervention and inhaled corticosteroids were administered to subjects in stage II and III, which improved their life qualities to some degree. CONCLUSIONS: Not only multi-disciplinary cooperation of clinical, endoscopic and histological assessment, but also awareness are crucial to TO diagnosis, especially in early stage of TO, which was difficult to identify. The stage of TO based on bronchoscopic visualization might be applied to guide the choice of clinical treatment strategy.