Tracheobronchopathia Osteochondroplastica - to Biopsy or not to Biopsy? A Relook at The Rare Disease.

BACKGROUND: Tracheobronchopathia osteochondroplastica (TPO) is a rare idiopathic disease involving the tracheobronchial tree. It is mostly an incidental finding with non-specific clinical manifestations. It has typical bronchoscopic, radiological features and biopsy is usually considered non-essential. The study aimed to determine whether biopsy makes a difference in the management of patients. METHODS: All patients diagnosed with TPO in our institution over 15 years (2005 to 2020) were included in this study. Their medical records, chest computed tomography (CT), and bronchoscopy reports were retrospectively reviewed, and data were analysed. All the CT images were reviewed by a senior chest radiologist. RESULTS: From the 20,000 bronchoscopies and 260,000 CT thorax images obtained, 28 cases were diagnosed as TPO based on either bronchoscopy or radiology or both. Among the 19 cases diagnosed through bronchoscopy, 16 underwent a biopsy. In addition to TPO features, biopsy showed additional diagnoses in 6 cases. In 9 cases, TPO was not initially diagnosed by CT but by bronchoscopy. In 8 patients, TPO was diagnosed incidentally on CT performed for other reasons. On follow-up with the treatment of underlying/co-existing concomitant aetiologies, clinical improvement was noted in all patients. None of them progressed to respiratory failure or airway obstruction until the last follow-up. CONCLUSION: Among patients who underwent bronchoscopic biopsy of TPO lesions, 38% had biopsy results showing an alternative aetiology, which led to changes in the treatment plan for all these patients. Hence, a bronchoscopic biopsy of TPO lesions should be performed to rule out other aetiologies.

Severe tracheobronchopathia osteochondroplastica in an asymptomatic patient.

Tracheobronchopathia osteochondroplastica (TO) is a rare condition affecting the lumen of the tracheobronchial tree. It is characterised by the presence of multiple osseous and cartilaginous nodules with posterior wall sparing. While it is a benign condition, it can cause narrowing of the tracheal lumen and subglottis to varying degrees. Approximately 400 cases have been reported worldwide, with an incidence of 0.3% in autopsies and between 1 in 125 and 1 in 5000 on bronchoscopy. As most patients are asymptomatic, this may contribute to underdiagnoses and relative low incidence. Severity of the condition is often unrelated to patient symptomatology. We present a patient with one of the most severe cases of TO seen at our institution. Despite being asymptomatic, significant tracheal and bronchial narrowing was found incidentally on laryngobronchoscopy.

Incidental Tracheocele as an Unusual Presentation of Pneumomediastinum in the Trauma Setting.

BACKGROUND Tracheocele are rarely encountered air cysts formed due to tracheal wall outpouching through a weak vantage point. The majority are acquired in the adult population and are associated with conditions that weaken the tracheal wall. Most tracheoceles are diagnosed incidentally since many are asymptomatic or present with nonspecific symptoms. Multidetector computed tomography (MDCT) scans are the most common imaging modality for diagnosis of silent tracheal cysts. Tracheocele have been very rarely documented in the trauma setting; therefore, in the setting of multiple body trauma diagnosis can be challenging. CASE REPORT We report a case of an acquired tracheocele after a high-impact blunt thoracic trauma with an admission diagnosis of suspected tracheal perforation. MDCT of the neck and chest demonstrated an irregularly multicystic-shaped air collection at the right posterolateral trachea upon evaluation. Flexible laryngoscopy and bronchoscopy results were unremarkable. CONCLUSIONS Tracheocele are rare and asymptomatic pseudo-diverticulum of the tracheal wall. Many are diagnosed incidentally with imaging studies for other conditions. Nevertheless, in the trauma setting its diagnosis can be challenging and misleading. Consideration of conditions such as tracheocele is important to prevent any unwarranted treatment modalities.

Multiple Tracheal Diverticula in a COVID-19 Positive Patient.

A tracheal diverticulum is a type of paratracheal air cyst and is usually an incidental finding after a computed tomography scan of the neck and thorax. With an incidence between 1% and 4% in adults, tracheal diverticula are rare entities that can be symptomatic in certain cases. We present a case of a COVID-19 positive patient who presented to our hospital and was diagnosed with multiple tracheal diverticula during his hospitalization.

Tracheal bronchus: a rare cause of recurrent pneumonia in adults.

Tracheal bronchus, also known as bronchus suis, is a rare congenital anomaly of the airway where an accessory bronchus originates directly from the trachea. With an estimated incidence of 0.001%-2.0%, this condition is rarely reported in literature. It is usually discovered as an incidental finding in an otherwise asymptomatic individual. However, it can act as a focus of recurrent infection or present as persistent radiographic infiltrates. Multidetector CT imaging and bronchoscopy play a crucial role in the identification of this entity. We hereby report the case of a middle-aged man who presented with recurrent right upper lobe pneumonia, which was found to be due to an underlying tracheal bronchus.

Respiratory distress and tracheal deviation secondary to sigmoid volvulus / Dificultad respiratoria y desviación traqueal secundaria a vólvulo sigmoideo ­ Un reporte de caso

INTRODUCTION: The trachea is a semiflexible tube of 1.5 to 2 cm in width and 10 to 13 cm in length. Its deviation might be caused by not only diverse thoracic but also abdominal pathologies, which may compromise the airway. We present a case of a severe tracheal deviation due to an abdominal pathology causing displacement of mediastinal structures. CLINICAL CASE: A 78-year-old woman presents with difficulty breathing. History of chronic bedridden and frequently constipated, last stool 5 days prior. On physical examination, cachectic complexion, dry mucous membranes, breathing superficially with scarce wheezing, SatO2 82% on room air. Abdomen distended with an absence of bowel sounds. Chest x-rays show severe tracheal deviation and abdominal x-ray with coffee bean sign. A laparotomy evidences a large sigmoid volvulus. A sigmoidectomy and descending colon colostomy is performed. Room air oxygen saturation improved after extubation to 96%.CONCLUSION: Desaturation and tracheal deviation were caused by a large sigmoid volvulus. Although these pathologies were thoracic, clinicians should suspect different underlying pathologies, in this case, abdominal

INTRODUCCIÓN: La tráquea es un tubo semiflexible de 1-5 a 2 cm de ancho y 10 a 13 cm de longitud. Puede presentar desviaciones en su trayecto, no solo por patologías torácicas, sino también abdominales, las cuales pueden comprometer la vía aérea. Presentamos el caso de una desviación severa de la tráquea por una patología abdominal que ocasionó desplazamiento de las estructuras mediastinales. REPORTE DE CASO: Mujer de 78 años que se presenta por dificultad respiratoria. Antecedente de postramiento crónico en cama y estreñimiento frecuente, con última deposición 5 días previos. En la exploración física presenta complexión caquéctica, mucosas secas, respiración superficial con sibilancias, saturando 82% al aire ambiente. Abdomen distendido con ausencia de ruidos intestinales. Radiografía torácica muestra desviación traqueal severa y la radiografía abdominal muestra signo del grano de café. En el abordaje por laparotomía se evidencia un vólvulo sigmoideo grande. Se realizó sigmoidectomía y colostomía del colon descendiente. La saturación al aire ambiente mejoró después de la extubación a 96%. CONCLUSIÓN: La desaturación y desviación traqueal fueron causadas por un vólvulo sigmoideo grande. Aunque estas patologías eran torácicas, el clínico debe sospechar diferentes patologías de base, como en este caso, abdominales.

An unusual cause for globus sensation: infected tracheal diverticulum with abscess formation.

CASE REPORT: A 43-year-old woman presented with a 3-week history of globus sensation and malaise. A computed tomography scan of her neck showed a large right paratracheal abscess secondary to an infected tracheal diverticulum. The patient was admitted under the ENT surgical team, and underwent incision and drainage of the abscess. There were no post-operative complications and she was discharged home after 2 days, on oral antibiotics. CONCLUSION: This case demonstrates that a tracheal diverticulum may become infected and present as a cervical abscess. To our knowledge, this is the fourth reported case in the international literature of abscess formation related to an infected tracheal diverticulum.

CT findings of non-neoplastic central airways diseases.

Non-neoplastic lesions of central airways are uncommon entities with different etiologies, with either focal or diffuse involvement of the tracheobronchial tree. Clinical symptoms of non-neoplastic tracheobronchial diseases are non-specific, and diagnosis is difficult, especially in the early stages. Three-dimensional computed tomography (3D-CT) is an evaluable tool as it allows to assess and characterize tracheobronchial wall lesions and meanwhile it enables the evaluation of airways surrounding structures. Multiplanar reconstructions (MPR), minimum intensity projections (MinIP), and 3D Volume Rendering (VR) (in particular, virtual bronchoscopy) also provide information on the site and of the length of airway alterations. This review will be discussed about (1) primary airway disorders, such as relapsing polychondritis, tracheobronchophathia osteochondroplastica, and tracheobronchomegaly, (2) airway diseases, related to granulomatosis with polyangiitis, Chron's disease, Behcet's disease, sarcoidosis, amyloidosis, infections, intubation and transplantation, (3) tracheobronchial malacia, and (4) acute tracheobronchial injury. 3D-CT findings, especially with MPR and 3D VR reconstructions, allows us to evaluate tracheobronchial disease morphologically in detail.

Tracheal rupture after vocal cord polyp resection: A case report.

INTRODUCTION: Transoral laser microsurgery (TLM) is one of the most common operations performed for glottic lesions. Several protection measures are taken to prevent tracheal damage. However, some protection measures and common postoperative complications may still cause delayed tracheal rupture in certain situations. Cases of tracheal rupture after surgery are extremely rare, and there are no previous reports of TLM of the glottis causing tracheal rupture. PATIENT CONCERNS: A middle-aged woman who underwent TLM for bilateral vocal cord polyps developed sudden neck pain, followed by cough and subcutaneous emphysema. DIAGNOSIS: She underwent head, neck, and chest computed tomography (CT), which revealed a 4-cm membranous tracheal tear located 4.5 cm distal to the glottis, pneumomediastinum, and subcutaneous emphysema extending from the base of skull to the chest. INTERVENTIONS: The patient underwent an emergency surgical surgical chest exploration and tracheal repair. OUTCOMES: One month after the surgery, the patient fully recovered with no tracheal stenosis or respiratory dysfunction. CONCLUSIONS: Conventional protective measures and common postoperative complications of TLM may also cause tracheal rupture.

Tracheobronchopathia Osteochondroplastica Associated with Fibrotic Interstitial Lung Disease.

Tracheobronchopathia osteochondroplastica (TPO) is a very rare, benign disorder involving the lumen of the trachea-bronchial tree. However, its etiology is unknown. In our first case, observation for several years showed that TPO worsened as interstitial lung disease was aggravated. In the second case, the lung parenchymal lesion on computed tomography (CT) was found to be compatible with interstitial lung abnormality (ILA). We believe that our cases suggest a common pathogenetic relationship between TPO and fibrotic interstitial lung disease. TGF-ß is likely a common factor in the pathogenesis of TPO and fibrotic interstitial lung disease.

An imaging study on tracheomalacia in infants with esophageal atresia: the degree of tracheal compression by the brachiocephalic artery is a good indicator for therapeutic intervention.

PURPOSE: Tracheomalacia (TM) is a frequent complication after esophageal atresia (EA) repair. This study aimed to review patients who underwent aortopexy for TM after EA repair and to compare their imaging features. METHODS: The patients who underwent thoracoscopic EA repair and contrast-enhanced computed tomography (CECT) at our hospital between 2013 and 2020 were retrospectively reviewed. The ratio of the lateral and anterior-posterior diameter of the trachea (LAR) where the brachiocephalic artery (BCA) crosses the trachea was defined. The LAR of the patients who underwent CECT for asymptomatic pulmonary disease was set as a normal reference. The Z-score of each LAR was calculated and compared between the patients that did or did not undergo aortopexy. RESULTS: A total of 51 patients represented the controls, 5 patients underwent aortopexy, and 12 patients were discharged without surgery. The mean LARs in the patients who underwent aortopexy, did not undergo aortopexy, and controls were 3.54, 1.54, and 1.15, respectively. The mean Z-score of the aortopexy group was 21.2. After successful aortopexy, each patient's LAR decreased to < 1.5. CONCLUSION: Aortopexy was preferred if the trachea was compressed by the BCA. The LAR is a useful indicator for predicting the therapeutic effect of aortopexy.

Chronic cough with dual pathologies of pulmonary ossification.

An 81-year-old man presented with chronic cough, which did not respond to the initiation of combined bronchodilator/inhaled corticosteroid therapy. CT of the chest revealed calcified nodules throughout the trachea sparing the posterior membrane, and tiny peripheral parenchymal nodules with basal interlobular septal thickening and calcification. Flexible bronchoscopy demonstrated endobronchial nodularity from the proximal trachea to the mid-sections of both main bronchi, sparing the posterior membrane. Histopathology revealed submucosal fibrous connective tissue and benign bone, confirming a diagnosis of tracheobronchopathia osteochondroplastica. CT was consistent with a concurrent diagnosis of dendriform pulmonary ossification. These two rare phenomena often present with non-specific symptoms, and the diagnosis can be made with imaging in both conditions. There is a role for bronchoscopy in the diagnosis of tracheobronchopathia osteochondropastica, and the endobronchial appearance could be diagnostic. The concurrence of both phenomena in our case might represent activity of a common cellular pathway of ossification in both sites.