A case of tracheobronchial amyloidosis with emphasis on differential diagnosis.

Tracheobronchial amyloidosis is a manifestation of amyloidosis of the respiratory tract characterized by focal or diffuse deposition of amyloid in the submucosa of the trachea and proximal bronchi. Tracheobronchial amyloidosis is not associated with systemic amyloidosis or pulmonary parenchymal involvement. It affects predominantly men aged over fifty. Depending on the part of the tracheobronchial tree that is affected, stenosis of the airways causes a variety of unspecific symptoms. Diagnosis is reached by means of typical presentation in CT scan followed by bronchoscopy and histopathological confirmation. Tracheobronchial amyloidosis should be borne in mind in the differential diagnosis of patients with chronic cough and/or dyspnea or recurrent respiratory infections.

The role of MRI in the diagnosis and management of tracheal diverticulum.

BACKGROUND: Multidetector CT is currently the best imaging method for detecting tracheal diverticulum (TD). Compared with CT, MRI is radiation-free and has higher resolution. However, the MRI characteristics of this disease have not been previously reported. The present retrospective study compared the MR and CT imaging features of TD, aiming to examine the role of MRI in TD diagnosis and management. METHODS: Imaging data were collected in 26 TD patients divided into two groups, including the uninfected and infected groups. The MR and CT imaging features (size/wall/channel) of uninfected patients were compared. The performances of MRI and CT in diagnosing and monitoring therapeutic efficacy in infected TD patients were comparatively assessed. RESULTS: The uninfected group comprised 25 cases with 25 lesions confirmed by CT, including 23 lesions (92%) detected by MRI, with an average diameter of 8.5 mm (range from 3 to 15 mm). Meanwhile, the average diameter was 7.8 mm as measured by CT (range from 2.8 mm to 14.7 mm). The lesion diameters of the two cases not detected by MRI were 2.3 mm and 2 mm. MRI detected walls of all the 23 lesions (23/23), while CT detected no wall (0/23). CT showed channels in 18 lesions (18/23) versus3 for MRI (3/23). The infected case presented with a paratracheal abscess; MRI clearly showed a relationship between the abscess and the trachea, while CT could not show the lesion source. MRI also sensitively showed the whole process of lesion absorption. CONCLUSIONS: MRI can be used as a supplementary method for TD diagnosis, providing information about the wall that cannot be obtained by CT. MRI is superior to CT in diagnosing infected TD cases presenting with a paratracheal abscess, and in monitoring therapeutic efficacy in these patients.

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.

Identification of the avian tracheal trematode Typhlocoelum cucumerinum (Trematoda: Cyclocoelidae) in a host-parasite-environment system: diagnosis, life cycle and molecular phylogeny.

Typhlocoelum cucumerinum is a tracheal parasite of birds widely distributed across the globe. Nevertheless, aspects of the biology of this cyclocoelid are still poorly understood. Herein, we report the finding of T. cucumerinum in definitive and intermediate hosts from an urban waterbody of Brazil. The parasite was initially detected during the necropsy of domestic Muscovy ducks (Cairina moschata) found dead in the locality. Coproparasitological tests in live animals revealed that 12/47 (25.53%) Muscovy ducks and 2/8 (25%) mallards (Anas platyrhynchos platyrhynchos) were infected with T. cucumerinum. Moreover, rediae and metacercariae morphologically similar to T. cucumerinum were found in 3/248 (1.33%) Biomphalaria straminea collected in the same waterbody frequented by the birds. The conspecificity between the adult and the larval stages was confirmed molecularly (100% similarity in Cox-1). Moreover, the phylogenetic position of T. cucumerinum was determined for the first time based on partial fragments of the 28S, Cox-1 and Nad-1 genes. The species grouped with other members of the subfamily Typhlocoelinae with sequences available, but the data obtained do not support the distinctiveness of the genera Typhlocoelum and Tracheophilus. Further studies involving a broader range of species can result in taxonomic rearrangements in Typhlocoelinae.

Tracheal Injury during Endoscopic Transoral Vestibular Approach Thyroidectomy: A Case Report.

This report describes the intraoperative course of endoscopic thyroidectomy by oral vestibular approach in a female patient. This operation is new, and its perioperative management is not yet mature. In this case, the surgery resulted in trachea injury that could not be detected easily. As a result, the patient suddenly developed acute dyspnea and circulatory disorder. This procedure requires caution in surgical execution and anesthesia management.

Complete tracheal rings and hypoplastic left heart variant: a rare and fatal association.

Congenital complete tracheal rings are usually associated with pulmonary slings. We report a rare association of congenital complete tracheal rings with hypoplastic left heart variant. A term infant with diagnosis of a mildly hypoplastic mitral valve, unicuspid aortic valve, and moderately hypoplastic aortic arch with severe coarctation underwent a hybrid procedure initially. Upon failing extubation attempts, complete tracheal rings were seen on direct laryngoscopy. The combination of the lesions resulted in a poor outcome. In patients with failure of extubation post-cardiac surgery, a diagnosis of complete tracheal rings should be included in the differential and a direct laryngoscopy should be considered.

Pulmonary pythiosis in a canine patient.

A Staffordshire terrier presented for evaluation of a chronic, nonproductive cough that was unresponsive to antibiotic therapy. A large mass identified in the pulmonary hilum was most consistent with tracheobronchial lymphadenopathy on radiographic and computed tomography (CT) images. Bronchoscopy confirmed a mass compressing the dorsal portion of the intrathoracic trachea. Bronchoscopic biopsies of the tracheal mass revealed necrosuppurative and eosinophilic inflammation with intralesional Pythium insidiousum hyphae. Pythiosis should be included as a differential diagnosis for tracheobronchial lymphadenopathy and bronchopneumopathy in dogs, especially when the patient is from or has visited a region endemic for Pythium insidiosum.

[Tracheobronchopathia osteochondroplastica: a clinicopathological analysis of 18 cases].

Objective: To investigate the clinical symptoms, imaging features, pathologic manifestations and diagnosis of tracheobronchopathia osteochondroplastica (TO). Methods: The clinical data, imaging and pathologic features and outcome of 18 TO patients diagnosed at the First Affiliated Hospital of Zhengzhou University from August 2011 to August 2018 were collected and analyzed. Results: The 18 TO patients included 10 males and 8 females; patients' age range was 31 to 64 years (mean 52 years). Six patients (6/18) were smokers. The main presenting clinical symptoms included cough in 15 cases, expectoration in eight cases (8/18), hemoptysis in five cases (5/18), chest tightness in four cases, wheezing in three cases and chest pain in two cases. The time interval between the initial symptoms and diagnosis was 1.5 to 360.0 months, and the average time interval was 45.2 months. Blood calcium and phosphorus were normal in 18 patients (18/18). Chest X-ray showed no direct evidence of TO. Six patients (6/18) showed irregular changes in the trachea or bronchial wall by chest CT scan. Three patients (3/18) had mild ventilatory obstruction. TO was classified as: 10 cases (10/18) were scattered type, seven cases (7/18) were diffuse type and one case (1/18) was confluent type. Epithelial squamous metaplasia, submucosal cartilage, submucosal ossification and hematopoietic bone marrow within the ossified areas were the characteristic histopathologic findings of TO. Conclusions: TO is a rare benign disorder that shows atypical presentation. CT scan is insensitive, the histopathology shows submucosal cartilage or ossification. TO should be diagnosed by comprehensive consideration of clinical symptoms, imaging and pathology.

Correlation of Symptoms with Bronchoscopic Findings in Children with a Prenatal Diagnosis of a Right Aortic Arch and Left Arterial Duct.

A right aortic arch (RAA) with a left arterial duct (LAD) together encircle the trachea and have the potential to cause tracheobronchial compression and published guidelines recommend bronchoscopy in symptomatic patients. The aim of the study was to describe the incidence of tracheal compression in a cohort of prenatally diagnosed RAA and LAD. Retrospective review of clinical course and imaging of prenatal cases of RAA and LAD assessed with flexible bronchoscopy over an 11-year period. 34 cases of prenatally diagnosed RAA with LAD underwent bronchoscopy at median age of 9 months (range 0.4-123) of whom 11 had respiratory symptoms and 23 were asymptomatic. In the neonatal period, three cases demonstrated respiratory symptoms. An aberrant left subclavian artery (ALSA) was identified in 29 cases. Pulsatile tracheal compression was identified in 32/34 (94%) cases and two cases showed normal tracheal appearances. Significant tracheal compression (> 70% occlusion) was present in 25/34 (74%) cases of which 16 were asymptomatic. Significant carinal compression (> 70% occlusion) was identified in 14/34 (42%) cases, an ALSA was observed in 13/14. Surgical relief of a vascular ring has been performed in 27 (79%) cases at a median age of 15 months (range 0.6-128 months). At surgery, a fibrous remnant of an atretic left aortic arch was identified in 11/27 (41%) cases. Significant tracheal compression may be present in infants even without symptoms. If early relief of airway compression is to be achieved to promote normal development of tracheal cartilage, early bronchoscopy should be considered.

Tracheal injury: late symptom after double-lumen tube intubation - A case report.

As a basic skill, endotracheal intubation, performed throughout the world by health care professionals is a relatively safe and effective maneuver. However, this technique is not risk free and could lead to many serious complications. We wanted to report that in a patient with double lumen tube intubation, airway trauma can cause late symptoms . We thought that such complications must be published to share experiences.

Tracheal Cartilaginous Sleeve in Syndromic Craniosynostosis: An Underrecognized Source of Significant Morbidity and Mortality.

Tracheal cartilaginous sleeve (TCS) is a rare and previously unrecognized source of morbidity and mortality in patients with certain craniosynostosis syndromes. There is a paucity of reporting on this airway anomaly, and the true incidence of TCS is largely unknown. The purpose of this study was to investigate the incidence of TCS among patients with syndromic craniosynostosis at our institution. Patients with syndromic craniosynostosis who underwent direct bronchoscopy and laryngoscopy were evaluated retrospectively by pediatric otolaryngologists for the presence of TCS and associated anomalies. Among patients with a diagnosis of syndromic craniosynostosis in our craniofacial database, 10 (37%) were found to have previous direct bronchoscopy and laryngoscopy reports. Of these 10 patients, 2 had Crouzon syndrome, 3 had Pfeiffer syndrome, 3 had Apert syndrome, 1 had Muenke syndrome, and 1 had Antley-Bixler syndrome. Eighty percent (8/10) of these patients were found to have some evidence of TCS. The most commonly observed associated findings included the following: tracheostomy dependency (7/10; 70%), hearing loss (6/10; 60%), obstructive sleep apnea (5/10; 50%), cervical spine anomalies (5/10; 50%), developmental delay (5/10; 50%), and enlarged cerebral ventricles (4/10; 40%). Larger multicenter studies are required to further characterize this airway anomaly and its impact on this patient population. Our results confirm the importance of thorough airway evaluation at initial presentation and the need for validated screening protocols.

Chronic Obstructive Pulmonary Disease Mismatch: A Case of Tracheal Hamartoma.

OBJECTIVE: To demonstrate the diagnostic challenge of tracheal hamartoma in a patient with chronic obstructive pulmonary disease (COPD). CLINICAL PRESENTATION AND INTERVENTION: A 65-year-old man with COPD was admitted with sudden onset of asphyxia attacks related to the position of his body. Computerized tomography (CT) of the neck showed a soft tissue mass with calcification, which occluded more than two-thirds of the proximal part of the trachea. The tumor was completely removed, and histopathology confirmed hamartoma. CONCLUSION: This case report showed the detection of a primary tracheal tumor on CT. This finding enabled the correct diagnosis and led to appropriate treatment in the form of surgery.

[IgG(4)-related disease involving the trachea and paratracheal soft tissue: a case report and literature review].

Objective: To investigate the clinical data of a patient with IgG(4)-related disease involving the trachea and paratracheal soft tissue and review the literature so as to improve the understanding level of the disorder. Methods: To analyze the clinical manifestation, laboratory examination, imaging, histopathology, treatment and prognosis of a patient with IgG(4)-related disease trachea and paratracheal soft tissue involved, who was admitted to the Department of Respiratory and Critical Care Medicine at Beijing Chaoyang Hospital. The relevant literatures were reviewed. Results: A 18-year-old female was admitted with chief complaint of cough, dyspnea, and neck mass. Neck CT suggested that tracheal stenosis was caused by surrounded soft tissue. Paratracheal mass biopsy showed dense collagen fibers with infiltration of many lymphocytes and plasma cells. Immunohistochemical stain found that IgG(4)-positive plasma cells were >50/high power field (HPF) and a ratio of IgG(4)/IgG positive cells was over 40% .The level of serum IgG(4) was significantly increased (2 930 mg/L). She was diagnosed as IgG(4)-related disease. The patient was treated with 80 mg intravenous methylprednisolone per day for three days, then prednisone 40 mg daily oral. Her dyspnea was significantly relieved.One month later, CT scan showed that the cervical tracheal stenosis was significantly improved. We identified 20 cases of IgG(4)-related disease involving the trachea and paratracheal soft tissue from databases, in which only 1 case was similar as this patient. The other 19 cases were of extratracheal involvement. Elevated serum IgG(4) was detected in 11/12 patients. Most patients were treated with glucocorticoid, some combined with immunosuppressive agents and rituximab. The clinical outcome was good. Conclusion: IgG(4)-related disease involving the trachea and paratracheal soft tissue is a rare condition. Serum IgG(4) level and histopathology should be considered for diagnosis. Glucocorticoid is effective.

[Tracheal Surgery]. / Chirurgie der Trachea.

Surgery of the trachea is a specialised field in which many disciplines work jointly due to the variety of indications and the extended topography. Not only because of its particular functional importance, but also because of its complex morphology, anatomy and physiology, this organ represents a special therapeutic challenge. A variety of diseases require surgical procedures of the trachea; the therapeutic strategy is influenced both by the disease itself as well as patient-dependent parameters. Regardless of the nature of the underlying disorder, good results require a high level of expertise in airway management, a careful diagnosis and interventional planning as well as an experienced surgical team that masters extended operative techniques. An optimal treatment decision always requires a multidisciplinary assessment of the patient's individual situation by interventional pulmonologists, thoracic surgeons, visceral surgeons, ENT (ear, nose and throat) surgeons and anaesthesiologists.

Tracheobroncopathia osteochondroplastica: Three case reports with literature review.

Tracheobroncopathia osteochondroplastica (TO) is a benign disease of the large airways seen very rarely. It is characterized by 1-3 mm sized ossified nodular lesions in submucosa. Its etiology is unclear, but it is stated that malignancy, chronic inflammation, amyloidosis, and genetic factors might have an effect on it. It was first described by Wilks in a 38-year-old man diagnosed with tuberculosis in 1857. Generally, patients are asymptomatic and TO is diagnosed incidentally. But symptoms become significant with infections and obstruction in tracheabronchial tree. Generally chest radiography is normal, so thorax computed tomography can be remarkable in diagnosis of TO. Besides, final diagnosis can be established by viewing ossified nodules in trachea and bronchus through the fiberoptic bronchoscopy. Amyloidosis, tuberculosis, sarcoidosis, bronchial carcinoma, and tracheobronchial calcinosis must be remembered in differential diagnosis. Also ossifications in submucosa and proof of bone marrow in histopathological examinations are important in diagnosis of TO. Mostly palliative treatment is performed to the symptoms . We want the clinicians to keep in mind for this very rarely seen tracheal disease with three case reports.

Airway stenting in the management of iatrogenic tracheal injuries: 10-Year experience.

BACKGROUND AND OBJECTIVE: Iatrogenic tracheal injury (ITI) is a rare yet severe complication of endotracheal tube (ETT) placement or tracheostomy. ITI is suspected in patients with clinical and/or radiographic signs or inefficient mechanical ventilation (MV) following these procedures. Bronchoscopy is used to establish a definitive diagnosis. METHODS: We conducted a retrospective, single-centre chart review of 35 patients between 2004 and 2014. Depending on the nature and location of ITI and need for MV, patients were triaged to surgical repair, endoscopic management with airway stents or conservative treatment consisting of ETT or tracheotomy cannula (TC) placement distal to the wound and bronchoscopic surveillance. RESULTS: Three of the four patients (11.43%) presenting with tracheoesophageal fistula (TEF) underwent surgery. Seven patients (20%) who did not require MV underwent endoscopic surveillance. Of the 24 ventilated patients (68.57%), 7 with ITI in the lower trachea were treated with silicone Y-stent (ETT or TC was placed inside the stent) and 17 patients with ITI in the upper trachea were managed by placing ETT or TC cuff distal to the injury. Overall management success, defined as complete healing of the ITI, was seen in 88.57% of patients. Four patients (11.43%) died of non-ITI-related comorbidities. CONCLUSION: Conservative management should be considered in non-ventilated patients with ITI and when ITI is located in the upper trachea of ventilated patients where ETT or TC bypasses the injury. Airway stenting should be considered in ventilated patients with ITI located in the lower trachea. Surgery should be reserved for TEF and conservative and endoscopic management failure.

Tracheoinnominate Fistula: Endovascular Treatment with a Stent Graft in a 4-Year-Old Child.

A 4-year-old boy presented with acute and profuse bleeding at the tracheostomy site. An emergency angiography was performed and identified a pseudoaneurysm at the innominate artery. A selective catheterization of the artery was executed and 2 Advanta V12 balloon-expandable covered stents were implanted in an overlapping manner to occlude the pseudoaneurysm. Final angiography demonstrated patency of the innominate artery and no signs of bleeding. The patient had no postoperative complications and no further bleeding during follow-up. A contrasted computed tomography scan was performed after 20 days and demonstrated no signs of pseudoaneurysm or bleeding. After 4 months, the patient was readmitted to tracheal dilatation and change of T-tube and died of respiratory complications.