Osteochondroplastic tracheobronchopathy: Four case reports. / Traqueobroncopatía osteocondroplástica: reporte de cuatro casos clínicos.

Introduction: Osteochondroplastic tracheobronchopathy is a rare benign chronic disease of unknown etiology. Bronchoscopy remains the gold standard for diagnosing osteochondroplastic tracheobronchopathy. Its typical findings are described as a cobblestone, rock garden, mountainscape, or stalactite cave appearance. The present work aims to show the main clinical features of this rare pathology. Clinical cases: The clinical data of four middle-aged patients, three men and one woman, were analyzed. The main clinical symptoms were chronic cough, dyspnea, and dysphonia. The patient's preliminary diagnosis was made by computed axial tomography of the chest, confirmed by bronchoscopy and histopathological examination. Treatment included medication for symptoms and, in one case, cryosurgery and argon plasma coagulation. Discussion: Diagnosing osteochondroplastic tracheobronchopathy was not easy, given its uncommon nature and non-specific symptoms often found in other pathologies. No case series articles on this pathology have been published in Peru. Therefore, we used the original articles published in other countries to reference our findings. Conclusion: Osteochondroplastic tracheopathy is a benign disease that typically affects adults. Men are more likely to be affected. Its clinical manifestations are non-specific and frequently of pharyngeal origin, and the cause is not yet defined. Chest computed axial tomography combined with bronchoscopy are the main diagnostic procedures. There is no standard treatment with consistent therapeutic effects.

Introducción: La traqueobroncopatía osteocondroplástica es una rara enfermedad crónica benigna de etiología desconocida. La broncoscopía sigue siendo el estándar de oro para el reconocimiento de traqueopatía osteocondroplástica. Sus hallazgos típicos se describen como un empedrado, un jardín de rocas, una apariencia de paisaje montañoso o de una cueva con estalactitas. El objetivo del presente trabajo es mostrar las principales características clínicas de una patología poco conocida. Casos clínicos: Se analizaron los datos clínicos de cuatro pacientes de mediana edad, tres fueron hombres y una mujer. Los principales síntomas clínicos fueron tos crónica, disnea, disfonía. Los pacientes tuvieron un diagnóstico preliminar mediante tomografía axial computarizada de tórax, confirmado por examen video broncoscópico e histopatológico. El tratamiento incluyó medicamentos para los síntomas y en un solo caso criocirugía y coagulación con argón plasma. Discusión: El diagnóstico de traqueobroncopatía osteocondroplástica no fue sencillo por ser una entidad rara, cuyos síntomas son inespecíficos y muy frecuentes en otras patologías. En Perú no se han publicado artículos de serie de casos sobre esta patología. Por lo tanto, tomamos como referencia artículos originales publicados en otros países para compararlos con nuestros hallazgos. Conclusión: La traqueopatía osteocondroplástica es una enfermedad benigna que predispone a los adultos, los hombres tienen más probabilidades de verse afectados. Sus manifestaciones clínicas son inespecíficas; frecuentemente de origen faríngeo y la causa no está aún definida. La tomografía axial computarizada de tórax combinada con video broncoscopía son los principales procedimientos para el diagnóstico. No existe un estándar de tratamiento con efectos terapéuticos consistentes.

Traqueobroncopatía osteocondroplástica: reporte de cuatro casos clínicos / Osteochondroplastic tracheobronchopathy: four case reports

INTRODUCCIÓN: La traqueobroncopatía osteocondroplástica es una rara enfermedad crónica benigna de etiología desconocida. La broncoscopía sigue siendo el estándar de oro para el reconocimiento de traqueopatía osteocondroplástica. Sus hallazgos típicos se describen como un empedrado, un jardín de rocas, una apariencia de paisaje montañoso o de una cueva con estalactitas. El objetivo del presente trabajo es mostrar las principales características clínicas de una patología poco conocida. CASOS CLÍNICOS: Se analizaron los datos clínicos de cuatro pacientes de mediana edad, tres fueron hombres y una mujer. Los principales síntomas clínicos fueron tos crónica, disnea, disfonía. Los pacientes tuvieron un diagnóstico preliminar mediante tomografía axial computarizada de tórax, confirmado por examen video broncoscópico e histopatológico. El tratamiento incluyó medicamentos para los síntomas y en un solo caso criocirugía y coagulación con argón plasma. DISCUSIÓN: El diagnóstico de traqueobroncopatía osteocondroplástica no fue sencillo por ser una entidad rara, cuyos síntomas son inespecíficos y muy frecuentes en otras patologías. En Perú no se han publicado artículos de serie de casos sobre esta patología. Por lo tanto, tomamos como referencia artículos originales publicados en otros países para compararlos con nuestros hallazgos. CONCLUSIÓN: La traqueopatía osteocondroplástica es una enfermedad benigna que predispone a los adultos, los hombres tienen más probabilidades de verse afectados. Sus manifestaciones clínicas son inespecíficas; frecuentemente de origen faríngeo y la causa no está aún definida. La tomografía axial computarizada de tórax combinada con video broncoscopía son los principales procedimientos para el diagnóstico. No existe un estándar de tratamiento con efectos terapéuticos consistentes.

INTRODUCTION: Osteochondroplastic tracheobronchopathy is a rare benign chronic disease of unknown etiology. Bronchoscopy remains the gold standard for diagnosing osteochondroplastic tracheobronchopathy. Its typical findings are described as a cobblestone, rock garden, mountainscape, or stalactite cave appearance. The present work aims to show the main clinical features of this rare pathology. CLINICAL CASES: The clinical data of four middle-aged patients, three men and one woman, were analyzed. The main clinical symptoms were chronic cough, dyspnea, and dysphonia. The patient's preliminary diagnosis was made by computed axial tomography of the chest, confirmed by bronchoscopy and histopathological examination. Treatment included medication for symptoms and, in one case, cryosurgery and argon plasma coagulation. DISCUSSION: Diagnosing osteochondroplastic tracheobronchopathy was not easy, given its uncommon nature and non-specific symptoms often found in other pathologies. No case series articles on this pathology have been published in Peru. Therefore, we used the original articles published in other countries to reference our findings. CONCLUSION: Osteochondroplastic tracheopathy is a benign disease that typically affects adults. Men are more likely to be affected. Its clinical manifestations are non-specific and frequently of pharyngeal origin, and the cause is not yet defined. Chest computed axial tomography combined with bronchoscopy are the main diagnostic procedures. There is no standard treatment with consistent therapeutic effects.

Tracheomediastinal fistula induced by concurrent chemoradiotherapy in small cell lung cancer: A case report and literature review.

Tracheomediastinal fistula is a rare but life-threatening complication of cancer. We report a case of tracheomediastinal fistula induced by concurrent chemoradiotherapy in limited stage small cell lung cancer. Despite the treatment response, the metastatic paratracheal lymph node increased gradually during concurrent chemoradiotherapy, resulting in the occurrence of tracheomediastinal fistula and mediastinitis. Without any surgical intervention, the patient achieved successful recovery from mediastinitis through antibiotic treatment, although the tracheomediastinal fistula remained open. In this report, we also review previous studies of tracheomediastinal and bronchomediastinal fistulas and summarize the clinical features.

Long-term outcomes of tracheal stents removal under fluoroscopy guidance: comparison of tracheal fistulas and tracheal stenosis.

BACKGROUND: Endoscopic removal is the most common method for removal of tracheal stents. Few studies have reported the technique of fluoroscopy-guided stent removal for tracheal fistula and tracheal stenosis. We aimed to study the safety and efficacy of fluoroscopy-guided stent removal as well as the optimal duration for stent usage. METHODS: We conducted a retrospective analysis of 152 patients who underwent fluoroscopy-guided stent removal from January 2011 to June 2017. Reasons for stent implantation were tracheal fistula in 85 patients (TF group), and tracheal stenosis in 67 patients (TS group). All patients underwent tracheal CT scans before stent removal and during follow up. The technical success rate, complications, and survival rate were compared between the two groups. RESULTS: The technical success rate of stent removal was 98.9 and 97.4%, respectively for the TF and TS group. Removal was routine for half of patients, and in the remainder, excessive granulation tissue was the common indications for stent removal, which was found after stenting at 142.1 ± 25.9 days in the TF group, and at 89.9 ± 15.0 day in the TS group. The total incidence of complications was 21.1 and 22.4%, respectively, for the TF and TS groups. Perioperative death occurred in one patient in the TF group, and two patients in the TS group. Recurrence of fistula or stenosis requiring re-stenting was the most comment complication in both groups. The 0.5-, 3-, 6-year survival rates were 90.3, 59.6, and 36.1% for TF group, and 80.4, 75.7, 75.7% for TS group. CONCLUSIONS: Fluoroscopic removal of tracheal stents is safe and effective for both tracheal fistula and tracheal stenosis, with no significant difference in outcomes. Clinicians should pay attention to the risk of hemoptysis for patients with malignant tumors and a combination with endoscopic hemostasis may help improve its safety.

Bronchoscopic Treatment of Tracheobronchial Fistula With Autologous Platelet-Rich Plasma.

Tracheobronchial fistula (TBF) is a challenging management condition. Several bronchoscopic procedures have been tried for fistula closure. However, none has been found to be superior to the others. We herein describe a novel technique involving the submucosal injection of autologous platelet-rich plasma (auto-PRP) around the fistula to close the TBF. After auto-PRP treatment, all 3 TBF patients have successfully healed. No treatment-related complications and fistula-related symptoms were detected. Thus, this application of auto-PRP for fistula closure is a feasible and cost-effective strategy and could be recommended as a valuable therapeutic alternative for repairing postoperative TBF.

Clinical Characteristics of Six Cases of Tracheobronchopathia Osteochondroplastica.

Objective: To investigate the clinical characteristics of tracheobronchopathia osteochondroplastica (TO). Methods: The clinical data of six patients with TO from November 2016 to November 2018 were retrospectively analyzed. The etiology, clinical manifestations, diagnosis, and treatment of TO were summarized. Result: All six patients with TO were middle-aged males, confirmed by histopathological examination. The main clinical symptoms were cough, sputum, hemoptysis, chest pain, and repeated pulmonary infection. Some patients could make a preliminary diagnosis by chest CT, and bronchoscopy showed that TO mainly occurred in the trachea and the main bronchus and was more likely to invade the right bronchus. The treatment mainly includes anti-infection, phlegm-resolving, and other symptomatic treatment. Conclusion: TO is a benign disease predisposing to adults, and males are more likely to be affected. Its clinical manifestations are lack of specificity, and the cause may be related to chronic infection. Bronchoscopy combined with histopathological examination is the primary approach for the diagnosis of TO. There is no well-recognized treatment standard for TO, and the judgment of therapeutic effect is inconsistent. It is necessary to improve the understanding of this disease from a clinical perspective.

Case report of endoprosthesis -Y implantation in severe respiratory failure in the MPSII patient; comparison with literature data.

BACKGROUND: The tracheobronchomalacia is a life-threatening complication of mucopolysaccharidosis (MPS) without known effective, optimal treatment. The severe expiratory collapse of the trachea and bronchi is one of causes of the high rate of deaths in the course of airway impairment in MPSII patients. CASE PRESENTATION: Due to the adynamic tracheobronchomalacia despite of enzymatic treatment (ERT) in our MPSII patient, a life-saving tracheal bifurcated type-Y endoprosthesis (a self-expanding, metal stent for the prosthesis of tracheal and bronchial stenosis) was implanted. In the followed months, the breathing efficiency improved, but then gradual worsening, progression of bronchi occlusion at the stent border resulted in patient's death. CONCLUSION: The Y-stent implantation appears to be a short-term, life-saving solution without satisfactory long-term effects due to the progress of peripheral bronchomalacia and increased tissue proliferation and granulation, that arises during the illness' course.

Tracheobronchopathia Osteochondroplastica: Five Cases Report and Literature Review.

Tracheobronchopathia osteochondroplastica (TO) is a rare disease. Here, we report 5 TO cases treated at our hospital. Bronchoscopy showed typical multiple firm and glossy nodules in all the 5 cases. Conservative treatment effectively alleviated the symptoms. Tracheobronchopathia osteochondroplastica is a manageable disease. Awareness in clinicians is critical to avoid unnecessary treatment in patients with TO.

Successful Rescue of a Ruptured Tracheoinnominate Fistula with Extracorporeal Membrane Oxygenation, Endovascular Stents, and Debranching Surgical Bypass.

We reported a case of ruptured tracheoinnominate fistula in a 14-year-old boy with history of repeated sternotomy. Tracheostomy was performed at age 2 years. Slide tracheoplasty was done at age 13 years. He presented to outpatient clinic with episodic hemosputum. Massive blood emanated from stoma during bronchoscopy evaluation. Venous-arterial extracorporeal membrane oxygenation was installed for resuscitation. A contrast-enhanced computed tomography (CT) and angiography confirmed the diagnosis. Immediate control of bleeding was achieved by an endovascular stent graft deployed at innominate artery. Massive hemorrhage recurred on day 7. An aortic arch stent was inserted and all arch vessels debranching via supraclavicular collar excision was performed. A covered stent was used to fenestrate the aortic stent and establish antegrade blood flow to all neck vessels via left common carotid artery. The patient remained stable at 10-month follow-up. Combination of extracorporeal membrane oxygenation, endovascular intervention, and surgical bypass could be effective in treating critical patients.

Intermediate bronchial fistula caused by mediastinal drainage tube compression and fungal infection: a case report.

BACKGROUND: Intermediate bronchial fistula formation caused by mediastinal drainage tube compression and fungal infection is rare. CASE PRESENTATION: A 50-year-old male patient with type 2 diabetes was observed air filling in mediastinal drainage tube, 12 days after esophagectomy for esophageal squamous carcinoma. Based on the results of computed tomography, bronchoscopy and pathology, the diagnosis of intermediate bronchial fistula caused by mediastinal drainage tube compression and fungal infection was made. Anti-fungal drug and temporary covered metallic stent was used. After stent removed, the fistula was healed with some granulation hyperplasia. He was free from respiratory symptom during 1 year follow-up. CONCLUSION: Intermediate bronchial fistula caused by the combination of mediastinal drainage tube compression and fungal infection is rare. Timely stenting could boost the healing of fistula via granulation tissue proliferation.

Tracheobronchial involvement of relapsing polychondritis.

Recent studies show that relapsing polychondritis patients with tracheobronchial involvement are distinct from others in terms of clinical characteristics, therapeutic management, and disease evolution. Tracheobronchial involvement affects 20 to 50% of patients and may reveal the disease. It should be sought at the time of diagnosis and at each follow-up visit. Respiratory impairment is confirmed by computed tomography (CT) of the chest, including the cervical portion of the trachea, with end-inspiratory and dynamic expiratory scans, and pulmonary function tests. These investigations should be performed, even in asymptomatic patients, at the time of diagnosis, and repeated as necessary during follow-up. Bronchoscopy and a fortiori endoscopic intervention should be considered with caution and performed only by expert endoscopists after careful evaluation of the risks and benefits of such procedures, which can lead to damage or perforation of the airways and bronchospasm. Early detection and management of tracheobronchial involvement in relapsing polychondritis has significantly improved the prognosis of patients, especially with the development of interventional fiberoptic bronchoscopy. However, relapsing polychondritis-related morbidity and mortality are still elevated, particularly in tracheobronchial disease.

Use of esophageal stent for the treatment of postoperative gastrointestinal-airway fistula after esophagectomy.

A gastrointestinal-airway fistula (GAF) after esophagectomy is a very serious postoperative complication that can cause severe respiratory complications due to digestive juice inflow. Generally, GAF is managed by invasive surgical treatment; less-invasive treatment has yet to be established. We performed esophageal stent placement (ESP) in three cases of GAF after esophagectomy. We assessed the usefulness of ESP through our clinical experience. All GAFs were successfully managed by ESP procedures. After the procedure, the stent positioning and expansion were appropriately evaluated by radiological assessments over time. The stent was removed after endoscopic confirmation of fistula closure on days 8, 23, and 71. Only one patient with a long-term indwelling stent developed a manageable secondary gastrobronchial fistula as a procedure-related complication. In conclusion, ESP was shown to be a less-invasive and effective therapeutic modality for the treatment of GAF.

Tracheal injury characterized by subcutaneous emphysema and dyspnea after improper placement of a Sengstaken-Blakemore tube: A case report.

RATIONALE: Tracheal injury characterized by subcutaneous emphysema and dyspnea can occur following the use of a Sengstaken-Blakemore tube. Should tracheal injury occur, it may be possible to manage resultant airway obstruction with a tracheal stent. PATIENT CONCERNS: We describe the case of a 51-year-old patient who developed a tracheal injury when a Sengstaken-Blakemore tube was inadvertently inserted into the patient's trachea. DIAGNOSES: Liver cirrhosis, gastric-fundus variceal bleeding, tracheal injury. INTERVENTIONS: Polyglycol and tissue glue were injected intravenously, and endoscopic variceal ligation was performed. A Sengstaken-Blakemore tube was used to stop the bleeding. A covered tracheal stent was placed via fiberoptic bronchoscopy to relieve the tracheal injury due to improper placement of a Sengstaken-Blakemore tube. OUTCOMES: After placement of the tracheal stent, the patient was able to breathe spontaneously and subsequently recovered. LESSONS: Some precautions must be taken to avoid placing a Sengstaken-Blakemore tube in the trachea. If a tracheal injury occurs following misplacement of a Sengstaken-Blakemore tube, it may be possible to manage resultant airway obstruction by placing a tracheal stent.

Tuberculosis and Other Granulomatous Diseases of the Airway.

Granulomatous diseases of the airway are challenging lesions to diagnose and effectively manage not only because they are uncommon but also because they can occur in different forms, each with unique clinical and radiological characteristics. Most such lesions can be effectively managed conservatively with repeated airway dilatation, use of intraluminal stents, and specific antimicrobial treatment. The only exception is those lesions presenting with localized airway obstruction wherein surgical resection may be indicated and beneficial.

Nonoperative Endoscopic Management of Benign Tracheobronchial Disorders.

Modern thoracic surgery requires knowledge and skill in advanced bronchoscopic techniques. Rigid bronchoscopy remains a workhorse for the management of central airway obstruction. Dilation of tracheal strictures is now much simpler with the advent of the balloon dilator, which can be passed through a therapeutic bronchoscope. Numerous adjuncts, such as laser, argon beam coagulation, electrocautery, and cryotherapy, can be used to improve airway patency. There are now numerous stenting options for strictures that require stenting to maintain airway patency.

Airway management changes associated with rising radiologic incidence of external laryngotracheal injury.

BACKGROUND: Traumatic laryngeal injuries are uncommon life-threatening injuries that require prompt, rational management of a potentially precarious airway. It is unclear whether the current incidence of laryngotracheal injury is due to enhanced injury detection or increased occurrence. The objective of this study was to evaluate the relations between diagnostic imaging with both initial airway management and surgical treatment in patients with external laryngotracheal injuries (ELTIs) in Alberta. METHODS: In this large-scale population-based analysis, we used regional health databases containing inpatient admissions, emergency department visits and trauma service activations employing International Classification of Diseases diagnostic codes to identify all ELTIs diagnosed from Apr. 1, 1995, to Dec. 31, 2011, in adults (age ≥ 16 yr). We evaluated health records and diagnostic imaging for injury features, airway management, operative interventions and hospital length of stay (LOS). RESULTS: Eighty-nine patients met the inclusion criteria. The incidence of ELTIs increased over time, paralleling a rise in detection during the period incorporating greater computed tomography (CT) use (p = 0.002). Endotracheal tube intubation was performed in 8/30 cases (27%) in the pre-CT era, compared to 38/59 cases (64%) in the post-CT era (p = 0.001); the use of surgical intervention remained consistent. The largest contributors to increased endotracheal tube placements were the emergency department and emergency medical services. No change in survival was detected, but mean LOS among patients admitted for minor, isolated ELTIs increased by 2.3 (95% confidence interval 0.14-4.8) days (p = 0.06), mostly for patients admitted under critical care for mechanical ventilation. CONCLUSION: Management of ELTIs shifted from predominantly conservative airway monitoring to endotracheal tube intubation over the study period in spite of no clinically significant change in injury severity or operative intervention frequency. The location of endotracheal tube placement suggests less comfort with ELTI among first-responder and emergency personnel.

CONTEXTE: Les lésions traumatiques du larynx sont des blessures rares qui peuvent être mortelles et nécessitent une prise en charge rapide et efficiente, en raison de l'état potentiellement précaire des voies respiratoires. On ignore si l'incidence actuelle des lésions laryngo-trachéales est attribuable à une amélioration de la détection ou à une augmentation de la fréquence réelle. Cette étude avait pour but d'évaluer le lien entre l'imagerie diagnostique, et la prise en charge initiale des voies respiratoires ainsi que le traitement chirurgical chez des patients ayant subi des lésions laryngo-trachéales externes (LLTE) en Alberta. MÉTHODES: Dans le cadre de cette analyse de grande envergure basée sur une population, nous avons interrogé des bases de données régionales sur les hospitalisations, les consultations aux services d'urgence et la prestation de services de traumatologie. Nous nous sommes servis des codes diagnostiques de la Classification statistique internationale des maladies pour repérer tous les cas de LLTE diagnostiqués entre le 1er avril 1995 et le 31 décembre 2011 chez des adultes (16 ans et plus). Nous avons examiné les dossiers de santé et les résultats d'imagerie diagnostique pour en extraire des données sur les caractéristiques des lésions, la prise en charge des voies respiratoires, les interventions chirurgicales et la durée de séjour à l'hôpital. RÉSULTATS: Au total, 89 patients répondaient aux critères d'inclusion. L'incidence des LLTE a augmenté au fil du temps; en parallèle, l'utilisation répandue de la tomographie par ordinateur a entraîné une augmentation de la détection de ces lésions (p = 0,002). Une intubation trachéale a été réalisée chez 8/30 patients (27 %) pendant la période prétomographie, et chez 38/59 patients (64 %) pendant la période post-tomographie (p = 0,001); le recours à la chirurgie est demeuré constant. L'augmentation du nombre d'intubations est principalement attribuable aux interventions effectuées par le personnel ambulancier et par les services d'urgence. Aucun changement du taux de survie n'a été enregistré; toutefois, la durée de séjour moyenne des patients hospitalisés en raison de LLTE mineures et isolées a augmenté de 2,3 jours (intervalle de confiance à 95 % : 0,14-4,8; p = 0,06), surtout pour les patients admis aux soins intensifs pour recevoir une ventilation mécanique. CONCLUSION: Durant la période à l'étude, la prise en charge des LLTE est passée d'un suivi essentiellement conservateur des voies respiratoires à la prépondérance de l'intubation trachéale, bien qu'aucun changement significatif n'ait été observé quant à la gravité des lésions ou à la fréquence des interventions chirurgicales. Le contexte où ont lieu les intubations laisse croire que le personnel ambulanciers et les premiers intervenants sont moins à l'aise de prendre en charge les LLTE.

[Tracheal Injury Successfully Treated without Surgery;Report of a Case].

A 73-year-old man was injured after crashing his automobile into a concrete wall, and was admitted to our hospital with breathing difficulties. Chest computed tomography showed extensive mediastinal emphysema, right traumatic pneumothorax, and a suspected tracheal injury at the membranous region. Because of the associated tracheomalacia, the site of the tracheal injury was difficult to find by bronchoscopy. In addition, as the patient's respiratory condition was stable, surgical treatment was not chosen. Due to the continuous air leakage from the right lung, the patient underwent surgery after mediastinal and subcutaneous emphysema improved. The postoperative course was uneventful.