Next-Generation Sequencing as an Auxiliary Tool in Pediatric Laryngeal Lymphoma Diagnosis.

Lymphomatous involvement of the larynx is a rare entity. We present a case of atypical laryngotracheitis as the initial manifestation of non-Hodgkin's lymphoma in a pediatric patient. The diagnosis was aided through the use of microbial cell-free DNA (mcfDNA) testing, which detected the presence of Epstein-Barr virus in the patient's plasma. This enabled the consideration of an Epstein-Barr virus-related lymphoproliferative process, leading to additional workup and the final diagnosis of lymphoma. To our knowledge, this is the first case of mcfDNA testing leading not simply to an infectious organism, but further to a new oncologic diagnosis. Plasma mcfDNA testing has the potential to inform clinical practice beyond classic infectious disease manifestations. In this article, we review both the possible future applications and the areas of further investigation that remain.

Pseudomembranous Tracheobronchitis With Severe Tracheal Stenosis and Masked Bronchial Obstruction.

BACKGROUND: Pseudomembranous tracheobronchitis (PMTB) is a rare condition characterized by the formation of endobronchial pseudomembranes. PMTB overlaps with necrotizing tracheobronchitis or plastic bronchitis. The reported infectious etiology mainly includes invasive aspergillosis. PMTB can cause serious airway obstruction; however, urgent tracheotomy is rarely required. CASE REPORT: A 46-year-old woman was transferred to the emergency department (ED) with a 1-week history of progressive dyspnea and cough that was preceded by fever and sore throat. She was previously healthy except for a 20-year history of mild palmoplantar pustulosis. Stridor was evident. Nasolaryngoscopy performed in the ED revealed severe tracheal stenosis caused primarily by mucosal edema and secondarily by pseudomembranes. Initially, tracheitis was considered the sole cause of dyspnea. Although she underwent urgent tracheotomy to prevent asphyxia, her respiration deteriorated progressively. Bronchoscopy revealed massive pseudomembranes obstructing the bilateral bronchi, which led to the clinical diagnosis of PMTB. Subsequent toilet bronchoscopy markedly improved her ventilation. The causative pathogen was not identified despite extensive work-up, including molecular biological testing. Histopathologic examination of the pseudomembranes revealed fibrin with abundant neutrophils, which was consistent with PMTB. Associated conditions, including immunodeficiency, were not found. Her condition improved with antibiotics and repeated toilet bronchoscopy. WHY SHOULD AN EMERGENCY PHYSICIANS BE AWARE OF THIS?: PMTB is an important differential diagnosis of airway emergencies. PMTB can present with critical edematous tracheal stenosis and masked bronchial pseudomembranous obstruction. Emergency physicians should include PMTB in the differential diagnosis in adult patients with acute central airway obstruction because it requires prompt multimodal treatment.

Caracterización de las infecciones en niños portadores de cánula de traqueotomía: revisión sistemática de la literatura / Characterization of infections in children with Tracheostomy Tube: A Systematic Review of the Literature

Resumen Introducción: actualmente los profesionales de la salud se enfrentan al manejo de las vías aéreas artificiales en grupos pediátricos, esto requiere de cuidados delicados y mucha atención para detectar, establecer y manejar situaciones apremiantes; por esta razón, existe un mayor riesgo de aparición de infecciones bacterianas traqueopulmonares. El objetivo del estudio fue analizar la caracterización de las infecciones en pacientes pediátricos portadores de cánula de traqueotomía en las diferentes publicaciones científicas. Materiales y métodos: se realizó una revisión sistemática mediante la búsqueda de la literatura existente entre los años 2015-2020 en las bases de datos Elsevier, PubMed, Google Académico y SciELO, teniendo en cuenta los criterios de inclusión artículos en idioma inglés, español y población de edad entre los 0-15 años con infección de cánula de traqueotomía en los años 2015-2020. Resultados: de 258 artículos distribuidos en las bases de datos, se seleccionaron 21 artículos que cumplían con los criterios de inclusión. Conclusiones: a pesar de que en la actualidad existan criterios clínicos, factores de riesgo y pruebas de laboratorio asociados a infecciones de la cánula postraqueotomía en pacientes pediátricos, se requiere mayor investigación para definir las guías clínicas de manejo en la toma de decisiones médicas. Asimismo, se consideró como limitación importante la cantidad de literatura existente con respecto al tema.

Abstract Introduction: Currently, health professionals face the management of artificial airways in pediatric groups, this requires delicate care and a lot of attention to detect, establish and manage pressing situations, which is why there is a greater risk of tracheo-pulmonary bacterial infections. The objective was to analyze the characterization of infections in pediatric patients with tracheostomy tubes in the different scientific publications. Method: A systematic review of the literature was carried out between the years 2015-2020 in Elsevier, PubMed, Google Academic and SciELO databases, taking into account the inclusion criteria of the population aged 0-15 years in the years 2015-2020. The amount of existing literature on the subject was considered an important limitation. Results: From 258 articles distributed in the databases, 21 articles were selected that met the inclusion criteria. Conclusions: Although there are currently clinical criteria, risk factors and laboratory tests associated with infections of the post-tracheotomy tube in pediatric patients, further research is required to define clinical guidelines for management in medical decision-making.

Analysis of the Clinical Features of Tracheobronchial Fungal Infections with Tumor-Like Lesions.

BACKGROUND: Tracheobronchial fungal infections (TBFI) cause life-threatening complications in immunocompromised hosts but are rarely reported. Misdiagnosis and delayed antifungal treatment are associated with the high mortality rate of patients with TBFI. OBJECTIVES: This study analyzed the bronchoscopic features of TBFI and their roles in the early diagnosis of TBFI. METHODS: The demographic, clinical, radiologic, and bronchoscopic data of 53 patients diagnosed with TBFI in our department during a 15-year period were retrospectively analyzed. RESULTS: Most of the TBFI patients were male, and mass was the most common radiologic abnormality. Obvious predilection in primary bronchus distributions was observed. 41.9% of the 43 Aspergillus tracheobronchitis (AT) patients, 70% of the 10 tracheobronchial mucormycosis (TM) patients, and 100% of the 3 endobronchial cryptococcosis patients had been misdiagnosed as having cancer on bronchoscopy because of the presence of tumor-like lesions. The most common features of AT were bronchial occlusion with a mass or mucosal necrosis, bronchial stenosis with mucosal hyperplasia, or uneven mucosa. The main descriptions of TM were bronchial stenosis or obstruction due to mucosal necrosis, uneven mucosa, or a mass. The endoscopic characteristics of endobronchial cryptococcosis included occlusion due to uneven mucosa or mass, or external compressive stricture. CONCLUSION: Immunocompromised patients and immunocompetent patients with underlying disease displaying tumor-like lesions on bronchoscopy should be differentially diagnosed with cancer. Bronchial biopsy is indispensable for the early diagnosis of TBFI.

Fatal necrotic tracheitis by Aviadenovirus in captive Alagoas curassows (Pauxi mitu) extinct from the wild.

Extinct from nature, captive young Alagoas curassows (Pauxi mitu) were found agonizing or dead with respiratory disease. Intranuclear inclusion bodies were found in the epithelia of the trachea, associated with marked necrotic tracheitis. An Aviadenovirus was isolated in chicken eggs and characterized genetically with 99% identity to the fowl Aviadenovirus A, as based on the hexon protein gene. This is the first report of respiratory disease caused by Aviadenovirus in any cracid species in Brazil, recommending for stricter biosecurity in the conservation premises. RESEARCH HIGHLIGHTS Fatal tracheitis in curassows extinct from nature was associated with Aviadenovirus A. Seven-month-old Alagoas curassows (Aves: Cracidae) died with haemorrhagic tracheitis. Aviadenovirus A with 99% identity to fowl adenovirus 1 was detected in dead curassows. Fatal tracheitis by Aviadenovirus was described in Pauxi mitu (Aves: Cracidae).

Pediatric Bacterial Tracheitis-A Variable Entity: Case Series with Literature Review.

OBJECTIVE: To review the presentation and treatment of children diagnosed with bacterial tracheitis at our institution and to review the available literature focusing on key presenting symptoms and clinical outcomes of children diagnosed with bacterial tracheitis. STUDY DESIGN: Case series with literature review. SETTING: Tertiary children's hospital and available literature. SUBJECTS AND METHODS: Case series of children with bacterial tracheitis retrospectively reviewed at a tertiary children's hospital. Those with a tracheostomy or those who developed bacterial tracheitis as a complication of prolonged intubation were excluded. RESULTS: Thirty-six children were identified (mean ± SD age, 6.7 ± 4.5 years). The most common presenting symptom was cough (85%), followed by stridor (77%) and voice changes/hoarseness (67%). A concurrent viral illness was found for 55%, and the most common bacteria cultured was methicillin-sensitive Staphylococcus aureus. Pediatric intensive care admission occurred for 69%, and 43% required intubation. No patient required tracheostomy. One patient (2.7%) died secondary to airway obstruction and subsequent respiratory arrest. Four patients had recurrence of bacterial tracheitis 4 to 12 months following their initial presentation. CONCLUSION: Bacterial tracheitis is an uncommon condition with an atypical presentation and variable clinical course but serious consequences if left unrecognized. Staphylococcus is the most common bacteria identified, and many patients will have a prodromal viral illness. Changes in patient epidemiology and presentation may have occurred over time.

Autoimmune Tracheal Cartilage Inflammation Responsive to Anti-TNF-α Therapy.

Tracheal inflammation, or tracheitis, is a pathologic process that can occur secondary to a number of systemic inflammatory diseases, or it may be idiopathic in nature. Regardless of the underlying etiology, tracheitis can, in its most severe form, be life-threatening, thus making its treatment an area of interest. Our case is one of a 50-year-old man with a remote history of inflammatory bowel disease achieving clinical cure following surgical resection who presented with progressive dyspnea due to tracheal stenosis that was presumed secondary to an autoimmune and inflammatory etiology. His disease was initially refractory to recurrent surgical interventions. He ultimately achieved clinical improvement with a combination of methotrexate and the tumor necrosis factor alpha (TNF-α) inhibitor, adalimumab. While both clinical trials and standardized treatment guidelines are lacking in this domain, this case illustrates a potential role for TNF-α inhibitors in the treatment of inflammatory tracheitis, irrespective of the underlying etiology.

Terapêutica antibiótica na traqueobronquite associada à ventilação mecânica: uma revisão da literatura / Antibiotic therapy in ventilator-associated tracheobronchitis: a literature review

RESUMO O conceito de traqueobronquite associada à ventilação mecânica é controverso, e sua definição não é unanimemente aceita, sobrepondo-se, muitas vezes, à da pneumonia associada à ventilação mecânica. A traqueobronquite associada à ventilação mecânica tem incidência semelhante à da pneumonia associada à ventilação mecânica, com elevada prevalência de agentes multirresistentes isolados, condicionando um aumento do tempo de ventilação mecânica e de internação, ainda que sem impacto na mortalidade. A realização de culturas quantitativas pode permitir melhor definição diagnóstica da traqueobronquite associada à ventilação mecânica, possivelmente evitando o sobrediagnóstico desta entidade. Uma das maiores dificuldades na diferenciação entre traqueobronquite associada à ventilação mecânica e pneumonia associada à ventilação mecânica reside na exclusão de um infiltrado pulmonar por meio da radiografia do tórax; também podem ser necessárias a tomografia computadorizada torácica, a ultrassonografia torácica ou ainda a colheita de amostras invasivas. A instituição de terapêutica antibiótica sistêmica não demonstrou melhorar o impacto clínico da traqueobronquite associada à ventilação mecânica, nomeadamente na redução do tempo de ventilação mecânica, de internação ou mortalidade, apesar da eventual menor progressão para pneumonia associada à ventilação mecânica, ainda que existam dúvidas relativas à metodologia utilizada. Deste modo, considerando a elevada prevalência da traqueobronquite associada à ventilação mecânica, o tratamento desta entidade, por rotina, resultaria em elevada prescrição antibiótica sem benefícios claros. No entanto, sugerimos a instituição de terapêutica antibiótica em doentes com traqueobronquite associada à ventilação mecânica e choque séptico e/ou agravamento da oxigenação, devendo ser realizados simultaneamente outros exames auxiliares de diagnóstico para exclusão da pneumonia associada à ventilação mecânica. Após esta revisão da literatura, entendemos que uma melhor diferenciação entre traqueobronquite associada à ventilação mecânica e pneumonia associada à ventilação mecânica pode diminuir, de forma significativa, a utilização de antibióticos em doentes críticos ventilados.

ABSTRACT The concept of ventilator-associated tracheobronchitis is controversial; its definition is not unanimously accepted and often overlaps with ventilator-associated pneumonia. Ventilator-associated tracheobronchitis has an incidence similar to that of ventilator-associated pneumonia, with a high prevalence of isolated multiresistant agents, resulting in an increase in the time of mechanical ventilation and hospitalization but without an impact on mortality. The performance of quantitative cultures may allow better diagnostic definition of tracheobronchitis associated with mechanical ventilation, possibly avoiding the overdiagnosis of this condition. One of the major difficulties in differentiating between ventilator-associated tracheobronchitis and ventilator-associated pneumonia is the exclusion of a pulmonary infiltrate by chest radiography; thoracic computed tomography, thoracic ultrasonography, or invasive specimen collection may also be required. The institution of systemic antibiotic therapy does not improve the clinical impact of ventilator-associated tracheobronchitis, particularly in reducing time of mechanical ventilation, hospitalization or mortality, despite the possible reduced progression to ventilator-associated pneumonia. However, there are doubts regarding the methodology used. Thus, considering the high prevalence of tracheobronchitis associated with mechanical ventilation, routine treatment of this condition would result in high antibiotic usage without clear benefits. However, we suggest the institution of antibiotic therapy in patients with tracheobronchitis associated with mechanical ventilation and septic shock and/or worsening of oxygenation, and other auxiliary diagnostic tests should be simultaneously performed to exclude ventilator-associated pneumonia. This review provides a better understanding of the differentiation between tracheobronchitis associated with mechanical ventilation and pneumonia associated with mechanical ventilation, which can significantly decrease the use of antibiotics in critically ventilated patients.

Nodular tracheobronchitis in a patient with lymphoma: an unusual presentation of viridans streptococcal respiratory tract infection.

Bacterial tracheobronchitis without lung parenchymal involvement is extremely rare in adults, except in patients who are intubated or mechanically ventilated. We present a case of nodular tracheobronchitis caused by viridans streptococci in a non-ventilated lymphoma patient. To our knowledge, this is the first report of viridans streptococcal infection that has caused nodular tracheobronchitis.

Association between follicular tracheitis and gastroesophageal reflux.

OBJECTIVE: Follicular tracheitis (also known as tracheal cobblestoning) is an entity that is poorly described and of unclear significance. The objective of this study was to better define follicular tracheitis and determine the association between the clinical finding of follicular tracheitis on bronchoscopy and objective evidence of gastroesophageal reflux disease. METHODS: Retrospective chart review of children with recurrent croup having undergone a rigid bronchoscopy and an investigation for gastroesophageal reflux between 2001 and 2013. RESULTS: 117 children with recurrent croup children age 6-144 months were included in the study. Follicular tracheitis was noted on 41% of all bronchoscopies. Fifty-nine percent of all children who underwent bronchoscopy were diagnosed with gastroesophageal reflux on at least one investigation. Forty-nine of 117 children underwent a pH probe study, and 51% were found to have evidence of reflux on this study. Nine children were diagnosed with eosinophilic esophagitis. Three patients underwent a biopsy of the follicular tracheitis lesions, which revealed chronic inflammation. There was no evidence of an association between findings of follicular tracheitis and a positive test for gastroesophageal reflux (p=0.52) or a positive pH probe study (p=0.64). There was no association between follicular tracheitis and subglottic stenosis (p=0.33) or an history of asthma and/or atopy (p=0.19). CONCLUSION: In children with recurrent croup, follicular tracheitis remains an unspecific finding associated with an inflammatory disorder of unknown etiology.

Laryngotracheal mucormycosis: Report of a case.

Airway mucormycosis is a deadly opportunistic infection that affects immunocompromised persons, particularly diabetics and those undergoing chemotherapy. Although it is typically a pulmonary or sinonasal infection, mucormycosis can affect the larynx and trachea, with devastating results. We report the case of a 46-year-old man with human immunodeficiency virus infection, hepatitis C infection, neurosyphilis, and recently diagnosed Burkitt lymphoma who presented with dysphonia and stridor after receiving one dose of intrathecal chemotherapy. Flexible laryngoscopy detected the presence of fibrinous material that was obstructing nearly the entire glottis. Surgical debridement revealed a firm mucosal attachment; there was little bleeding when it was removed. After debridement, the patient's dyspnea improved only to recur 2 days later. After an awake tracheotomy, laryngoscopy and bronchoscopy identified necrosis extending from the supraglottic area to the carina tracheae. Biopsies demonstrated hyphal architecture consistent with mucormycosis. Despite continued debridements, the fibrinous material reaccumulated. The patient was placed in hospice care; his airway remained patent, but he died from other causes several weeks after presentation. The management of airway mucormycosis is challenging and complex. Fungal airway infections should be considered in the differential diagnosis of an immunosuppressed patient who presents with dyspnea, dysphonia, and vocal fold immobility. Timely diagnosis and management are critical for a successful outcome, although the prognosis is poor if the infection is widespread, even with the best of efforts.

A 60-year-old woman with cough, dyspnea, and atelectasis 19 years after liver transplant.

A 60-year-old black woman presented with nonproductive cough of 1-month duration. She had also experienced rapidly progressive dyspnea for 1 week and one bout of vomiting a day before presentation. Her symptoms had failed to improve with a course of amoxicillin-clavulanate. Her medical history was significant for diabetes mellitus and liver transplant 19 years earlier for hepatitis C cirrhosis, for which she was receiving tacrolimus and mycophenolate. She was a current smoker with 40 pack-years of smoking history.

Tracheobronchitis with dyspnea in a patient with ulcerative colitis.

We herein report the case of a 42-year-old man with a one-year history of ulcerative colitis who presented with exacerbated bloody diarrhea, a productive cough and increasing breathing difficulties. Colonoscopy revealed typical deep ulcers in the rectosigmoid colon and atypical multiple sucker-like ulcers in the transverse colon, and computed tomography of the chest demonstrated wall thickening of the trachea and bronchi. In addition, bronchoscopy showed ulcers in the trachea, and histopathology disclosed findings of necrosis and inflammation of the subepithelial tissue of the trachea. Based on these findings, the patient's respiratory symptoms were strongly suspected to be due to ulcerative colitis-related tracheobronchitis. Treatment with systemic corticosteroids subsequently resulted in a rapid clinical improvement.

Treatment of Disseminated Talaromyces marneffei with Tracheal Infection: Two Case Reports.

Talaromyces marneffei infection involving the trachea presents as diffuse irregular nodules that grow on the tracheal lumen and/or rarely present as concurrent severe eosinophilia. Herein, we report two patients without HIV infection whose main tracheal and/or principal bronchi were involved by T. marneffei infection, which manifested as diffuse proliferative nodules. In case 1, the infection primarily affected the main trachea, resulting in structural cartilage damage, tracheal stenosis, and tracheal absence. In case 2, there were diffuse proliferative nodules in the trachea and bronchi with marked eosinophilia. The final diagnosis was made based on a positive culture from bronchiolar lavage fluid, skin, and tracheal polyps. Case 1 was administered antifungal treatment combined with surgery, but relapse occurred during a 3-month follow-up period. Case 2 was treated by intravenous liposomal amphotericin B combined with atomized inhaled liposomal amphotericin B, and he later showed improvement; there was no relapse during the 12-month period of antifungal treatment. Importantly, atomized inhaled amphotericin B antifungal treatment for T. marneffei infection has never been previously reported.

Ulcerative lesions as a rare cause of laryngotracheitis in the pediatric population.

OBJECTIVE: The goal of this study is to describe a unique finding of ulcerative lesions of the larynx in two pediatric patients presenting with prolonged acute laryngotracheitis and compare to previously described reports to determine the typical clinical picture, need for intervention, and management model. METHODS: We present two cases of ulcerative lesions of the larynx in immunocompetent children, one with PCR positive HSV, which presented as severe croup requiring intensive care unit admission. Literature review was completed to assess for current knowledge of this entity. Our cases are discussed in the context of previously reported cases of HSV laryngotracheitis. Descriptive analysis was completed focusing on presentation, physical exam findings, treatment, length of therapy, and outcomes. RESULTS: Literature review uncovered six case reports including 10 individual cases of prolonged croup with findings of HSV laryngitis and one retrospective review describing 15 cases of prolonged croup found to be caused by ulcerative laryngitis. All patients underwent direct laryngoscopy and bronchoscopy for evaluation. Analysis was completed comparing the studies to our patients with significant findings including high intubation rate of 77%, ulcerative stomatitis in 63%, and treatment with antiviral medication directed at HSV in 85% with improvement in symptoms. CONCLUSION: It is important to consider HSV as a possible pathogen in cases of prolonged or atypical croup. Laryngoscopy should be used for diagnostic intervention and identification of ulcerative lesions. Stomatitis may be an indication for earlier direct inspection. Treatment with anti-viral therapy and with discontinuation or taper of steroid is suggested.

An unusual case of invasive Aspergillus ulcerative tracheobronchitis without involvement of lung parenchyma in a post-renal transplant patient.

We present the case of a 54-year-old male, who presented with respiratory complaints four months after he underwent renal transplantation. Bronchoscopy showed ulcerated mucosa of the left main bronchus and computed tomography (CT) of the thorax showed foci of air within the bronchial wall. A biopsy from the lesion showed septate fungal hyphae, dichotomously branching at acute angles. A locally invasive Aspergillus ulcerative tracheobronchitis with no parenchymal involvement is an important cause of tracheobronchitis in post-renal transplant patients. An early diagnosis and institution of appropriate treatment can improve the outcome. A combination treatment of caspofungin and voriconazole can be considered if patient is not responding to voriconazole alone.