Inferring the diagnostic potential of 18F-FDG-PET/CT in post-renal transplantation from a unique case harboring multiple rare complications.

Renal transplantation is undoubtedly an effective treatment for patients with end-stage renal disease, but it is certainly not a cure. Patients require lifelong immunosuppression to maintain optimal allograft function, and post-operative risk complications such as cancer in the transplant recipient cannot be ignored. Besides, infection is a silent complication that follows transplantation. Relatedly, herein, we present a report of a 40-year-old patient who underwent renal transplantation and promptly developed a diffuse large B-cell tumor in the liver and Aspergillus infection in the trachea. In addition, an inflammatory necrotizing granuloma was also observed in the muscles. Of importance, we also described the potential of 18F-FDG-PET/CT, which was instrumental in monitoring and evaluating these relevant post-operative complications in this rare case.

Aspergillus tracheobronchitis with Birt-Hogg-Dubè syndrome as a rare cause of chronic cough.

BACKGROUND: Aspergillus tracheobronchitis (ATB) is confined as a condition of chronic superficial infection of tracheobronchial tree. Its diagnosis is difficult due to atypical manifestations and low detective rate of Aspergillus thus far. CASE PRESENTATION: Herein, we presented a 45-year-old male patient with a sole chronic productive cough for five years referred to our cough specialist clinic. Chest high-resolution computed tomography showed multiple lung cysts predominantly located in the subpleural lesions and near the mediastinum. Neither bacteria nor fungi were identified by sputum culture. However, metagenomic next-generation sequencing in sputum detected Aspergillus fumigatus DNA. The genetic testing of whole blood suggested the germline mutation of the tumor suppressor gene folliculin, supporting a diagnosis of Birt-Hogg-Dubé (BHD) syndrome. His productive cough symptom significantly improved after receiving itraconazole treatment for 2 months. After discontinuation of antifungal treatment, there was no relapse for four months follow-up. A diagnosis of ATB with BHD syndrome was eventually established in this patient. CONCLUSION: ATB should be considered in any patient with prolonged unexplained productive cough. Next-generation sequencing technologies may be useful to identify ATB which is uncommon and easily ignored in clinical practice.

[Necrotizing tracheobronchitis due to invasive aspergillosis in an immunocompetent patient]. / Traqueobronquitis necrotizante secundaria a aspergilosis invasiva en paciente inmunocompetente.

Aspergillus tracheobronchitis is a rare form of invasive aspergillosis reported exceptionally in the immunocompetent patient. Its diagnosis is difficult, and the treatments proposed so far are of little effectiveness, all of which constitute a real problem for the health team. We present the case of an immunocompetent 28 years old woman, with no epidemiological background, who developed necrotizing tracheobronchitis due to invasive aspergillosis and received voriconazole with local instillation by bronchoscopy with a good response.

La traqueobronquitis aspergilar es una forma poco frecuente de aspergilosis invasiva reportada excepcionalmente en el paciente inmunocompetente. Su diagnóstico es difícil, y los tratamientos propuestos hasta ahora son de escasa efectividad, todo lo cual constituye un verdadero problema para el equipo de salud. Presentamos el caso de una paciente de 28 años, inmunocompetente y sin antecedentes epidemiológicos, que desarrolló traqueobronquitis necrotizante por aspergilosis invasiva y recibió tratamiento con voriconazol con instilación local por broncoscopia con buena respuesta.

Traqueobronquitis necrotizante secundaria a aspergilosis invasiva en paciente inmunocompetente / Necrotizing tracheobronchitis due to invasive aspergillosis in an immunocompetent patient

Resumen La traqueobronquitis aspergilar es una forma poco frecuente de aspergilosis invasiva reportada excepcionalmente en el paciente inmunocompetente. Su diagnóstico es difícil, y los tratamientos propuestos hasta ahora son de escasa efectividad, todo lo cual constituye un verdadero problema para el equipo de salud. Presentamos el caso de una paciente de 28 años, inmunocompetente y sin antecedentes epidemiológicos, que desarrolló traqueobronquitis necrotizante por aspergilosis invasiva y recibió tratamiento con voriconazol con instilación local por broncoscopia con buena respuesta.

Abstract Aspergillus tracheobronchitis is a rare form of invasive aspergillosis reported exceptionally in the immunocompetent patient. Its diagnosis is difficult, and the treatments proposed so far are of little effectiveness, all of which constitute a real problem for the health team. We present the case of an immunocompetent 28 yearsold woman, with no epidemiological background, who developed necrotizing tracheobronchitis due to invasive aspergillosis and received voriconazole with local instillation by bronchoscopy with a good response.

Acute mediastinitis associated with tracheobronchial tuberculosis and aspergillosis: a case report and literature review.

Acute mediastinitis (AM) is a rare but life-threatening disease. Here, we report a case of AM secondary to endobronchial tuberculosis (EBTB) and pseudomembranous Aspergillus tracheobronchitis (PMATB) co-infection. EBTB was confirmed by tissue culture for Mycobacterium tuberculosis and GeneXpert MTB/RIF (Cepheid, Sunnyvale, CA, USA) detection (simultaneous detection of M. tuberculosis and resistance to rifampin) using endobronchial biopsies; PMATB was confirmed by histopathology. Even with antibiotic treatment and systemic support treatment, the patient died of massive hemoptysis on day 10 after admission. When immunocompromised hosts have AM, especially with central airway involvement, EBTB and aspergillosis should be considered potential causes. Bronchoscopy is helpful for rapid diagnosis and administering precise treatment.

Severe Fever with Thrombocytopenia Syndrome Complicated with Pseudomembranous Aspergillus Tracheobronchitis in a Patient without Apparent Risk Factors for Invasive Aspergillosis.

Severe fever with thrombocytopenia syndrome (SFTS) is a tick-borne infectious disease. A 91-year-old woman was admitted to our intensive-care unit with SFTS, and she developed dyspnea with wheezes 5 days after admission. Bronchoscopy showed scattered white mold in her central airway. An airway tissue biopsy and culture of bronchial lavage fluid revealed fungal hyphae in the necrotic tissue, confirmed as Aspergillus fumigatus. She was thus diagnosed with pseudomembranous aspergillus tracheobronchitis. She had no common risk factors for invasive aspergillosis (IA). Patients with SFTS, even those without apparent risk factors for IA, may be at risk of developing IA.

[Obstructive tracheobronchitis due to Aspergillus fumigatus in an immunocompetent patient]. / Traqueobronquitis obstructiva por Aspergillus fumigatus en un paciente inmunocompetente.

BACKGROUND: Aspergillus tracheobronchitis (ATB) is an uncommon type of invasive pulmonary aspergillosis in which fungal involvement is limited to the tracheobronchial tree. While the more severe forms, such as pseudomembranous and ulcerative ATB, occur almost exclusively in immunocompromised patients, the milder obstructive form may occur in patients without immune deficiency. CASE REPORT: The case of a 32 year-old man with no previous history of illness, who was evaluated for recurrent right lower lobe pneumonia, is presented. Microbiological sputum studies revealed growth of Serratia marcescens, and a limited growth of Aspergillus fumigatus, the latter interpreted as a contaminant in the specimen. Bronchoscopy revealed a dense mucous plug at level B10 of the right lower lobe, which could not be removed; no other macroscopic findings of interest were observed. During his hospital admission, the patient expectorated the mucous plug and had a significant subsequent bronchorrhoea. A substantial number of colonies of A. fumigatus grown in the sputum cultures. The patient was given voriconazole, leading to a clinical resolution, with no recurrences. CONCLUSIONS: Obstructive ATB is characterised by the excessive production of thick, hyphae-laden mucus, which can obstruct the airway lumen and generate relapsing post-obstructive pneumonias. It is important to consider this diagnosis in immunocompetent patients with recurrent respiratory infections and who show repeated isolation of Aspergillus colonies in the sputum, even in small quantities.

Analysis of the clinical features of invasive bronchopulmonary aspergillosis.

INTRODUCTION: Early diagnosis of invasive bronchopulmonary aspergillosis (IBPA) is difficult, so the mortality rate is high. OBJECTIVE: To discuss the clinical features of IBPA. METHODS: We retrospectively analyzed the clinical features, imaging findings, laboratory test, diagnosis and treatment of 115 patients with IBPA diagnosed from October 2004 to June 2013 in Zhongshan Hospital, Fudan University. RESULTS: The main clinical manifestations were cough in 58 patients (50.4%), expectoration in 37 patients (32.2%), fever in 18 patients (15.7%), shortness of breath in 26 patients (22.6%), hemoptysis in 26 patients (22.6%) and chest pain in 7 patients (6.0%). The main CT findings were nodules in 35 patients (30.4%), consolidation shadows or patchy shadows in 62 patients (53.9%) and cavity in 14 patients (12.2%). Percutaneous pulmonary biopsy was conducted in 25 patients (21.7%), TBLB in 58 patients (50.4%) and thoracoscopic surgery in 32 patients (27.8%). The positive rate of GM test was 73.5% (72/98). Thirty patients who received lobectomy were followed up for 1-3 years. Fifty-five patients who received monotherapy with antifungal agents were followed up for 1-3 years, and 12 patients were healed. The lesions for 23 patients were obviously absorbed, 10 patients had aggravation and two patients died. CONCLUSIONS: The clinical manifestations of IBPA were unspecific. The main symptoms were cough and expectorate. Patients with different immunologic function had different imaging findings. The halo sign and new moon sign for diagnosis was not as common as reported. Interventional therapy under bronchoscope is very important for patients with ATB.

Unique Case of Pseudomembranous Aspergillus Tracheobronchitis: Tracheal Perforation and Horner's Syndrome.

Pseudomembranous aspergillus tracheobronchitis is an uncommon form of invasive pulmonary aspergillosis, and it is generally seen in immunocompromised patients. We report about a mildly immunocompromised case with pseudomembranous aspergillus tracheobronchitis, which caused tracheal perforation, and Horner's syndrome. A 44-year-old female with uncontrolled diabetes mellitus, complaining of fever and dyspnea, was admitted to the hospital. She was hospitalized with community-acquired pneumonia and diabetic ketoacidosis. Insulin infusion and empirical antibiotics were firstly commenced. Bronchoscopy showed left vocal cord paralysis with extensive whitish exudative membranes covering the trachea and the main bronchi. Liposomal amphotericin B was added due to the probability of fungal etiology. Mucosal biopsy revealed aspergillus species. Second bronchoscopic examination demonstrated a large perforation in the tracheobronchial system. Despite all treatments, respiratory failure developed on the 25th day and the patient died within 2 days. Pseudomembranous aspergillus tracheobronchitis is fatal in about 78 % of all cases despite appropriate therapy. Early diagnosis and efficient antifungal therapy may improve the prognosis.

Lesson of the month 1: A cough that doesn't fit the mould.

Aspergillus disease has a wide spectrum of manifestations within the lungs; however invasive Aspergillus is most commonly associated with immunocompromise or pre-existing respiratory disease. Here we present a case of Aspergillus tracheobronchitis causing right middle lobe collapse, masquerading as late-onset asthma in a patient with no pre-existing risk factors following massive inhalation of Aspergillus spores from working with compost. This case highlights the importance of having a high index of suspicion for Aspergillus-related disease even in those with no traditional risk factors.

Airway Obstruction and Respiratory Failure Due to Aspergillus Tracheobronchitis / 대한구급학회지

Invasive aspergillosis is a serious threat and a leading cause of death in immunocompromised patients. Aspergillus tracheobronchitis is an infrequent but severe form of invasive pulmonary aspergillos in which the fungal infection is entirely or predominantly confined to the tracheobronchial tree. We report an extraordinary case of acute airway obstruction and respiratory failure due to Aspergillus tracheobronchitis in an immunocompromised patient. Fiberoptic bronchoscopy revealed extensive obstruction of both the main and lobar bronchus with yellowish nodules strongly adhered to the bronchial wall; both histologic examination and culture of these nodules revealed Aspergillus fumigatus. Even with early detection of an intraluminal growth of Aspergillus and prompt institution of antifungal therapy, the patient died of refractory hypoxemia a few days later. This report shows that Aspergillus tracheobronchitis should be considered in immunocompromised patients with suspected lung infection even when the main radiographic finding is atelectasis.

Tracheobronchial manifestations of Aspergillus infections.

Human lungs are constantly exposed to a large number of Aspergillus spores which are present in ambient air. These spores are usually harmless to immunocompetent subjects but can produce a symptomatic disease in patients with impaired antifungal defense. In a small percentage of patients, the trachea and bronchi may be the main or even the sole site of Aspergillus infection. The clinical entities that may develop in tracheobronchial location include saprophytic, allergic and invasive diseases. Although this review is focused on invasive Aspergillus tracheobronchial infections, some aspects of allergic and saprophytic tracheobronchial diseases are also discussed in order to present the whole spectrum of tracheobronchial aspergillosis. To be consistent with clinical practice, an approach basing on specific conditions predisposing to invasive Aspergillus tracheobronchial infections is used to present the differences in the clinical course and prognosis of these infections. Thus, invasive or potentially invasive Aspergillus airway diseases are discussed separately in three groups of patients: (1) lung transplant recipients, (2) highly immunocompromised patients with hematologic malignancies and/or patients undergoing hematopoietic stem cell transplantation, and (3) the remaining, less severely immunocompromised patients or even immunocompetent subjects.