Respirology Case Reports最新文献

Pulmonary vein stenosis-a complication of atrial fibrillation ablation unfamiliar to most pulmonologists-was identified in a 42-year-old man who presented with haemoptysis. Initial bronchoscopy at a previously visited hospital revealed a central airway lesion, but biopsy was declined because of its haemorrhagic nature. Repeat bronchoscopy at our hospital showed oedematous changes and vascular proliferation in the left main bronchus, suggesting adenoid cystic carcinoma. However, cryobiopsy excluded malignancy and suggested a vascular malformation with thickened, irregularly dilated muscular vessels. Contrast-enhanced CT revealed occlusion of the left superior pulmonary vein, interpreted as a secondary change. The patient had a history of catheter ablation for atrial fibrillation. Based on these findings, the stenosis was determined to be a complication of catheter ablation, and surgical bypass was performed. Severe pulmonary vein stenosis was successfully diagnosed using cryobiopsy. This condition is unfamiliar to pulmonologists; however, differentiation from airway tumours is crucial for determining treatment.

Extraskeletal Ewing sarcoma (EES) is a rare malignant tumour within the "Ewing family of tumours," first described by Tefft et al. in 1969. It accounts for less than 1 case per million, typically affecting adolescents and young adults. We report a rare case of pleural EES in a 42-year-old woman presenting with chest pain and low-grade fever persisting for one month. Physical examination showed decreased breath sounds in the right lung base on auscultation. Ultrasound demonstrated right-sided pleural effusion, an unusual manifestation of EES that can hinder accurate diagnosis. CT scans identified multiple pulmonary nodules, further confirmed by thoracoscopy. Biopsy established the diagnosis of EES. The patient was subsequently treated with multi-agent VAC/IE chemotherapy. Despite initiation of systemic therapy, the disease followed an aggressive course, and the patient succumbed during follow-up. We also review existing literature to highlight clinical, pathological, and radiological features of pleural EES, aiming to expand knowledge of this rare presentation.

[This corrects the article DOI: 10.1002/rcr2.70435.].

We present a case of active interstitial lung disease (ILD) preceding the onset of rheumatoid arthritis (RA). A 68-year-old man had dyspnea, decreased forced vital capacity, and elevated Krebs von den Lungen-6 levels. Non-specific interstitial pneumonia-like patterns, including ground-glass opacities, fibrosis, and subpleural spared area, were observed on computed tomography. Both rheumatoid factor and anti-cyclic citrullinated peptide antibody were positive; however, he had no clinical evidence of arthritis. Two years later, he developed arthritis, which was diagnosed as RA with high disease activity. He was started on salazosulfapyridine, which had no effect on the arthritis. Filgotinib, a selective Janus kinase (JAK) 1 inhibitor, was added for arthritis treatment; thereafter, the ILD improved alongside the arthritis. He maintained remission for 4 years with no exacerbation, flare, or adverse events. JAK1 inhibitor may be safe and beneficial in the long-term control and treatment of arthritis and ILD.

A 15-year-old autistic boy with rumination syndrome presented with progressive oesophageal dilation on CT. Achalasia was excluded based on imaging. This case illustrates a rare example of structural change likely due to behavioural disorder and highlights the diagnostic value of CT when manometry is unfeasible.

Silicone implants can trigger adjuvant-induced autoimmune/inflammatory syndrome (ASIA). We present a 53-year-old woman who had undergone bilateral intramuscular silicone breast implantation at X-24 years. Following implant rupture at X year, she developed cough, fatigue, and arthralgia, accompanied by worsening sarcoid-like pulmonary lesions. After implant removal, her clinical symptoms and radiological findings improved. Histopathological examination findings identified foreign-body granulomas, fulfilling diagnostic criteria for ASIA. This case suggests that silicone leakage after implant rupture, rather than the duration of exposure, is a critical factor in disease development and progression. The sarcoid-like pulmonary lesions in this case may represent a sarcoid-like granulomatous reaction triggered by silicone exposure in the setting of ASIA, rather than true systemic sarcoidosis. Early implant removal appears beneficial for both the diagnosis and treatment of ASIA.

Percutaneous drainage can be used to treat infected pulmonary bullae unresponsive to antibiotics; however, it carries a high risk of bronchopleural fistula and empyema. We report the case of an elderly man with an infected pulmonary bulla successfully treated with percutaneous drainage followed by open-window thoracostomy (OWT) for persistent air leakage. Although the infection was controlled, closing the OWT was challenging due to recurrent bronchial fistulae despite repeated surgeries. This case highlights the need to carefully identify and close all bronchial openings in the cyst wall to achieve successful closure after OWT in patients with severely infected bullae.

An 81-year-old ex-smoker with chronic cough and weight loss had imaging showing right upper-lobe nodules, tree-in-bud changes and bronchial obstruction. Bronchoscopy revealed a right upper lobe bronchus polypoidal mass with histological evidence of actinomycosis. This case highlights actinomycosis mimicking malignancy and the need to exclude wider differentials and cancer mimics.

Whipple's disease (WD) is a rare chronic multisystem infectious disease caused by the actinomycete Tropheryma whipplei. Pulmonary involvement is uncommon, and its clinical manifestations lack specificity, with diverse imaging findings, making it prone to misdiagnosis. We report a rare case of a 50-year-old woman who presented with a 2-week history of cough. Chest CT showed multiple thick-walled cavities in both lungs, a highly unusual presentation for WD pneumonia. Routine microbiological tests, including acid-fast staining and culture of bronchoalveolar lavage fluid (BALF), were negative, which made the diagnosis challenging. Metagenomic next-generation sequencing (mNGS) of BALF detected T. whipplei, confirming the diagnosis of WD pneumonia. After oral doxycycline treatment, follow-up chest CT showed complete resolution of the pulmonary cavities. This case demonstrates that multiple thick-walled cavities may be a characteristic imaging manifestation of WD pneumonia, highlights the diagnostic value of mNGS for this rare infection, and supports oral doxycycline monotherapy as an effective treatment option for isolated pulmonary TW infection.

Bronchial foreign body aspiration (BFBA) in adults is uncommon and frequently overlooked, particularly in tuberculosis-endemic settings where it may mimic asthma or pulmonary tuberculosis, leading to prolonged diagnostic delay. We report a 55-year-old woman with a two-year history of treatment-refractory wheezing and progressive dyspnea, repeatedly misdiagnosed as asthma. Chest computed tomography revealed a smooth intraluminal lesion within the right basal trunk bronchus with surrounding post-obstructive inflammatory changes but no distal atelectasis. Flexible bronchoscopy identified and successfully removed an impacted sapodilla seed, resulting in immediate resolution of wheezing and complete radiologic recovery at follow-up. This case highlights the importance of meticulous clinical examination, recognition of atypical features of adult-onset wheezing, and early use of chest CT to identify endobronchial obstruction. Timely bronchoscopy remains the definitive diagnostic and therapeutic modality, preventing unnecessary prolonged treatment and irreversible pulmonary sequelae.