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Diagnostically Challenging Disseminated Bacillus Calmette-Guerin Infection With Lung and Liver Involvement: A Case Report. 弥散性卡介苗芽孢杆菌感染累及肺和肝脏的诊断:1例报告。
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-18 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70448
Shinichi Chang, Makiko Yomota, Ayumi Takizawa, Noriyo Yanagawa, Tsunekazu Hishima, Naoko Kubota, Yukio Hosomi

Intravesical Bacillus Calmette-Guerin (BCG) therapy is the standard treatment for non-muscle-invasive bladder cancer. While it is effective, it sometimes causes a disseminated BCG infection, which is often difficult to diagnose microbiologically. We report herein a 73-year-old, male patient in whom a persistent fever, dyspnoea, and jaundice developed after he received his tenth round of intravesical BCG therapy. Chest computed tomography revealed diffuse micronodules, and lung and liver biopsies demonstrated multiple, well-formed, non-caseating granulomas. Despite negative microbiological test results, a disseminated BCG infection was suspected. Antimycobacterial therapy was started but discontinued after 2 weeks due to a drug-induced eruption. The clinical and radiological findings continued to improve without further treatment. The present case highlights the importance of a detailed assessment of radiological and histopathological findings for the diagnosis and management of diffuse lung diseases, including complications related to intravesical BCG therapy.

膀胱内卡介苗治疗是非肌肉浸润性膀胱癌的标准治疗方法。虽然它是有效的,但有时会引起播散性卡介苗感染,这通常很难从微生物学上诊断出来。我们在此报告一位73岁男性患者,他在接受第10轮膀胱内卡介菌治疗后出现持续发烧、呼吸困难和黄疸。胸部计算机断层扫描显示弥漫性微结节,肺和肝活检显示多发形态良好的非干酪化肉芽肿。尽管微生物检测结果阴性,但仍怀疑播散性卡介苗感染。开始抗真菌治疗,但在2周后因药物引起的皮疹而停止治疗。临床和放射学表现在没有进一步治疗的情况下继续改善。本病例强调了详细评估放射学和组织病理学结果对弥漫性肺部疾病的诊断和管理的重要性,包括膀胱内卡介苗治疗相关的并发症。
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引用次数: 0
Pulmonary Hamartoma Initially Misdiagnosed as Adenocarcinoma: A Case Report and Review of the Literature. 肺错构瘤最初误诊为腺癌:1例报告及文献复习。
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-17 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70445
Erfaneh Hosseini, Maryam Mazraehei Farahani, Ramin Sami, Mohammad Behgam Shadmehr

Pulmonary hamartomas are common benign lung tumours that rarely present with hemoptysis and may mimic malignancy. We report a 39-year-old non-smoker with 2 months of hemoptysis and a 3-cm right upper-lobe lesion. CT-guided core biopsy with IHC suggested adenocarcinoma (CK7, TTF-1, Napsin-A positive), but PET-CT showed no metastasis. The patient underwent VATS resection and final histopathology confirmed pulmonary hamartoma. Symptoms resolved postoperatively with no recurrence at 6-month follow-up. This case highlights potential false-positive biopsy/IHC results from entrapped benign epithelium and the role of surgical excision for definitive diagnosis.

肺错构瘤是常见的良性肺肿瘤,很少表现为咯血,可能与恶性肿瘤相似。我们报告一位39岁非吸烟者,咯血2个月,右上叶病变3厘米。ct引导下IHC核心活检提示腺癌(CK7, TTF-1, Napsin-A阳性),但PET-CT未显示转移。患者行VATS切除术,最终病理证实为肺错构瘤。术后症状消失,随访6个月无复发。本病例强调了良性上皮夹闭的潜在假阳性活检/免疫组化结果和手术切除的最终诊断作用。
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引用次数: 0
Delayed Massive Haemoptysis Following EBUS-TBNA Revealing a Pseudoaneurysm of an Ectopic Bronchial Artery: A Rare Complication at the Crossroads of Interventional Pulmonology and Radiology. EBUS-TBNA后迟发性大咯血显示异位支气管动脉假性动脉瘤:介入肺科与放射学交叉的罕见并发症。
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-17 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70446
Julien G Cohen, Matthieu Papillard, Romain Messe

Endobronchial ultrasound-guided transbronchial needle aspiration (EBUS-TBNA) is widely considered a safe and minimally invasive procedure for the evaluation of mediastinal lymphadenopathy. Although complications are rare, severe haemorrhagic events may occur, particularly when vascular structures are punctured. A 31-year-old woman with Budd-Chiari syndrome underwent EBUS-TBNA for hypermetabolic mediastinal lymph nodes. Sampling was uneventful, but she developed massive haemoptysis 7 days later. Contrast-enhanced CT and bronchoscopy identified two 4R lymph node pseudoaneurysms, confirmed by angiography. The lesion was successfully treated with selective microcoil embolisation. No recurrence was observed at two-month follow-up, despite resuming anticoagulation. Delayed pseudoaneurysm is an exceptionally rare but potentially life-threatening complication of EBUS-TBNA. Pre-procedural Doppler assessment, awareness of vascular variants, and timely imaging are essential. Endovascular embolisation is an effective, minimally invasive treatment. Early recognition and multidisciplinary management are crucial to prevent catastrophic outcomes.

超声引导下的支气管穿刺(EBUS-TBNA)被广泛认为是一种安全、微创的评估纵隔淋巴结病的方法。虽然并发症是罕见的,严重的出血事件可能发生,特别是当血管结构被刺穿。31岁女性Budd-Chiari综合征患者因高代谢纵隔淋巴结接受EBUS-TBNA治疗。采样正常,但7天后出现大量咯血。增强CT和支气管镜检查发现两个4R淋巴结假性动脉瘤,经血管造影证实。选择性微线圈栓塞成功治疗病变。尽管恢复抗凝治疗,但随访2个月未见复发。迟发性假性动脉瘤是EBUS-TBNA异常罕见但可能危及生命的并发症。术前多普勒评估、血管变异意识和及时成像是必不可少的。血管内栓塞是一种有效的微创治疗方法。早期识别和多学科管理对于防止灾难性后果至关重要。
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引用次数: 0
Economic Impact of Biologics for Severe Asthma: A Case Series of 14 Patients From Northern Malaysia. 生物制剂对严重哮喘的经济影响:马来西亚北部14例患者的病例系列。
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-17 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70447
Arvindran Alaga, Izzatul Nadzirah Ismail

There remains a paucity of evidence from Asia on the cost-effectiveness of biologics for severe asthma. This pilot cost-offset analysis aims to evaluate the economic impact of adding biologics (omalizumab or benralizumab) to severe asthma standard care as a proof of concept for its cost-effectiveness, and to assess improvement in exacerbation rates and asthma control. Fourteen patients from the Severe Asthma Registry of Hospital Sultanah Bahiyah, Kedah who received biologics for at least 12 months from January 2018 to December 2024 were included. The economic impact of biologic treatment was evaluated by comparing the cost of biologic administration with the potential savings from reduced exacerbations requiring emergency department visits and hospital admission. Cost savings were observed with biologics, alongside reduced exacerbation and improved asthma control in real-world settings. Future research could explore a full cost-effectiveness analysis and a budget impact analysis to inform strategic pricing and reimbursement decisions.

来自亚洲的关于生物制剂治疗严重哮喘的成本效益的证据仍然缺乏。该试点成本抵消分析旨在评估在重度哮喘标准治疗中添加生物制剂(omalizumab或benralizumab)的经济影响,作为其成本效益的概念证明,并评估恶化率和哮喘控制的改善。来自吉打州Sultanah Bahiyah医院严重哮喘登记处的14名患者在2018年1月至2024年12月期间接受了至少12个月的生物制剂。生物治疗的经济影响是通过比较生物治疗的成本与减少需要急诊科就诊和住院的恶化所节省的潜在成本来评估的。在现实环境中,生物制剂可以节省成本,同时减少急性发作和改善哮喘控制。未来的研究可以探索全面的成本效益分析和预算影响分析,以告知战略定价和报销决策。
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引用次数: 0
Endobronchial Watanabe Spigot for Acute Empyema With Bronchopleural Fistula: A Case Series. 支气管内Watanabe龙头治疗急性肺气肿合并支气管胸膜瘘:一个病例系列。
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-16 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70444
Kento Yokota, Takayuki Niitsu, Satoshi Tanaka, Daiki Nagira, Saki Nishibeppu, Naoya Takada, Satoshi Tobita, Moto Yaga, Kiyonobu Ueno, Yasunobu Funakoshi

Empyema with bronchopleural fistula (BPF), defined by persistent air leak with pleural infection, undermines negative-pressure drainage and often leads to highly invasive procedures such as fenestration. While endobronchial Watanabe spigots (EWSs) have mainly been used for postoperative or chronic fistulas, their role as an early intervention in acute infectious empyema with BPF remains uncertain. We report three consecutive frail patients with acute empyema and persistent air leak due to BPF treated using a prospectively predefined early EWS approach. After adequate drainage and antibiotics failed to seal the fistula, endobronchial occlusion was performed at the earliest bronchoscopic opportunity before considering fenestration. In all cases, EWS placement achieved complete cessation of air leak, allowed chest tube removal within 6-26 days and obviated fenestration. Diverse microbiology, including polymicrobial infection and nontuberculous mycobacteria, was controlled without observed device-related infection or migration. Early EWS may offer a minimally invasive, fenestration-sparing option in selected patients.

脓胸合并支气管胸膜瘘(BPF),定义为持续的空气泄漏并胸膜感染,破坏负压引流,通常导致高侵入性手术,如开窗。虽然支气管内Watanabe导管(ews)主要用于术后或慢性瘘管,但其在急性感染性脓胸合并BPF的早期干预中的作用仍不确定。我们报告了三例连续体弱的患者,由于BPF导致急性脓胸和持续漏气,使用前瞻性预先定义的早期EWS方法治疗。在充分引流和抗生素未能封闭瘘管后,在考虑开窗之前,在最早的支气管镜检查机会进行支气管内闭塞。在所有病例中,EWS放置完全停止了空气泄漏,允许在6-26天内取出胸管,并避免了开窗。多种微生物,包括多微生物感染和非结核分枝杆菌,都得到了控制,没有观察到与器械相关的感染或迁移。早期EWS可以为特定患者提供微创、保留开窗的选择。
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引用次数: 0
Unexpected Response to Nab-Paclitaxel in Poor Performance Status Lung Adenocarcinoma With a Unique 'Pan-Positive' Tumour Marker Profile: Case Report. nab -紫杉醇在表现不佳的肺腺癌中具有独特的“泛阳性”肿瘤标志物的意外反应:病例报告
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-15 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70442
Masato Shimozono, Takafumi Kato, Nobuharu Ohshima, Masashi Kitani, Masaomi Maeda, Sumie Nakamura, Kei Kusaka, Masahiro Shimada, Atsuhisa Tamura

Patients with advanced non-small cell lung cancer and poor performance status (PS) are often excluded from chemotherapy. We present a 63-year-old male with metastatic, poorly differentiated lung adenocarcinoma, ECOG PS 3, and an exceptionally broad, markedly elevated panel of tumour markers. He tolerated initial single-agent nab-paclitaxel with significant clinical and serological improvement, allowing treatment escalation. This report details the dynamic, often discordant, changes in this marker panel throughout treatment. This case suggests that carefully selected patients with poor PS may derive clinical benefit from chemotherapy and that monitoring a marker profile may provide insights into mechanisms of therapeutic efficacy and resistance.

晚期非小细胞肺癌和不良表现状态(PS)的患者通常被排除在化疗之外。我们报告一例63岁男性转移性低分化肺腺癌,ECOG ps3,肿瘤标记物异常广泛,显著升高。他最初耐受单药nab-紫杉醇,临床和血清学改善显著,允许治疗升级。本报告详细描述了在整个治疗过程中该标志物组的动态变化,这些变化通常是不一致的。该病例表明,精心挑选的不良PS患者可能从化疗中获得临床益处,监测标志物谱可能提供治疗效果和耐药性机制的见解。
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引用次数: 0
Interesting Case of Mesocardia: A Rare Cardiac Positional Anomaly. 有趣的中心肌炎病例:罕见的心脏位置异常。
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-11 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70437
Nasir Majeed, Harshana Bandara, Aqeel Umar, Paul Marsden

A 37-year-old man presenting with chronic cough was incidentally found to have mesocardia on chest imaging, with a midline heart and associated upper thoracic scoliosis. Further tests revealed eosinophilic asthma, treated successfully. This rare, isolated mesocardia case highlights the importance of recognising abnormal cardiac positioning on routine radiographs.

一名37岁男性,慢性咳嗽,偶然发现胸片上有中心肌病,心脏中线和相关的上胸椎脊柱侧凸。进一步检查发现嗜酸性粒细胞哮喘,治疗成功。这个罕见的,孤立的心间质病例强调了在常规x线片上识别异常心脏定位的重要性。
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引用次数: 0
Beyond Cystic Fibrosis: Recognising Shwachman-Diamond Syndrome in the Respiratory Clinic. 超越囊性纤维化:识别呼吸系统临床的Shwachman-Diamond综合征。
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-10 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70440
Freda Yang, Ladina Weitnauer, Imogen Felton, Nicholas J Simmonds

Cystic fibrosis (CF) and Shwachman-Diamond Syndrome (SDS) share overlapping features, including recurrent respiratory infections and pancreatic insufficiency, which can complicate diagnosis. We report a case of a six-year-old girl who presented with productive cough, steatorrhea and recurrent infections. Initial evaluation showed abnormal liver enzymes, elevated immunoreactive trypsin, neutrophil dysfunction and skeletal anomalies. Sweat test was equivocal and CFTR genetic panel was negative. Further genomic analysis identified compound heterozygous mutations in the SBDS gene: c.258+2T>C (known pathogenic variant) and c.284T>A (novel variant), confirming SDS. Over a 42-year follow-up, she experienced intermittent neutropenia, recurrent respiratory infections and pregnancy-related complications. This case emphasises the importance of considering SDS in CF-like presentations with atypical features and equivocal CF testing. Milder SDS phenotypes can survive into adulthood. Further work is needed to refine genotype-phenotype correlations and guide long-term management.

囊性纤维化(CF)和Shwachman-Diamond综合征(SDS)具有重叠特征,包括反复呼吸道感染和胰腺功能不全,这可能使诊断复杂化。我们报告一个六岁的女孩谁提出生产性咳嗽,脂肪漏和反复感染的情况。初步评估显示肝酶异常,免疫反应性胰蛋白酶升高,中性粒细胞功能障碍和骨骼异常。汗液试验结果不明确,CFTR基因板阴性。进一步的基因组分析发现了SBDS基因的复合杂合突变:C .258+2T>C(已知致病变异)和C . 284t >A(新变异),证实了SDS。在42年的随访中,她出现了间歇性中性粒细胞减少症、复发性呼吸道感染和妊娠相关并发症。本病例强调了在非典型特征和CF检测不明确的CF样表现中考虑SDS的重要性。轻微的SDS表型可以存活到成年。需要进一步的工作来完善基因型-表型相关性并指导长期管理。
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引用次数: 0
Airway Management of Patient With Mounier-Kuhn Syndrome: A Case Report. Mounier-Kuhn综合征患者气道管理1例报告。
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-10 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70429
Ali Karami, Vida Naderi Boldaji, Fatemeh Khalili

Mounier-Kuhn syndrome (MKS) is a rare disorder characterised by tracheobronchomegaly, which poses significant anaesthetic challenges due to air leaks during mechanical ventilation. We report a 65-year-old male with previously undiagnosed MKS who underwent cardiac surgery. Following intubation with an 8-mm endotracheal tube (ETT), a persistent air leak resulted in inadequate ventilation despite tube repositioning and upsizing to a 9-mm ETT. Preoperative chest imaging revealed significant tracheal dilation. To achieve adequate ventilation, we implemented a modified technique: advancing the ETT into the right main bronchus, then retracting until bilateral breath sounds were audible, supplemented by gauze packing to reinforce the seal. This case underscores the importance of preoperative recognition of MKS to anticipate ventilation difficulties. The described technique, adapted from tracheoesophageal fistula management, effectively resolved the air leaks complication.

穆尼埃-库恩综合征(MKS)是一种罕见的以气管支气管扩张为特征的疾病,在机械通气过程中由于空气泄漏导致麻醉困难。我们报告一位65岁男性先前未确诊的MKS,他接受了心脏手术。用8mm气管内管(ETT)插管后,尽管气管重新定位并增大到9mm气管内管,但持续的空气泄漏导致通气不足。术前胸部影像学显示气管明显扩张。为了获得足够的通气,我们实施了一种改进的技术:将ETT推进至右主支气管,然后收回,直到双侧呼吸音可听到,并辅以纱布填塞以加强密封。本病例强调术前识别MKS以预测通气困难的重要性。该方法借鉴了气管食管瘘的治疗方法,有效地解决了漏气并发症。
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引用次数: 0
A Case of Right-Sided Heart Failure and Severe Pulmonary Hypertension Secondary to Partial Anomalous Pulmonary Venous Connection. 部分肺静脉连接异常继发于右侧心力衰竭和严重肺动脉高压1例。
IF 0.8 Q4 RESPIRATORY SYSTEM Pub Date : 2025-12-09 eCollection Date: 2025-12-01 DOI: 10.1002/rcr2.70438
Ahmed Tareq Alabsi, Khalifa Abdulrahman Yusuf, Alya Salman Aldoseri, Shadi Fayez Kanhosh, Abdulrahman Hasan Al-Madani, Ahmed Taha Shaarawy

Partial anomalous pulmonary venous connection (PAPVC) is a rare congenital anomaly in which one or more pulmonary veins drain into the systemic venous circulation. PAPVC is often misdiagnosed as idiopathic pulmonary hypertension in the adult population. We report a case of a 48-year-old woman with long-standing pulmonary hypertension who presented with recurrent pulmonary oedema and right-sided heart failure. Transthoracic echocardiography showed severe pulmonary hypertension with preserved left ventricular function. Computed tomographic pulmonary angiography excluded pulmonary embolism but confirmed an anomalous drainage of the right upper pulmonary vein into the superior vena cava, consistent with isolated PAPVC without an atrial septal defect. Right heart catheterization confirmed severe precapillary pulmonary hypertension with a positive vasoreactivity response to adenosine. She was successfully managed medically with diltiazem and diuretics, showing sustained clinical improvement. This case highlights isolated PAPVC as a critical, under-recognised cause of severe pulmonary hypertension in adults.

部分肺静脉连接异常(PAPVC)是一种罕见的先天性异常,其中一条或多条肺静脉流入全身静脉循环。成人PAPVC常被误诊为特发性肺动脉高压。我们报告一例48岁的妇女长期肺动脉高压谁提出了复发性肺水肿和右侧心力衰竭。经胸超声心动图显示严重肺动脉高压,左心室功能保留。计算机断层肺血管造影排除肺栓塞,但证实右上肺静脉异常引流至上腔静脉,与孤立性PAPVC一致,无房间隔缺损。右心导管检查证实严重毛细血管前肺动脉高压伴腺苷阳性血管反应。她成功地用地尔硫卓和利尿剂治疗,显示出持续的临床改善。本病例强调孤立的PAPVC是成人严重肺动脉高压的一个关键原因,但未得到充分认识。
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引用次数: 0
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Respirology Case Reports
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