{"title":"Allergic bronchopulmonary mycosis induced by pembrolizumab for bladder cancer: A case report","authors":"Hiroki Iida , Kenichiro Atsumi , Naohiro Kadoma , Sae Takashima , Yukari Shirakura , Ayana Suzuki , Kakeru Hisakane , Ryo Matsuoka , Koji Nagata , Masahiro Seike , Takashi Hirose","doi":"10.1016/j.rmcr.2025.102179","DOIUrl":"10.1016/j.rmcr.2025.102179","url":null,"abstract":"<div><div>Pembrolizumab is an immune checkpoint inhibitor (ICI) of programmed cell death-1 category, used for treating various types of cancer. ICIs can sometimes result in immune-related adverse events. Allergic bronchopulmonary mycosis (ABPM) induced by ICI has rarely been reported. We hereby report the case of an 83-year-old woman who experienced non-Aspergillus ABPM with eosinophilia induced by pembrolizumab that had been prescribed for treating bladder cancer. Steroid monotherapy with prednisone was successful and pembrolizumab could be resumed. Through the present case report, we urge the clinicians to be aware of the potential risk of ABPM as a T-helper type 2-associated immune-related adverse event.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"54 ","pages":"Article 102179"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143396078","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Effective management of refractory chylothorax associated with elderly onset Gorham-Stout disease: A case report","authors":"Haruna Yamaki , Masaru Ejima , Takaya Takeguchi , Chihiro Kagohashi , Shunya Hanawa , Natsushi Kubota , Kotaro Hanawa , Seishi Higashi , Satoko Hanada , Reiko Taki","doi":"10.1016/j.rmcr.2025.102195","DOIUrl":"10.1016/j.rmcr.2025.102195","url":null,"abstract":"<div><div>Gorham-Stout disease (GSD) is a rare vascular disease of lymphatic origin characterized by progressive osteolysis that commonly causes chylothorax owing to the leakage of lymphatic fluid from dissolved bones. We report a case of refractory chylothorax that was diagnosed as elderly onset GSD and treated successfully using multidisciplinary approaches. A 78-year-old male presented with persistent cough and shortness of breath caused by massive left pleural effusion. Thoracentesis and pleural biopsy revealed an initial diagnosis of idiopathic chylothorax. The patient underwent continuous thoracic drainage and several percutaneous thoracic duct embolization with subcutaneous octreotide acetate injection and a low-fat diet. Thoracoscopic surgery was performed to control the secondary empyema through the drain tube and repair the pleural lesions with lymphatic leakage. A sufficient decrease in pleural fluid volume allowed temporary removal of the thoracic drainage tube. However, thoracic drainage was resumed for refractory chylothorax when the patient gradually developed neck pain after three months. Computed tomography, magnetic resonance imaging, and bone scintigraphy revealed growing osteolytic lesions in the cervical and thoracic spine regions, and biopsy demonstrated numerous dilated thin-walled capillary structures with lymphocyte infiltrates. After diagnosing GSD and introducing sirolimus, the patient's symptoms improved, and thoracic drainage was discontinued. Multidisciplinary treatments successfully halted disease progression for over a year. The case report emphasizes the systematic examination to identify rare etiology in refractory chylothorax treatment.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102195"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143685859","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Amal Al-Naimi , Sara G. Hamad , Abdalla E. Zarroug
{"title":"Congenital cervical lung herniation in an infant with arterial tortuosity syndrome","authors":"Amal Al-Naimi , Sara G. Hamad , Abdalla E. Zarroug","doi":"10.1016/j.rmcr.2025.102193","DOIUrl":"10.1016/j.rmcr.2025.102193","url":null,"abstract":"<div><div>Arterial Tortuosity Syndrome (ATS) is a rare inherited connective tissue disorder that is characterized by elongated tortuous large and medium-sized arteries. ATS, as a multi-systemic disease, manifests with skin laxity, joint hypermobility and predisposition to hernia formation. Inguinal and diaphragmatic hernias were reported as the most common types of herniations in children with ATS.</div><div>However, no previous reports have documented an association with congenital cervical lung herniation in those children. We present a case of congenital cervical lung herniation in an infant with ATS that was resolved clinically and radiologically at the age of 43 months with conservative management.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"54 ","pages":"Article 102193"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143637251","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Two fatal cases of anti-melanoma differentiation-associated gene 5 antibody (anti-MDA5) rapidly progressive interstitial lung disease (RP-ILD): Exploring pitfalls and differences","authors":"Dina Ismail , Fatema Ezzy , Bibi Ayesha","doi":"10.1016/j.rmcr.2025.102224","DOIUrl":"10.1016/j.rmcr.2025.102224","url":null,"abstract":"<div><h3>Introduction</h3><div>Rapidly progressive interstitial lung disease (RP-ILD) poses a critical challenge in clinical practice, characterized by nonspecific symptoms that rapidly progress to respiratory failure despite intensive management. Anti-melanoma differentiation-associated gene 5 antibody (anti-MDA5), associated with dermatomyositis-related ILD, is a key biomarker in such cases. This report highlights two fatal cases of anti-MDA5-positive RP-ILD, emphasizing diagnostic and management challenges.</div></div><div><h3>Case presentation</h3><div>The first case involved a 56-year-old male without prior autoimmune disease who developed acute respiratory distress syndrome (ARDS) and multiorgan failure. The second case concerned a 49-year-old female with systemic lupus erythematosus (SLE), whose condition similarly culminated in multiorgan failure despite aggressive treatment. Both patients faced delayed diagnoses and suboptimal responses to immunosuppressive therapy, underscoring the complexity of managing RP-ILD.</div></div><div><h3>Discussion</h3><div>Prolonged testing turnaround for MDA5 and reliance on nonspecific clinical indicators remain significant barriers to early diagnosis. Dermatological signs such as Gottron's papules, while indicative of RP-ILD, are associated with poorer prognoses. Current serum biomarkers under investigation lack the sensitivity and specificity of MDA5, complicating early detection efforts. Aggressive immunosuppression in deteriorating patients frequently leads to infectious complications, further compromising outcomes.</div></div><div><h3>Conclusion</h3><div>To address these challenges, early recognition of dermatological indicators, prompt immunosuppressive therapy, and tailored patient management are critical. Additionally, timely referral for extracorporeal membrane oxygenation (ECMO) as a bridge to lung transplantation may improve survival. Advancing research into serum biomarkers holds promise for enhancing diagnostic precision and therapeutic decision-making in RP-ILD.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102224"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143878836","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Marc Assaad, Roshan Acharya, Elspeth Springsted, Frank Biscardi
{"title":"A rare presentation of pulmonary transthyretin amyloidosis","authors":"Marc Assaad, Roshan Acharya, Elspeth Springsted, Frank Biscardi","doi":"10.1016/j.rmcr.2025.102189","DOIUrl":"10.1016/j.rmcr.2025.102189","url":null,"abstract":"<div><div>Virchow described amyloidosis in 1853. Amyloidosis is the extracellular deposition of an insoluble misfolded polymerized tetramer of fibrillary protein that accumulates within tissues leading to organ dysfunction. Amyloidosis affects 10 people per one million every year. Specific varieties of amyloidosis result from diversely involved proteins. Tissue involvement can lead to multiorgan system dysfunction. Lung involvement from amyloidosis is very rare, and tissue biopsy is crucial for diagnosis. Genetic testing might be requested based on the suspected type. Lung involvement is very rare and mainly asymptomatic, and some cases are only diagnosed postmortem. When symptomatic, the clinical manifestations are nonspecific and include cough, dyspnea, and respiratory infections. The infiltrative process might affect pulmonary mechanics producing abnormal pulmonary function testing. Radiological manifestations of pulmonary amyloidosis involve the pleura, the tracheobronchial tree, the alveoli, and the mediastinal lymph nodes. Treatment of pulmonary amyloidosis is based on treating the underlying disease process. We present herein a case of 78-year-old male with senile cardiac amyloidosis, who presents to our clinic with multiple nodular pulmonary opacities.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"54 ","pages":"Article 102189"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143620162","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Remarkable response to crizotinib in a patient with advanced lung adenocarcinoma harboring the MPRIP-ROS1 fusion gene: A case report","authors":"Yasuyuki Kishikawa, Kohei Otsubo, Daisuke Shibahara, Yoshimasa Shiraishi, Yasuto Yoneshima, Eiji Iwama, Isamu Okamoto","doi":"10.1016/j.rmcr.2025.102243","DOIUrl":"10.1016/j.rmcr.2025.102243","url":null,"abstract":"<div><div>For patients with advanced non-small cell lung cancer (NSCLC), genetic testing is crucial to identify alterations in targetable driver genes. ROS1-tyrosine kinase inhibitors have shown efficacy against NSCLC with common <em>ROS1</em> fusion genes, but the impact of rare fusion partners on therapeutic outcomes is not well understood. Here, we describe a 75-year-old female with advanced lung adenocarcinoma who was treated with crizotinib after the identification of the extremely rare <em>MPRIP-ROS1</em> fusion. Despite stepwise dose reductions due to adverse effects, the patient exhibited a significant tumor response to crizotinib. The sustained response, even at reduced doses, highlights the potential for targeted therapies in managing NSCLC with <em>MPRIP-ROS1</em> fusion. This case also underscores the importance of comprehensive genomic profiling using hybrid capture-based next-generation sequencing to identify rare driver gene alterations that may not be detected by conventional target sequencing-based methods.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"56 ","pages":"Article 102243"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144489453","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"A case of lung adenocarcinoma with EGFR exon 19 deletion/insertion mutation (T751_I759delinsS) showing response to Osimertinib","authors":"Kazuhisa Nakashima, Seiko Tanaka, Mika Nakao, Akari Tanino, Yoshihiro Amano, Tamio Okimoto, Takeshi Isobe","doi":"10.1016/j.rmcr.2025.102251","DOIUrl":"10.1016/j.rmcr.2025.102251","url":null,"abstract":"<div><div>Epidermal growth factor receptor (EGFR) tyrosine kinase inhibitors are effective for treating EGFR mutation-positive non-small cell lung cancer. However, the diversity of EGFR mutations presents a challenge, as treatment strategies for rare variants remain undefined.</div><div>A 29-year-old nonsmoking man presented to his primary physician in September 2019 with back pain. Chest computed tomography revealed multiple nodular lesionsin the right lung and pleura, leading to a diagnosis of lung adenocarcinoma originating in the right middle lobe (cT4N1M1a, stage IVA). Initial testing with the Cobas<sup>Ⓡ</sup> EGFR Mutation Detection Kit v2.0, using real-time polymerase chain reaction (PCR), yielded negative results for EGFR mutations. The patient was subsequently referred to our hospital for further treatment. In January 2020, he began combination therapy with atezolizumab, bevacizumab, carboplatin, and paclitaxel. Additionally, a bronchoscopy was conducted at our hospital to identify potential undetected driver gene mutations. Next-generation sequencing (NGS) analysis, performed as part of a clinical trial, revealed the presence of pT751_I759 delinsS, a rare EGFR exon 19 deletion–insertion mutation variant. The companion diagnostic test confirmed the mutation through re-examination using the peptide nucleic acid-locked nucleic acid PCR-clamp method. After the previous treatment regimen lost efficacy, osimertinib was initiated in April 2021. Tumor shrinkage was observed, and the treatment was sustained for 11 months.</div><div>This case involved a young patient diagnosed with lung adenocarcinoma. Given the clinical presentation, a driver gene mutation was strongly suspected. NGS identified the rare mutation pT751_I759delinsS, and the findings suggested the potential efficacy of osimertinib for this variant.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102251"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144535045","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Bronchial pleomorphic adenoma successfully diagnosed and resected with left lower sleeve lobectomy; a case report and literature review","authors":"Katsuhiro Itogawa , Tomohiro Oba , Mitsuru Maki , Masako Amano , Akiko Adachi , Hidekazu Matsushima","doi":"10.1016/j.rmcr.2025.102253","DOIUrl":"10.1016/j.rmcr.2025.102253","url":null,"abstract":"<div><div>Tracheobronchial tumor is a relatively uncommon type of respiratory tumor. Seromucous gland tumors, such as adenoid cystic carcinoma, mucoepidermoid carcinoma, and pleomorphic adenoma, are a part of this category. Among them, pleomorphic adenoma is especially rare. This report describes a case of bronchial pleomorphic adenoma successfully diagnosed and removed with surgery. A 70-year-old Japanese man presented with abnormal chest shadow and intermittent fever. A subsequently conducted chest computed tomography revealed a 40mm-sized mass in the left lower bronchus and distal obstructive pneumonia. Because a transbronchial biopsy was not diagnostic, a left lower sleeve lobectomy was performed. As a result, the mass was completely resected and a diagnosis of pleomorphic adenoma was made. Treatment options for pleomorphic adenoma include surgery and endoscopic resection, because observation is sometimes inadvisable due to the risk of metastasis or malignant transformation. Furthermore, complete resection is diagnostically appropriate because pleomorphic adenoma is sometimes undiagnosed or misdiagnosed with partially resected or biopsied specimen, due to the heterogeneity in pathology. This case underscores the challenges in diagnosing pleomorphic adenoma and highlights the importance of complete surgical resection for definitive diagnosis and treatment.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102253"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144569887","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"IgG4-related pleuritis diagnosed by local anesthesia thoracoscopy; a case report and literature review","authors":"Genki Inui , Tomoya Harada , Karen Makishima , Aditya Sri Listyoko , Shunsuke Ohga , Miyu Nishigami , Hiroyuki Tanaka , Hiroki Ishikawa , Hiroki Kohno , Yoshihiro Funaki , Miki Takata , Ryota Okazaki , Masato Morita , Masahiro Kodani , Akira Yamasaki","doi":"10.1016/j.rmcr.2025.102235","DOIUrl":"10.1016/j.rmcr.2025.102235","url":null,"abstract":"<div><div>IgG4-related pleuritis is rare in patients with IgG4-related diseases (IgG4RD). We report a case of IgG4-related pleuritis diagnosed in a 77-year-old Japanese man with right pleural effusion. The pleural effusion exhibited lymphocyte-predominant exudates with elevated adenosine deaminase (ADA) and IgG4 levels, along with the presence of plasma cells. A pleural biopsy via local anesthesia thoracoscopy (LAT) confirmed the diagnosis of IgG4-related pleuritis, and the patient was treated with prednisolone and azathioprine. Although ADA is a well-known useful marker for diagnosing tuberculous pleuritis, a review of 14 reported cases of IgG4-related pleuritis demonstrated a positive correlation between IgG4 and ADA in pleural effusion (ρ = 0.705, p < 0.05), suggesting that ADA levels could be elevated in IgG4-related pleuritis. Furthermore, all cases with available cytology reports showed the presence of plasma cells, indicating that detecting plasma cells could aid in diagnosis. Pleural biopsy remains the gold standard for the diagnosis of IgG4-related pleuritis. LAT is a safe and effective diagnostic procedure for older patients, enabling direct visualization and biopsy of pleural lesions without intubation. IgG4-related pleuritis presents with specific findings, including dense white granulomatous lesions, vesicular nodular changes, nonspecific inflammatory changes, and pleural thickening. IgG4-positive plasma cells can be detected even in biopsies with diffuse, nonspecific findings. Therefore, LAT is a valuable and safe tool for diagnosing IgG4-related pleuritis. In conclusion, exudative pleural effusion with elevated ADA levels should prompt the consideration of IgG4-related pleuritis in the differential diagnosis. LAT is a minimally invasive and highly accurate diagnostic tool for IgG4-related pleuritis.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"56 ","pages":"Article 102235"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144108127","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Enfortumab vedotin induced interstitial lung disease: A first case report with pathological evidence from transbronchial lung cryobiopsy","authors":"Shota Kaburaki , Toru Tanaka , Koichiro Kamio , Yosuke Tanaka , Kazuo Kasahara , Yasuhiro Terasaki , Masahiro Seike","doi":"10.1016/j.rmcr.2025.102237","DOIUrl":"10.1016/j.rmcr.2025.102237","url":null,"abstract":"<div><div>Enfortumab vedotin (EV), an antibody–drug conjugate targeting Nectin-4, has demonstrated efficacy against advanced urothelial carcinoma. While initially considered rare, EV-induced interstitial lung disease (ILD) is increasingly recognized, yet its pathological features remain poorly characterized. We report a case of a 60-year-old man with metastatic urothelial carcinoma who developed fever, fatigue, and cough after two cycles of EV therapy. His treatment history included right nephroureterectomy, platinum-based chemotherapy, and immune checkpoint inhibitors nivolumab and pembrolizumab. Laboratory tests revealed elevated serum ILD markers (Krebs von den Lungen-6527.7 U/mL, surfactant protein-D 294.5 ng/mL), and chest computed tomography showed new infiltrative shadows with air bronchogram predominantly in subpleural regions of the right lower lobe, consistent with organizing pneumonia pattern. Bronchoalveolar lavage from the right middle lobe showed 92 % macrophages with negative cultures. Transbronchial lung cryobiopsy revealed fibrosing nonspecific interstitial pneumonia with prominent fibrosis around bronchovascular bundles, lymphocytic infiltration in vessel walls and alveolar septa with myxofibrous thickening, epithelial injury, and fibrin exudation into alveolar spaces—representing previously undocumented features of EV-induced ILD. Drug discontinuation alone proved insufficient, but the patient improved markedly with methylprednisolone pulse therapy. This case highlights two key findings: detailed histopathological characterization through cryobiopsy documents distinct pathological features of EV-induced ILD; and early bronchoscopic evaluation helped guide therapeutic decision-making, supporting aggressive corticosteroid therapy. These findings advance our understanding of both the pathological features and management of EV-induced ILD, particularly relevant as EV–pembrolizumab combination becomes standard first-line treatment.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"56 ","pages":"Article 102237"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144115796","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}