Respiratory Medicine Case Reports最新文献

{"title":"Sarcomatoid Diffuse Pleural Mesothelioma in an 80-Year-Old Woman with Long-Term Response to Nivolumab Plus Ipilimumab: A Case Report","authors":"Yukiko Yoshida ,&nbsp;Hidenori Mizugaki ,&nbsp;Yuma Sato ,&nbsp;Ken Kuwahara ,&nbsp;Noriyuki Yamada ,&nbsp;Hajime Asahina ,&nbsp;Hiroshi Yokouchi ,&nbsp;Yoshihiro Matsuno ,&nbsp;Satoshi Oizumi","doi":"10.1016/j.rmcr.2025.102242","DOIUrl":"10.1016/j.rmcr.2025.102242","url":null,"abstract":"<div><div>An 80-year-old woman presented to our hospital with the chief complaint of left lateral thoracic pain. Chest computed tomography showed multiple nodular lesions in the left pleura and massive left pleural effusion. Based on clinical findings, imaging, and thoracoscopic pleural biopsy, a diagnosis of sarcomatoid diffuse pleural mesothelioma clinical T2N0M0 Stage IB was made. We administered nivolumab plus ipilimumab as first-line treatment, which resulted in significant reduction of pleural lesions and complete resolution of pleural effusion after two courses. No immune-mediated adverse events except fever were observed. Treatment was discontinued after 16 courses, with no disease recurrence for more than 2 years following the initial treatment. This case suggests that nivolumab plus ipilimumab can be an effective treatment option for older adults with pleural mesothelioma who maintain a good performance status.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"56 ","pages":"Article 102242"},"PeriodicalIF":0.8,"publicationDate":"2025-06-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144510904","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":"High-frequency jet ventilation in managing airway during whole-lung lavage under general anesthesia: A case report","authors":"Renhua Ju ,&nbsp;Xiaonan Du ,&nbsp;Ling Yin ,&nbsp;Yang Yu","doi":"10.1016/j.rmcr.2025.102206","DOIUrl":"10.1016/j.rmcr.2025.102206","url":null,"abstract":"<div><div>Pulmonary alveolar proteinosis (PAP) is a rare disorder characterized by the accumulation of surfactant-derived lipoproteins within alveoli and impaired macrophage function, leading to progressive dyspnea, hypoxemic respiratory failure, secondary infections, and pulmonary fibrosis. We report a case of a 43-year-old male with a history of occupational exposure to airborne dust from lathe work, presenting with exertional dyspnea. High-resolution computed tomography (HRCT) revealed bilateral patchy ground-glass opacities with interlobular septal thickening. Histopathological analysis of lung biopsy specimens showed eosinophilic amorphous material in alveolar spaces, which exhibited positive periodic acid-Schiff (PAS) staining with diastase resistance, confirming PAP. The patient underwent whole-lung lavage (WLL) of the right lung under general anesthesia. Severe baseline hypoxemia complicated intraoperative oxygen saturation maintenance. The intermittent use of high-frequency jet ventilation (HFJV) in the operative lung markedly improved oxygenation (SpO₂ increased from 85 % to 96 %) while ensuring effective saline distribution into distal alveoli. The procedure was completed without complications, highlighting the efficacy and safety of HFJV in managing complex airway conditions during WLL for PAP.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102206"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143850165","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":"Expanding applications of 3D-Printed stents to non-stenotic airways","authors":"Ilana Roberts Krumm ,&nbsp;Yaron B. Gesthalter","doi":"10.1016/j.rmcr.2025.102209","DOIUrl":"10.1016/j.rmcr.2025.102209","url":null,"abstract":"<div><div>Tracheobronchial stents have advanced significantly since their introduction in the 1980s, with virtual airway modeling and three-dimensional (3D) printing enabling the production of patient-specific custom airway stents. Since their FDA approval in 2019, 3D-printed custom stents have offered a promising solution for complex airway conditions. However, their use has primarily focused on tracheal stenosis and tracheobronchomalacia. The two cases presented here demonstrate novel applications of 3D printed stents in non-malignant airway diseases, specifically extrinsic vascular compression and bronchopleural fistula (BPF) from airway dehiscence.</div><div>The first case describes a 27-year-old male with tetralogy of Fallot complicated by extrinsic vascular compression of the bilateral mainstem bronchi, leading to recurrent mucus plugging, lung collapse, and respiratory failure. We designed a custom undersized Y stent to maintain airway patency while minimizing risks of vascular erosion. The second case details a 38-year-old post-lung transplant patient with a non-healing BPF with airway dehiscence and resultant respiratory failure. A bifurcated 3D-printed stent was successfully deployed to bypass the large fistula, resolving a chronic air leak.</div><div>These cases illustrate the versatility and potential of 3D-printed stents in addressing complex airway pathologies beyond tracheal stenosis or malacia and highlight critical considerations in stent design and deployment.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102209"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143834924","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":"Squamous cell carcinoma of the bronchus associated with human papillomavirus infection","authors":"Akane Toriyama ,&nbsp;Satsuki Kishikawa ,&nbsp;Shinichi Sasaki ,&nbsp;Shiaki Oh ,&nbsp;Shigeki Tomita ,&nbsp;Takashi Yao","doi":"10.1016/j.rmcr.2024.102162","DOIUrl":"10.1016/j.rmcr.2024.102162","url":null,"abstract":"<div><div>A woman in her 70s presented with an abnormal chest radiograph. Chest computed tomography showed a tumor arising from the intermediate bronchus. A bronchoscopic biopsy revealed squamous cell carcinoma, and right middle and lower bilobectomy was subsequently carried out. Histopathological examination of the resected specimen revealed squamous cell carcinoma associated with human papillomavirus infection.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"53 ","pages":"Article 102162"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11788789/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143123955","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":"A three dimensional printed endobronchial stent for the treatment of a broncho-esophageal fistula","authors":"Illaa Smesseim ,&nbsp;Sophia van Beelen ,&nbsp;Jolanda M. van Dieren ,&nbsp;Koen J. Hartemink ,&nbsp;Johanna van Sandick ,&nbsp;Jacobus A. Burgers","doi":"10.1016/j.rmcr.2025.102169","DOIUrl":"10.1016/j.rmcr.2025.102169","url":null,"abstract":"<div><div>Broncho-esophageal fistulas (BEFs) are a rare but serious complication that can occur after esophagectomy, often resulting in aspiration, respiratory issues, and infection. Management depends on fistula size and location, with options including conservative treatments, surgical closure and stenting. Conventional treatment involves esophageal stents, which may be insufficient for larger or more complex fistulas. This case report describes the first use of a 3D-printed airway stent in combination with an esophageal stent to treat a broncho-esophageal fistula. A 70-year-old patient with distal esophageal adenocarcinoma, treated with neoadjuvant chemoradiation, underwent robot-assisted minimally invasive esophagectomy. The procedure was complicated by a broncho-esophageal fistula, leading to multiple interventions. Despite dual stenting with a custom 3D airway stent, the fistula persisted, and the patient was transitioned to supportive care due to disease progression. This case, the first to use a 3D-printed airway stent for a broncho-esophageal fistula, demonstrates that the stent did not achieve closure, likely due to excessive pressure against the endobronchial wall. This underscores the need for improved 3D stent designs, offering important insights for interventional pulmonologists.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"53 ","pages":"Article 102169"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143155575","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":"Endobronchial hamartomas as a rare cause of chronic cough","authors":"Selsabil Daboussi ,&nbsp;Asma Saidane ,&nbsp;Abdellatif Syrine ,&nbsp;Samira Mhamdi ,&nbsp;Faten Gargouri ,&nbsp;Houssem Messaoudi ,&nbsp;Saber Hachicha ,&nbsp;Chiraz Aichaouia ,&nbsp;Zied Moatemri","doi":"10.1016/j.rmcr.2025.102210","DOIUrl":"10.1016/j.rmcr.2025.102210","url":null,"abstract":"<div><div>Hamartochondromas are rare benign lung tumors arising from the mesenchymal cells. The endobronchial location is not common (1.4 %).The symptoms are not specific and misleading mimicking a wide spectrum of diseases (Asthma, COPD, Bronchitis …) resulting in a diagnosis delay. We presented here a case of a 64-year patient who had complained of a persistent non-resolving chronic cough despite symptomatic treatments. The diagnosis of an endobronchial hamartoma was made via a repeat bronchial biopsy. The flexible endoscopy showed a white smooth polypoid mass occluding the lumen of the left laterobasal bronchus. A routine surveillance was initially considered. After a 12-year regular follow-up, he was admitted in our department of Pneumology for a recurrent pneumonia. The chest CT scan showed an endobronchial mass occluding the left laterobasal bronchus associated with an obstructive pneumonia. So, he underwent surgery. This benign neoplasia was totally removed by a segmentectomy. The post-operative macroscopic examination revealed a white, small, smooth, endobronchial mass with lobulated margins. The definitive histological exam showed a mixture of mature cartilage islands, mesenchymal tissue and fat. Therefore, the diagnosis of an endobronchial hamartoma was assessed. He was doing well one week after his hospital discharge. We also highlighted this benign lung tumor main clinical presentations, radiological findings as well as the therapeutic strategies and the outcomes.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102210"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143839500","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":"Efficacy and safety of lenvatinib in a case of thymic carcinoma complicated with interstitial lung disease and anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis: A case report","authors":"Yuki Hatakeyama ,&nbsp;Jun Sakakibara-Konishi ,&nbsp;Masato Tarumi ,&nbsp;Kosuke Tsuji ,&nbsp;Hirofumi Takahashi ,&nbsp;Megumi Furuta ,&nbsp;Yuta Takashima ,&nbsp;Hidenori Kitai ,&nbsp;Tetsuaki Shoji ,&nbsp;Yasuyuki Ikezawa ,&nbsp;Satoshi Konno","doi":"10.1016/j.rmcr.2025.102181","DOIUrl":"10.1016/j.rmcr.2025.102181","url":null,"abstract":"<div><div>Based on the results of a multicenter phase II study of patients with previously treated thymic carcinoma, lenvatinib administration for unresectable thymic cancer has been covered under insurance in Japan since 2021. However, patients with interstitial lung disease (ILD) were excluded from that study; therefore, the efficacy and safety of lenvatinib in these patients remain unknown. Herein, we report the case of a woman in her 50s who was diagnosed with thymic carcinoma complicated with ILD. In August 2016, the patient developed ILD with anti-melanoma differentiation-associated gene 5 (MDA5) antibody-positive dermatomyositis (DM). She received triple therapy comprising prednisolone, tacrolimus and azathioprine. In October 2021, the patient complained of lateral chest pain and back pain. In January 2022, computed tomography (CT) revealed an anterior mediastinal tumor, and percutaneous biopsy resulted in a diagnosis of thymic carcinoma with Masaoka classification IVb. In March 2022, first-line treatment with four cycles of carboplatin (area under the curve, 6) + paclitaxel (200 mg/m²) was initiated. Although a partial response was achieved, in September 2022, CT demonstrated progressive disease (PD). Therefore, in October 2022, Lenvatinib (24 mg) was started as the second-line treatment. The best response was stable disease; moreover, although lenvatinib dose reduction was required owing to adverse events, such as biliary-tract infection and stomatitis. The patient did not experience ILD exacerbation. Lenvatinib (14 mg) was continued until PD was observed in March 2023. Our findings suggest that lenvatinib is a viable treatment option for thymic carcinoma with ILD.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"54 ","pages":"Article 102181"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143454507","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":"Metastatic tracheal melanoma misdiagnosed as chronic obstructive pulmonary disease: A case report","authors":"Arshdeep Singh Marwaha ,&nbsp;Donald Cockcroft ,&nbsp;Julian Tam ,&nbsp;Brianne Philipenko","doi":"10.1016/j.rmcr.2025.102262","DOIUrl":"10.1016/j.rmcr.2025.102262","url":null,"abstract":"<div><h3>Introduction/objective(s)</h3><div>Metastatic tracheal melanoma is rare, with fewer than 20 reported cases. This case describes a 62-year-old female with a history of cutaneous melanoma excised 10 years prior, initially misdiagnosed with severe COPD. We highlight the diagnostic challenges when rare metastases mimic common conditions.</div></div><div><h3>Description</h3><div>Diagnosed with COPD based on dyspnoea and spirometry, the patient later developed worsening symptoms, including haemoptysis, requiring hospitalisation. A chest radiograph was unremarkable, but CT pulmonary angiogram revealed a 1.6 × 1.3 cm tracheal mass. Bronchoscopy confirmed 80–90 % luminal stenosis due to a friable mass, which biopsy identified as tracheal melanoma (BRAF V600E positive). She underwent tumor debulking via rigid bronchoscopy, followed by radiation therapy and vemurafenib.</div></div><div><h3>Discussion</h3><div>This case represents the longest interval between cutaneous melanoma and tracheal metastasis. Spirometry showed a COPD-like scooping pattern rather than the expected large airway obstruction, delaying diagnosis. New-onset severe airflow obstruction in patients with minimal smoking history should prompt alternative considerations. Advanced imaging and bronchoscopy are essential for early detection. Treatment includes surgical debulking, radiation, and targeted therapy, with follow-up showing symptom resolution and normalised spirometry.</div></div><div><h3>Conclusion</h3><div>Metastatic tracheal melanoma can mimic COPD, leading to misdiagnosis. The prolonged latency highlights the need for vigilance in melanoma follow-up. Rare airway lesions should be considered in atypical COPD presentations, reinforcing the importance of advanced diagnostic tools for timely identification and treatment.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102262"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144666025","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":"Acute myocardial infarction following inhaled treprostinil initiation","authors":"Bilal Alqam ,&nbsp;Sarah Medrek","doi":"10.1016/j.rmcr.2025.102184","DOIUrl":"10.1016/j.rmcr.2025.102184","url":null,"abstract":"<div><div>An 81-year-old man with a history of interstitial lung disease attributed to idiopathic pulmonary fibrosis, severe aortic stenosis, and stable coronary artery disease was started on inhaled treprostinil for pulmonary hypertension associated with interstitial lung disease to optimize hemodynamics prior to the valve replacement procedure. However, two days after starting this treatment, the patient presented with an inferior-posterior ST elevation myocardial infarction. Urgent coronary angiography revealed a 95 % proximal right coronary artery stenosis, successfully treated with percutaneous coronary intervention and drug-eluting stent placement. This case raises a question of whether there could be a potential association between inhaled treprostinil initiation and acute myocardial infarction in patients with underlying coronary artery disease. With the documented stability of the patient's coronary artery disease prior to medication initiation, it is plausible to question whether treprostinil may have played a role in plaque destabilization.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"54 ","pages":"Article 102184"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143527248","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":"Non-classical pulmonary exacerbation in cystic fibrosis revealing ALK-Translocated lung cancer: A case report","authors":"Mohamad Hadhud ,&nbsp;Johnathan Arnon ,&nbsp;Anat Hershko-Moshe ,&nbsp;Adi Hollander ,&nbsp;Noa Hurvitz-Lehmann ,&nbsp;Assaf Potruch ,&nbsp;Henny Azmanov ,&nbsp;Rottem Kuint ,&nbsp;Nurith Hiller ,&nbsp;Elie Picard ,&nbsp;Naama Sebbag-Sznajder ,&nbsp;Shira Leebhoff ,&nbsp;Michael Wilschanski ,&nbsp;Myriam Grunewald ,&nbsp;Liron Birimberg-Schwartz ,&nbsp;Malena Cohen-Cymberknoh","doi":"10.1016/j.rmcr.2025.102171","DOIUrl":"10.1016/j.rmcr.2025.102171","url":null,"abstract":"<div><div>Lung cancer is uncommon among people with cystic fibrosis (pwCF). We describe the case of a 35-year-old man with mild, stable CF disease who presented with severe respiratory distress, systemic symptoms, elevated liver enzymes and hypereosinophilia along with a lung mass and pleural effusion. The patient was subsequently diagnosed with non-small cell lung carcinoma (NSCLC), featuring anaplastic lymphoma kinase (ALK) translocation. Following treatment with a targeted tyrosine kinase inhibitor (TKI) there was a rapid tumor regression, however, his dyspnea and hypoxemia subsequently worsened. A trial of Elexacaftor/Tezacaftor/Ivacaftor (ETI) led to significant clinical improvement and enhanced pulmonary function. <em>In vitro</em> testing using patient-derived intestinal organoids was performed in parallel and also demonstrated a significant response to ETI. The deterioration observed following the initiation of ALK inhibitor treatment and subsequent improvement with CFTR modulators suggest that ALK inhibitor therapy may potentially impair CFTR activity. A better understanding of the relationship between these pathways could provide valuable insights and contribute to the development of more effective and tailored treatment strategies for patients with coexisting conditions<em>.</em> To our knowledge, this is the first reported case of ALK-translocated lung cancer in a CF patient, underscoring the necessity for a high degree of clinical suspicion in atypical presentations of pulmonary exacerbation and potentially linking ALK-EML4 activation pathways, TKI therapy and CFTR. Care for pwCF with lung cancer requires a unique multi-disciplinary approach to optimize their complex multifactorial treatment.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"53 ","pages":"Article 102171"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143155577","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}