Respiratory Medicine Case Reports最新文献

{"title":"Lung transplantation in a patient with severe bronchiolitis obliterans secondary to Castleman disease.","authors":"Lise Hommelgaard, Michael Perch, Elisabeth Bendstrup","doi":"10.1016/j.rmcr.2025.102266","DOIUrl":"10.1016/j.rmcr.2025.102266","url":null,"abstract":"<p><p>Castlemann disease (CD) is a rare but often benign condition of the lymph nodes. Seldomly coinciding with CD are other conditions, such as paraneoplastic phemhigus (PMP) and bronchiolitis obliterans (BO). When these occur, patients are at risk of developing severe complications, such as respiratory failure, which may subsequently result in death. Diagnosing and managing conditions related to CD poses a challenge and may cause diagnostic delay. In this case report, we describe the course of illness in a 31-year-old female diagnosed with CD. Prior to the diagnosis of CD, the patient had been diagnosed with both paraneoplastic phemfigus and an irreversible airway obstruction. Standard surgical treatment for CD, as well as standard medical treatment for airway obstruction, had little effect. Radiologic findings supported the diagnosis of bronchiolitis obliterans. A progressive decline in pulmonary function eventually led to the patient being evaluated for and undergoing lung transplantation. The co-occurrence of CD and PMP has previously been described as increasing the risk of developing BO, irreversible pulmonary function restrictions and poor prognosis. Lung transplant may be a possible treatment for some patients with terminal respiratory failure.</p>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"102266"},"PeriodicalIF":0.7,"publicationDate":"2025-07-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12357036/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144876781","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":"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":"Whole lung lavage in the setting of exogenous lipoid pneumonia","authors":"Gaurav A. Jategaonkar ,&nbsp;John Fanous ,&nbsp;Ryan Dunn ,&nbsp;Karen Swanson ,&nbsp;Michael B. Gotway ,&nbsp;Jehad Azar ,&nbsp;Henry D. Tazelaar ,&nbsp;Ana C. Zamora ,&nbsp;Ann M. Rusk","doi":"10.1016/j.rmcr.2025.102274","DOIUrl":"10.1016/j.rmcr.2025.102274","url":null,"abstract":"<div><h3>Introduction</h3><div>Lipoid pneumonia results from lipid deposition in the lungs, arising either exogenously or endogenously. Exogenous lipoid pneumonia occurs from aspiration or use of oil-based products such as mineral oil or nasal topicals. Beyond removing the offending agent, treatment options in adults are limited. Corticosteroids may provide relief, but refractory cases necessitate alternative therapies. Whole lung lavage has shown promise in select pediatric and adult cases. We report a case of steroid-refractory exogenous lipoid pneumonia successfully treated with whole lung lavage.</div></div><div><h3>Case presentation</h3><div>A 56-year-old non-smoker with coronary artery disease and recurrent pneumonia presented with 3 months of fever, weight loss, night sweats, and dyspnea following COVID-19 infection the previous year. Imaging revealed multifocal ground-glass opacities and pleural effusions, with negative infectious workup. Despite corticosteroid treatment, symptoms recurred, leading to multiple Emergency Department visits. Biopsy via bronchoscopy confirmed exogenous lipoid pneumonia, attributed to chronic mineral oil use for constipation. Despite cessation of mineral oil and prolonged steroid courses, his condition worsened. Bronchoalveolar lavage revealed 59 % lipid-laden macrophages. A multidisciplinary team recommended whole lung lavage. The patient underwent the procedure on the left lung, was successfully extubated the following day, and discharged 4 days post-procedure.</div></div><div><h3>Discussion</h3><div>Exogenous lipoid pneumonia is typically managed by stopping the offending agent and corticosteroids, but no standard treatment exists for steroid-refractory cases. Whole lung lavage offers a potential option for symptomatic and imaging improvement. This case underscores the emerging role of whole lung lavage in severe, steroid-resistant exogenous lipoid pneumonia.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102274"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144704428","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":"Clinical efficacy of elexacaftor-tezacaftor-ivacaftor in two siblings with homozygous I1234V mutation cystic fibrosis: A prospective case series","authors":"Mohammed Alzaid ,&nbsp;Turki Alshahrani ,&nbsp;Raed Alotaibi ,&nbsp;Gawahir Mukhtar ,&nbsp;Alanood Alanazi ,&nbsp;Hammad Alsadoon ,&nbsp;Safa Eltahir ,&nbsp;Ahmed Aldraihem ,&nbsp;Wadha Alotaibi","doi":"10.1016/j.rmcr.2025.102264","DOIUrl":"10.1016/j.rmcr.2025.102264","url":null,"abstract":"<div><h3>Background</h3><div>The missense CFTR variant I1234V (c.3700A &gt; G) produces class II protein-folding defects and is prevalent in the Middle East, yet clinical evidence for elexacaftor/tezacaftor/ivacaftor (ETI) in homozygous carriers is sparse. We prospectively evaluated ETI efficacy and safety in two paediatric siblings with homozygous I1234V cystic fibrosis (CF).</div></div><div><h3>Methods</h3><div>A single-centre, prospective case series was undertaken at King Fahad Medical City. Baseline assessments included spirometry, body-mass index (BMI), sputum microbiology, liver biochemistry and high-resolution chest CT. ETI was initiated according to weight-based dosing and patients were reviewed at 6 and 8 months. Primary outcomes were change in percent-predicted forced expiratory volume in 1 second (ppFEV₁) and BMI; secondary outcomes were Pseudomonas aeruginosa status, radiological changes and adverse events.</div></div><div><h3>Results</h3><div>After eight months of ETI, ppFEV₁ increased from 36 % to 46 % in the 11-year-old girl and from 57 % to 73 % in the 9-year-old boy. Corresponding BMI rose from 11.71 kg/m² (z = −4.37) to 15.48 kg/m² (z = −1.22) and from 12.69 kg/m² (z = −3.12) to 15.74 kg/m² (z = −0.55), respectively. Chronic P. aeruginosa was eradicated in both patients. Chest CT demonstrated reduced mucus plugging and peribronchial wall thickening with partial regression of cystic bronchiectasis.</div></div><div><h3>Conclusions</h3><div>ETI produced clinically meaningful improvements in lung function, nutritional status, microbiological clearance and radiological appearance in two children homozygous for the rare I1234V mutation. These real-world findings support extending ETI access to patients with rare class II CFTR variants and justify larger multicentre studies to confirm efficacy and long-term safety.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102264"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144704429","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}