Respiratory Medicine Case Reports最新文献

{"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":"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":"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":"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}

{"title":"A successful diagnostic case of Varicella zoster virus pneumonia by a Film Array assay for meningitis/encephalitis","authors":"Nobuhiro Asai ,&nbsp;Atsuko Yamada ,&nbsp;Akiko Nakamura ,&nbsp;Tomoko Ohno ,&nbsp;Narimi Miyazaki ,&nbsp;Yuzuka Kawamoto ,&nbsp;Mina Takayama ,&nbsp;Daisuke Sakanashi ,&nbsp;Toshihiro Ohta ,&nbsp;Yuichi Shibata ,&nbsp;Hideo Kato ,&nbsp;Mao Hagihara ,&nbsp;Nobuaki Mori ,&nbsp;Hiroshige Mikamo","doi":"10.1016/j.rmcr.2025.102180","DOIUrl":"10.1016/j.rmcr.2025.102180","url":null,"abstract":"<div><h3>Introduction</h3><div>Although varicella zoster virus (VZV) infection is generally considered self-limiting and shows a favorable outcome, herpes zoster (HZ) can cause several complications such as neurological pain or meningitis, which lowers the patients’ quality of life. HZ rarely manifests VZV pneumonia, resulting in a poor prognosis with a high mortality rate of 10–30 % if requiring a mechanical ventilation. Here, we present a rare successfully managed case of VZV pneumonia.</div></div><div><h3>Case presentation</h3><div>A 90-year-old man with a medical history of angina pectoris, prostate cancer, dementia, and brain infarction was diagnosed with pneumonia and was admitted to our institute. Four days before the admission, he had visited our outpatient clinic, where he had received anti-herpes therapy due to HZ on the right limb. Although TAZ/PIPC was started empirically, his respiratory status deteriorated. On day 3, we performed a Film Array Meningitis/Encephalitis (ME) PCR panel on a sputum specimen and VZV was identified. Thus, he was diagnosed as having VZV pneumonia and antiviral therapy with acyclovir was started. Since the patient's condition was improved, the antiviral therapy was ended on day 18. Despite that his VZV pneumonia was successfully cured, he passed away due to old age on day 63.</div></div><div><h3>Conclusion</h3><div>Due to poor medical conditions, some elderly patients cannot tolerate bronchoalveolar lavage fluid for diagnosing VZV pneumonia. Film Array ME panel on sputum specimen is a useful method to the diagnosis of VZV pneumonia.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"54 ","pages":"Article 102180"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143511740","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":"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":"Dendriform pulmonary ossification in military combat veterans: A case series","authors":"Jeremy T. Hua ,&nbsp;Carlyne D. Cool ,&nbsp;Tami J. Bang ,&nbsp;Silpa D. Krefft ,&nbsp;Richard C. Kraus ,&nbsp;Cecile S. Rose","doi":"10.1016/j.rmcr.2024.102156","DOIUrl":"10.1016/j.rmcr.2024.102156","url":null,"abstract":"<div><div>Dendriform pulmonary ossification (DPO) is a rare condition characterized by mature bone formation in the lung. DPO has been linked to various conditions, but little is known about the link between DPO and hazardous airborne exposures. We queried research databases of military personnel evaluated for deployment-related respiratory diseases at two occupational pulmonary medicine clinics (Colorado, USA) for diagnoses of DPO, and summarized demographics, Gulf War military deployment history, medical history, and pulmonary function testing. Chest imaging was independently reviewed and scored by a thoracic radiologist, and all cases had undergone lung tissue biopsy. We identified five male combat veterans with DPO, median age 49 years [range: 32–64]. All had deployed to Southwest Asia or Afghanistan during the First or Second Gulf War, and all reported frequent, intense exposure to diesel exhaust, burn pit emissions, and sandstorms. Lung physiology was abnormal in all cases. The most prevalent chest imaging and histopathology findings were airway-centric injury, inflammation, and retained particulate matter, suggesting substantial hazardous exposure during military deployment. This case series of a rare lung disease from the only two contemporary Colorado clinics serving previously deployed veterans highlights a potential link between airborne hazards and lung injury leading to DPO. A high index of clinical suspicion combined with a detailed occupational history may reveal additional exposure-related associations with DPO. Access to large medical databases of military veterans with linkage to exposure histories may further elucidate risk factors for lung injury with ossification, paving the way for targeted prevention.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"53 ","pages":"Article 102156"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11773146/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143061332","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":"Intuitive stent simulation with internal cavity airway models created by a consumer-grade three-dimensional printer: A case series","authors":"Naoya Kitamura ,&nbsp;Toshihiro Ojima ,&nbsp;Yoshifumi Shimada ,&nbsp;Keitaro Tanabe ,&nbsp;Ryo Yokoyama ,&nbsp;Naru Kitade ,&nbsp;Koichiro Shimoyama ,&nbsp;Tomoshi Tsuchiya","doi":"10.1016/j.rmcr.2025.102167","DOIUrl":"10.1016/j.rmcr.2025.102167","url":null,"abstract":"<div><div>Airway stenting, which can cause life-threatening complications, requires advanced techniques as well as safe and reliable procedures. Learning effective techniques and accurately identifying and simulating malignant airway stenosis before the procedure are crucial. However, traditional methods that use three-dimensional (3D) computed tomography images displayed on two-dimensional screens have limitations in accurately visualizing and simulating airway conditions. To address these issues, this study presents the creation of 3D airway models using a consumer-grade 3D printer. These cost-effective and time-efficient models replicate the airway lumen, allowing precise stent placement simulations and enhancing the learning process of airway stenting techniques. In a case series of five patients with malignant airway stenosis, these models facilitated effective stent placement, enhanced the understanding of airway anatomy, and contributed to procedural success. This study's findings suggest that the use of 3D airway models has substantial potential for broader clinical applications in airway stenting. However, evidence for the widespread adoption of this technology is insufficient, and further research is needed.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"53 ","pages":"Article 102167"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11788788/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143123635","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":"Idiopathic CD4 lymphocytopenia: An uncommon but fatal cause of Pneumocystis pneumonia","authors":"Julia Schearer ,&nbsp;Christopher Merrick","doi":"10.1016/j.rmcr.2025.102177","DOIUrl":"10.1016/j.rmcr.2025.102177","url":null,"abstract":"<div><div><em>Pneumocystis</em> pneumonia (PCP) is a fungal infection with primary occurrence in immunocompromised individuals by the etiologic agent <em>Pneumocystis jirovecii</em>. In those without previously identified risk factors, maintaining an index of suspicion for this diagnosis with search for underlying immunosuppressive cause is of utmost importance. Here, we present a case of fatal PCP in a patient without a known history of immunocompromise subsequently diagnosed with idiopathic CD4 lymphocytopenia (ICL) with an absolute CD4⁺ T-cell count of 7 cells/uL.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"54 ","pages":"Article 102177"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143349530","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 rare case of a giant pulmonary solitary fibrous tumor: Diagnostic and therapeutic challenges in the absence of malignancy","authors":"Nouman Aziz ,&nbsp;Waseem Nabi ,&nbsp;Sukhrob Makhkamov ,&nbsp;Yasmine Elsherif ,&nbsp;Adnan Bhat ,&nbsp;Sonu Sahni","doi":"10.1016/j.rmcr.2025.102191","DOIUrl":"10.1016/j.rmcr.2025.102191","url":null,"abstract":"<div><h3>Background</h3><div>Solitary fibrous tumors (SFTs) are rare mesenchymal neoplasms predominantly arising from the pleura but occasionally occurring in extrapleural locations. Pulmonary parenchymal SFTs of extreme size are exceedingly uncommon and present diagnostic and therapeutic challenges.</div></div><div><h3>Case presentation</h3><div>We report the case of a 60-year-old female with a 14 x 12 × 23 cm pulmonary SFT presenting with a two-week history of productive cough and significant weight loss. Imaging revealed a massive heterogeneous mass in the left lower lobe, compressing lung parenchyma, causing pleural effusion and atelectasis, and raising concerns for metastatic spread to the liver and spleen. Histological examination confirmed the diagnosis of SFT, with spindle-cell morphology and positive immunohistochemical staining for CD34 and STAT6. Despite the tumor's size and compressive effects, histopathology showed no necrosis, pleomorphism, or high mitotic activity, indicative of a non-aggressive phenotype. The patient underwent successful surgical resection via thoracotomy and is under long-term follow-up.</div></div><div><h3>Discussion</h3><div>This case highlights the unique diagnostic complexity of SFTs, particularly with tumors of exceptional size. While larger SFTs often suggest malignant potential, the absence of typical markers of malignancy in this case emphasizes the unpredictable behavior of these tumors. Imaging and immunohistochemical evaluation are critical for diagnosis, and surgical resection remains the cornerstone of management.</div></div><div><h3>Conclusion</h3><div>This rare case underscores the importance of thorough diagnostic evaluation and surgical management in addressing giant pulmonary SFTs. Despite their potential for malignant transformation, careful histopathological analysis and multidisciplinary collaboration can guide optimal treatment and long-term surveillance for these unpredictable tumors.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102191"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143685860","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}