Respiratory Medicine Case Reports最新文献

{"title":"Adverse effects of vanzacaftor-tezacaftor-deutivacaftor in a cystic fibrosis population: A case series","authors":"Jordan Saag,&nbsp;Sinead O'Bryant,&nbsp;Kandice Amos,&nbsp;Olivia Marett,&nbsp;Brian Garcia,&nbsp;Stefanie Krick","doi":"10.1016/j.rmcr.2026.102474","DOIUrl":"10.1016/j.rmcr.2026.102474","url":null,"abstract":"<div><div>Vanazacaftor-Tezacaftor-Deutivacaftor (VTD) is a novel cystic fibrosis transmembrane receptor (CFTR) modulator therapy. This development added another highly effective modulator therapy to the market as an alternative to the standard of care, Elexacaftor-Tezacaftor-Ivacaftor (ETI). Phase III trial data comparing VTD and ETI demonstrated non-inferiority in outcomes and similar adverse effect profile and rates. Here we present a case series demonstrating known and potentially unknown side effects, specific to VTD. From January 2025 to June 2025, 38 out of 400 patients at our adult CF center, were switched from ETI to VTD. Six of those patients experienced potential VTD related adverse events. Liver function testing (LFT) elevations were seen in 2/6 cases, worsening pulmonary symptoms in 3/6 cases, dermatologic manifestations in 2/6 cases, hypertension in 1/6 cases, neuropsychiatric symptoms in 2/6 cases, and gastrointestinal symptoms in 2/6 cases. Notably, one case led to hospitalization, with development of a severe rash, thrombocytopenia, and LFT elevations. In each case, all symptoms and laboratory manifestations resolved within days after switching back to ETI. This case series demonstrates both previously recognized and unrecognized adverse effects of VTD in a “real-world population.” The majority of our patients’ presenting symptoms were described in phase III clinical trials, including LFT elevations, pulmonary exacerbations, rash, and neuropsychiatric symptoms. On the contrary, thrombocytopenia, gastrointestinal manifestations, and hypertension have not been described thus far with VTD. Further research and more robust monitoring are needed to determine the overall incidence and risk of lesser-known side effects of novel CFTR modulators.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"63 ","pages":"Article 102474"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148731537","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":"Left main bronchus injury incurred during video double-lumen endobronchial tube intubation: A case report","authors":"Lijie Shi,&nbsp;Hui Li,&nbsp;Yuzhu Zhang,&nbsp;Nianhai Feng,&nbsp;Xiaona An","doi":"10.1016/j.rmcr.2026.102488","DOIUrl":"10.1016/j.rmcr.2026.102488","url":null,"abstract":"<div><div>Endotracheal intubation is a basic skill performed by healthcare professionals worldwide which is generally considered safe and effective. Nevertheless, this method is associated with some risks that, can result in significant issues. Double-lumen endobronchial tubes (DLTs) are commonly used in thoracic surgery as the preferred tool for one-lung ventilation. In recent years, visualization technology has highlighted the advantages of video double-lumen endobronchial tubes (VDLTs), leading to their increased clinical use. Nevertheless, the potential safety hazards remain unaddressed. Herein, we report a case of left main bronchus injury during VDLT intubation in a 59-year-old female patient undergoing right upper and lower lobe resection. After smooth intubation with a 35-Fr VDLT, position was confirmed by fiberoptic bronchoscopy. At the end of surgery, a 2-cm laceration was found on the membranous portion of the left main bronchus. The injury was repaired with 4-0 absorbable sutures, and the patient recovered uneventfully and was discharged on day 5. Such complications should be published to share experiences and raise awareness among anesthesiologists and thoracic surgeons, emphasizing that even with advanced visualization, airway injury remains a possible risk factor.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"63 ","pages":"Article 102488"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148843913","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":"Vitamin C deficiency presenting with mucosal bleeding and worsening dyspnea in severe pulmonary arterial hypertension: a case report","authors":"Lucy Tarcha,&nbsp;Jill Elwing,&nbsp;Jean Elwing","doi":"10.1016/j.rmcr.2026.102509","DOIUrl":"10.1016/j.rmcr.2026.102509","url":null,"abstract":"<div><h3>Background</h3><div>Scurvy is rare in developed countries but continues to occur in individuals with limited dietary variety. Vitamin C deficiency is an underrecognized and potentially reversible cause of mucocutaneous bleeding, anemia, and systemic symptoms. We report a case of vitamin C deficiency presenting with mucosal bleeding and progressive dyspnea in a patient with severe pulmonary arterial hypertension (PAH).</div></div><div><h3>Case presentation</h3><div>A 46-year-old woman with severe PAH and chronic iron deficiency anemia presented with progressive dyspnea, fatigue, extensive bruising, and mucosal bleeding. She was on stable triple therapy for PAH without recent medication changes. Laboratory evaluation demonstrated severe anemia with normal iron stores and profound vitamin C deficiency (&lt;5.7 μmol/L). Oral ascorbic acid supplementation and dietary modification led to rapid resolution of mucosal bleeding and improvement in dyspnea, with stabilization of hemoglobin levels.</div></div><div><h3>Conclusions</h3><div>This case highlights vitamin C deficiency as a reversible cause of mucocutaneous bleeding and anemia that may mimic or contribute to clinical deterioration in patients with complex cardiopulmonary disease. Consideration of micronutrient deficiencies may prevent unnecessary diagnostic evaluation and improve outcomes.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"64 ","pages":"Article 102509"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149161014","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":"Long-term CPAP treatment for OSA in pycnodysostosis: A 15-year longitudinal follow-up of a patient","authors":"Abir Bouhamdi ,&nbsp;Yassin Chefchaou ,&nbsp;Meryem Benjelloun ,&nbsp;Sanae Labyad ,&nbsp;Abdellah Abida ,&nbsp;Zouhayr Souirti ,&nbsp;Sana Abourazzak ,&nbsp;Siham Tizniti ,&nbsp;Mounia Serraj ,&nbsp;Bouchra Amara ,&nbsp;Mohamed Chakib Benjelloun ,&nbsp;Mohammed Elbiaze","doi":"10.1016/j.rmcr.2026.102442","DOIUrl":"10.1016/j.rmcr.2026.102442","url":null,"abstract":"<div><div>Pycnodysostosis is a rare genetic disorder associated with craniofacial anomalies that promote the occurrence of obstructive sleep apnea (OSA). We report the 15-year longitudinal follow-up of a patient with severe OSA treated with continuous positive airway pressure (CPAP). From the age of 2 years, the patient presented severe OSA confirmed by polysomnography and CPAP was initiated. Throughout follow-up, adherence was excellent (≥90%). Despite multidisciplinary follow-up and the addition of a dental prosthesis aimed at targeted expansion of the oral cavity, persistent maxillo-mandibular retrognathia was still present at age 17 years along with OSA persistence. This case highlights the importance of early and periodic follow-up and evaluation of alternative therapeutic approaches to improve upper airway patency in children with craniofacial syndromes such as pycnodysostosis.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"62 ","pages":"Article 102442"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148231665","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":"Tezepelumab in severe asthma with pulmonary mucus plugs and eosinophilic chronic rhinosinusitis after dupilumab intolerance and suboptimal mepolizumab response: A case report","authors":"Satoru Senoo,&nbsp;Kei Fukushima,&nbsp;Kohei Takata,&nbsp;Shunsuke Matsumori,&nbsp;Takahiro Baba,&nbsp;Yuka Sugisaki,&nbsp;Hiroki Omori,&nbsp;Hiroaki Matsuura,&nbsp;Akihiko Taniguchi,&nbsp;Rika Omote,&nbsp;Nobuaki Miyahara,&nbsp;Toshiaki Okada","doi":"10.1016/j.rmcr.2026.102458","DOIUrl":"10.1016/j.rmcr.2026.102458","url":null,"abstract":"<div><h3>Background</h3><div>Tezepelumab, a monoclonal antibody targeting thymic stromal lymphopoietin (TSLP), has demonstrated efficacy in the treatment of severe asthma and chronic rhinosinusitis with nasal polyps. However, real-world evidence remains limited, particularly for patients with eosinophilic chronic rhinosinusitis (ECRS) who exhibit inadequate or unfavorable responses to other biologics.</div></div><div><h3>Case presentation</h3><div>We report the case of a woman with severe asthma complicated by ECRS, who exhibited divergent clinical responses to multiple biologic agents. Treatment with dupilumab initially improved both asthma and sinonasal symptoms, but was discontinued because of persistent marked peripheral eosinophilia and the development of pulmonary ground-glass opacities. Subsequent therapy with mepolizumab led to improvement in eosinophilia and partial resolution of radiologic abnormalities; however, asthma control deteriorated, and chest computed tomography revealed atelectasis and mucus plugs, accompanied by worsening of ECRS. Tezepelumab was subsequently introduced, resulting in marked improvement in asthma control, resolution of pulmonary mucus plugs, and improvement in sinonasal symptoms and radiologic findings.</div></div><div><h3>Conclusion</h3><div>This case highlights the heterogeneous therapeutic responses to biologics targeting different inflammatory pathways in patients with severe asthma and ECRS. Upstream inhibition of TSLP with tezepelumab provided comprehensive control of both pulmonary and sinonasal disease, indicating that tezepelumab may be an effective therapeutic option for selected patients with complex type 2 airway inflammation.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"63 ","pages":"Article 102458"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148503808","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":"Fresh frozen plasma pleurodesis for bronchopleural fistula following secondary spontaneous pneumothorax due to ruptured emphysematous bullae in a high-risk elderly COPD patient","authors":"Afroza Akhter,&nbsp;Shaila Sharmin,&nbsp;Tanima Biswas","doi":"10.1016/j.rmcr.2026.102476","DOIUrl":"10.1016/j.rmcr.2026.102476","url":null,"abstract":"<div><h3>Introduction</h3><div>Persistent air leak (PAL) secondary to bronchopleural fistula (BPF) is a serious complication of secondary spontaneous pneumothorax (SSP) resulting from ruptured emphysematous bullae in patients with chronic obstructive pulmonary disease (COPD). Management is particularly challenging in elderly high-risk individuals who are unsuitable for surgical intervention. Fresh frozen plasma (FFP), containing fibrinogen and coagulation factors, may act as a biological sealant and facilitate pleural closure.</div></div><div><h3>Case report</h3><div>A 76-year-old man, a former smoker with COPD and multiple comorbidities presented with severe acute dyspnea and persistent cough due to right-sided pneumothorax with lung collapse. A water-sealed intercostal chest drain was inserted; although respiratory distress improved, a persistent air leak continued. High-resolution computed tomography confirmed right-sided pneumothorax with associated lung atelectasis and multiple emphysematous bullae. Given the high surgical risk, a total of three units of ABO-compatible fresh frozen plasma (FFP) were administered intrapleurally through the existing chest tube on alternate days. A gradual reduction in air leak was observed, with complete cessation by day 17. The chest tube was removed on day 20 following radiographic confirmation of full lung re-expansion. At 6-month follow-up, the patient remained clinically stable without recurrence of pneumothorax or the need for further pleural intervention.</div></div><div><h3>Conclusion</h3><div>Intrapleural FFP pleurodesis may represent a safe, feasible, and cost-effective alternative for managing persistent air leak in high-risk elderly COPD patients when surgical options are contraindicated. Complete resolution may require a prolonged course; however, the favorable outcome without complications reinforces the value of a carefully selected management strategy in this challenging clinical context.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"63 ","pages":"Article 102476"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148731472","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":"Spontaneous regression of pulmonary pleomorphic carcinoma after thoracoscopic surgery for hemothorax: A case report","authors":"Masashi Nishimura ,&nbsp;Akimasa Sekine ,&nbsp;Sanshiro Haga ,&nbsp;Eri Hagiwara","doi":"10.1016/j.rmcr.2024.102147","DOIUrl":"10.1016/j.rmcr.2024.102147","url":null,"abstract":"<div><div>A 69-year-old man visited our hospital due to left pleural effusion by chest X-ray. Thoracentesis indicated hemothorax and the specimen of pleural effusion confirmed malignant cells. Because anemia became apparent due to persistent bloody pleural effusion, thoracoscopic hemostasis was performed with surgical biopsy for a tumor extending beyond the visceral pleural of the left upper lobe and the patient was finally diagnosed as having a primary pleomorphic carcinoma. Then, the tumor rapidly shrunk and completely resolved in three months. Our case indicates that spontaneous regression possibly develops following thoracoscopic surgery for hemothorax in pulmonary pleomorphic carcinoma patients.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"62 ","pages":"Article 102147"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147994219","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":"Osimertinib-associated cardiac dysfunction in epidermal growth factor receptor-mutant non-small cell lung cancer: a single-center three-case series requiring hospitalization","authors":"Masao Takemura ,&nbsp;Shun Inoue ,&nbsp;Akari Neoi ,&nbsp;Daisuke Yoshida ,&nbsp;Nao Shibata ,&nbsp;Kentaro Hara ,&nbsp;Takami Kobayashi ,&nbsp;Ikuo Wakamatsu ,&nbsp;Kohei Taguchi ,&nbsp;Kazue Umetsu ,&nbsp;Shunichi Kouno ,&nbsp;Kana Ikeda ,&nbsp;Mitsuru Motegi","doi":"10.1016/j.rmcr.2026.102422","DOIUrl":"10.1016/j.rmcr.2026.102422","url":null,"abstract":"<div><h3>Background</h3><div>Osimertinib is frequently used for epidermal growth factor receptor (EGFR)-mutant non-small cell lung cancer (NSCLC), whereas cardiotoxicity signals, including left ventricular ejection fraction (LVEF) decline, heart failure (HF), and corrected QT interval prolongation, have been documented.</div></div><div><h3>Cases</h3><div>We describe three patients without clinical HF at baseline who were hospitalized for symptomatic HF with newly documented LVEF decline during osimertinib therapy. Case 1 (a 61-year-old woman with prior anthracycline exposure) developed early HF at approximately 1 to 2 months. Recovery was incomplete despite discontinuation and guideline-directed HF therapy, and she died at approximately 10 months after HF onset owing to cancer progression. Case 2 (a 72-year-old woman) presented at 12 months. Osimertinib was continued under HF therapy, and LVEF recovered. Case 3 (a 74-year-old woman; fourth-line osimertinib) presented at 60 months (ultra-late). Following a 2-month interruption and HF therapy, osimertinib was reintroduced without recurrence, and LVEF recovered.</div></div><div><h3>Conclusion</h3><div>These cases illustrate that osimertinib-associated cardiac dysfunction can develop from early to ultra-late phases and that selective rechallenge may be considered following HF therapy optimization and recovery with close monitoring. Comparative risk or incidence was not evaluated.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"61 ","pages":"Article 102422"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147803169","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":"Erythema nodosum as a rare presentation of pulmonary tuberculosis in adolescents: A case report","authors":"Harold Escudero-Quintero ,&nbsp;Diana Marcela Mosquera-Romero ,&nbsp;Juliana Taborda-Merchán","doi":"10.1016/j.rmcr.2026.102438","DOIUrl":"10.1016/j.rmcr.2026.102438","url":null,"abstract":"<div><h3>Aim and background</h3><div>Tuberculosis (TB), caused by the <em>Mycobacterium tuberculosis</em> complex, is a multisystemic infectious disease with variable clinical manifestations. Pulmonary involvement is the most common presentation, whereas cutaneous manifestations are rare. Early diagnosis and timely treatment are essential to prevent organ damage, reduce long-term sequelae, and minimize psychosocial impact, particularly in children and adolescents. This case highlights the importance of maintaining a high index of suspicion for TB, even in atypical presentations such as dermatologic findings, especially in high-endemicity settings.</div></div><div><h3>Case description</h3><div>We report the case of an 11-year-old female with confirmed active pulmonary TB who presented with tender nodular lesions on the lower limbs. The diagnosis was established based on clinical, microbiological, immunological, and radiological criteria.</div></div><div><h3>Conclusion</h3><div>A comprehensive clinical approach—integrating detailed history-taking, targeted diagnostic workup, and multidisciplinary evaluation—is key to ensuring treatment success and reducing complications in high-burden diseases like tuberculosis.</div></div><div><h3>Clinical significance</h3><div>Erythema nodosum is a rare initial manifestation of TB in children and adolescents, with limited cases reported in the literature.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"62 ","pages":"Article 102438"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148168972","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 Dieulafoy disease: A case report and literature review","authors":"Zhenggong Wang,&nbsp;Yunfeng He,&nbsp;Miao Huang,&nbsp;Xiongjun Zhang,&nbsp;Rui Ma","doi":"10.1016/j.rmcr.2026.102454","DOIUrl":"10.1016/j.rmcr.2026.102454","url":null,"abstract":"<div><div>Bronchial Dieulafoy disease (BDD) is a rare disease characterized by abnormal dilation or malformation of the submucosal artery of the bronchus. This disease features a low incidence, but its bleeding is rapid and occult, which easily leads to delayed diagnosis, endangers life, and is difficult to handle clinically. Currently, its diagnosis mainly relies on bronchoscopy and angiography, and treatment methods include endoscopic treatment, interventional embolization, and surgical operation. There are still many unsolved problems regarding the exact pathogenesis, treatment timing, treatment method selection, and long-term management of this disease. This article reports a case of a 54-year-old male patient who was admitted due to repeated hemoptysis for 2 years. During hospitalization, bronchoscopy was performed, and the result showed a nodule in the anterior segment of the right upper lung (brown vascular shadow seen on NBI). In view of the patient's repeated hemoptysis and high risk of massive bleeding, Bronchial Artery Embolization (BAE) was performed for him after obtaining the patient's consent. The operation successfully embolized the patient's diseased bronchial artery, and the patient did not experience hemoptysis again after the operation. During the follow-up period outside the hospital, the patient did not have hemoptysis again.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"62 ","pages":"Article 102454"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13312020/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148354680","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}