{"title":"A management of a patient with chronic thromboembolic pulmonary hypertension prior to surgical resection for lung cancer","authors":"Satoshi Isomatsu , Kenichiro Takeda , Yu Shionoya , Toshihiko Sugiura , Shizu Miyata , Shun Imai , Jun Nagata , Yu Taniguchi , Akira Naito , Rika Suda , Ayako Shigeta , Nobuhiro Tanabe , Takuji Suzuki","doi":"10.1016/j.rmcr.2025.102269","DOIUrl":"10.1016/j.rmcr.2025.102269","url":null,"abstract":"<div><div>The basic treatment for chronic thromboembolic pulmonary hypertension (CTEPH) includes lifelong anticoagulant therapy and pulmonary endarterectomy; moreover, balloon pulmonary angioplasty (BPA) and vasodilators are also known to be effective. Surgery is the standard treatment for localized lung cancer. However, no established treatment guidelines exist for cases of coexisting CTEPH and lung cancer.</div><div>The patient was a 55-year-old woman who experienced dyspnea on exertion. She was diagnosed with non-small cell lung cancer, and surgery was scheduled; however, she was also diagnosed with CTEPH during preoperative examinations. She was referred to our hospital, where right heart catheterization revealed a mean pulmonary artery pressure (mPAP) of 39 mmHg. We prioritized the treatment of CTEPH, starting oral riociguat followed by BPA three times. The mPAP decreased to 27 mmHg. Then, right upper lobectomy and lymph node dissection were performed. After surgery, there was no significant worsening of right heart failure, and no recurrence of the lung cancer.</div><div>This case report presents a method for managing both CTEPH and primary lung cancer. The essence was the intensification of CTEPH treatment in anticipation of lobectomy.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102269"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144687220","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}
Arshdeep Singh Marwaha , Donald Cockcroft , Julian Tam , Brianne Philipenko
{"title":"Metastatic tracheal melanoma misdiagnosed as chronic obstructive pulmonary disease: A case report","authors":"Arshdeep Singh Marwaha , Donald Cockcroft , Julian Tam , 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":"Delayed neoadjuvant therapeutic response in rare giant pulmonary tumor: A case report","authors":"Kai Zhang , Yuanyuan Wang , Yi Li","doi":"10.1016/j.rmcr.2025.102270","DOIUrl":"10.1016/j.rmcr.2025.102270","url":null,"abstract":"<div><div>Primary pulmonary synovial sarcoma (PPSS) is an extremely rare intrathoracic soft tissue sarcoma, but there remains a notable absence of consensus guidelines for the diagnosis and treatment of these tumors. In this case, we reported a 35-year-old PPSS patient who received neoadjuvant chemotherapy consisting of adriamycin and ifosfamide combined with anlotinib, a multi-target tyrosine kinase inhibitor. Although initial response assessment was not encouraging, subsequent imaging studies revealed therapeutic efficacy. This case underscores the critical role of multidisciplinary team and neoadjuvant therapy in PPSS management, aiming to raise awareness of this rare malignancy.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102270"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144713252","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":"Massive hemoptysis due to pulmonary artery pseudoaneurysm in invasive aspergillosis in a patient with interstitial lung disease and suspected Aspergillioma: A case report","authors":"Niroshan Ranjan , Angela Ishak , Medha Cherabuddi , Seth Garrett , Radhika Sheth , Anish Wadhwa , Javier I. Diaz Mendoza","doi":"10.1016/j.rmcr.2025.102288","DOIUrl":"10.1016/j.rmcr.2025.102288","url":null,"abstract":"<div><h3>Background</h3><div>Invasive pulmonary aspergillosis (IPA) is a serious complication, particularly in patients with existing structural lung disease like interstitial lung disease (ILD). Angioinvasive IPA can lead to pulmonary artery pseudoaneurysm (PAP), which is a rare but life-threatening source of massive hemoptysis.</div></div><div><h3>Case presentation</h3><div>We present the case of a 78-year-old white American male with ILD (potentially from Agent Orange exposure) and chronic hypoxic respiratory failure, dependent on home oxygen, who presented with recurrent, massive hemoptysis. Computed Tomography Angiography (CTA) showed a 1.5 cm PAP in the right upper lobe cavity. Despite no classical causative immunosuppression, a diagnosis of pulmonary aspergillosis was supported by bronchoalveolar lavage (BAL) culture and galactomannan testing. Although invasive disease was suspected, the possibility of an aspergilloma with a semi-invasive component could not be excluded due to lack of prior imaging. Urgent coil embolization was performed to control the hemorrhage, followed by antifungal treatment with voriconazole.</div></div><div><h3>Conclusion</h3><div>This case underscores the need for early suspicion of pulmonary aspergillosis in ILD patients, utilizing BAL and CTA for diagnosis, and combining endovascular embolization with antifungals. Given the absence of prior imaging and underlying fibrotic lung disease, distinguishing invasive disease from semi-invasive or chronic forms can be challenging.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"58 ","pages":"Article 102288"},"PeriodicalIF":0.7,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145120733","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":"Cytokine release syndrome caused by dual immunotherapy with tremelimumab and durvalumab: A case report","authors":"Takanori Ito, Saiki Yoshimura, Osamu Kanai, Naoki Fujimoto, Takuma Imakita, Issei Oi, Kohei Fujita, Kiminobu Tanizawa","doi":"10.1016/j.rmcr.2025.102283","DOIUrl":"10.1016/j.rmcr.2025.102283","url":null,"abstract":"<div><div>Cytokine release syndrome (CRS) is an acute systemic inflammatory syndrome characterized by fever and organ dysfunction. CRS is increasingly recognized as an immune-related adverse event caused by the use of immune checkpoint inhibitors (ICIs) to treat solid organ tumors. Herein, we report a life-threatening case of CRS that developed immediately after chemotherapy with tremelimumab and durvalumab in a patient with non-small-cell lung cancer. We administered high-dose corticosteroids and tocilizumab to the patient, which resulted in improvement. Regardless of the ICI class or immunoglobulin subclass, CRS should be considered when suspicious symptoms arise after ICI administration.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"58 ","pages":"Article 102283"},"PeriodicalIF":0.7,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145099227","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":"Endobronchial ultrasound-guided transbronchial cryobiopsy through a working channel potentially improving safety and efficacy","authors":"Mikito Suzuki, Kaito Yano, Tomohiro Imoto, Reiko Shimizu, Kazuo Nakagawa","doi":"10.1016/j.rmcr.2025.102293","DOIUrl":"10.1016/j.rmcr.2025.102293","url":null,"abstract":"<div><div>Endobronchial ultrasound (EBUS)-guided transbronchial cryobiopsy (TBCB) is a novel method for the biopsy of hilar and mediastinal lesions. Conventional cryobiopsy requires simultaneous removal of the cryoprobe and EBUS scope from the airway, resulting in delayed observation of the biopsy site and application of hemostasis, which may lead to an increased risk of complications, especially bleeding. Moreover, during repeated passes of the cryoprobe, re-identification of the lesion is time-consuming. We developed a novel method for direct collection through a working channel during EBUS-TBCB. We performed the procedure under local anesthesia without intubation. After EBUS-transbronchial needle aspiration, the cryoprobe was introduced into the target lesion through the puncture tract for real-time EBUS visualization. A freezing time of up to 4 s allowed the cryobiopsy specimens to pass through the working channel. Retaining the EBUS scope within the airway allowed immediate observation of the biopsy site, and to immediately suction and apply hemostasis using an EBUS balloon, which led to safer cryobiopsy even without intubation. The next cryopass could then be performed. In our two representative cases, we made a definite diagnosis with three and four cryobiopsy specimens, with procedure lengths of 2.2 and 3.0 min, respectively. Only mild bleeding was observed in both cases.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"58 ","pages":"Article 102293"},"PeriodicalIF":0.7,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145099225","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 , 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 , Johnathan Arnon , Anat Hershko-Moshe , Adi Hollander , Noa Hurvitz-Lehmann , Assaf Potruch , Henny Azmanov , Rottem Kuint , Nurith Hiller , Elie Picard , Naama Sebbag-Sznajder , Shira Leebhoff , Michael Wilschanski , Myriam Grunewald , Liron Birimberg-Schwartz , 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}
Jordi Costa i Colomer , Marta Garcia-Moyano , Luis Maiz , Santiago Perez-Tarazona , Marta Ruiz de Valbuena , Leticia Ceberio , Patricia Correcher , Natalia Juliá-Palacios , Montse Morales , Gema Pérez-Yagüe , Jesús Villarrubia , Raquel Pérez-Rojo
{"title":"The impact of interstitial lung disease in patients with acid sphingomyelinase deficiency (ASMD) - A case series","authors":"Jordi Costa i Colomer , Marta Garcia-Moyano , Luis Maiz , Santiago Perez-Tarazona , Marta Ruiz de Valbuena , Leticia Ceberio , Patricia Correcher , Natalia Juliá-Palacios , Montse Morales , Gema Pérez-Yagüe , Jesús Villarrubia , Raquel Pérez-Rojo","doi":"10.1016/j.rmcr.2025.102202","DOIUrl":"10.1016/j.rmcr.2025.102202","url":null,"abstract":"<div><h3>Background</h3><div>Lysosomal storage diseases (LSD) are inherited diseases caused by mutations affecting genes encoding the function of lysosomal enzymes. Acid sphingomyelinase deficiency (ASMD) is an ultra-rare, progressive, and often fatal LSD with an estimated prevalence of 1-in-50,000–250,000 individuals. The PrevASMD study established the ASMD prevalence in Spain at 35 patients. Almost all present asymptomatic interstitial lung disease (ILD), since >90 % patients with chronic visceral ASMD have radiographic evidence of ILD.</div></div><div><h3>Objective</h3><div>This case series aims to emphasize the importance of ASMD in adult and pediatric patients with ILD, and to underline the consideration of pulmonary involvement as a key feature of the multisystemic manifestation of ASMD.</div></div><div><h3>Methods</h3><div>This case series describes seven adult and pediatric cases of ASMD in Spain. All presented with pulmonary function impairment. Data were collected on clinical presentation, diagnostic workup, pulmonary function tests, imaging studies, and patient outcomes during follow up.</div></div><div><h3>Results</h3><div>Sphingomyelin accumulation in ASMD leads to multi-systemic disease involving spleen, liver, lungs, bone marrow, and lymph nodes. Almost all patients presented with asymptomatic ILD, but not all of them exhibited respiratory symptoms and not all were referred to pulmonologists. Periodic assessments included pulmonary function testing (forced vital capacity and diffusing capacity for carbon monoxide), O2 saturation, and exercise tolerance testing.</div></div><div><h3>Conclusion</h3><div>ASMD patients presented with different target organ involvement, with lung involvement contributing significantly to patient morbi-mortality. ASMD diagnostic suspicion among pulmonologists treating ILD is thus crucial, as multidisciplinary monitoring of ILD as a feature of ASMD can ensure its correct management and follow-up.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102202"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143873726","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"A successful diagnostic case of Varicella zoster virus pneumonia by a Film Array assay for meningitis/encephalitis","authors":"Nobuhiro Asai , Atsuko Yamada , Akiko Nakamura , Tomoko Ohno , Narimi Miyazaki , Yuzuka Kawamoto , Mina Takayama , Daisuke Sakanashi , Toshihiro Ohta , Yuichi Shibata , Hideo Kato , Mao Hagihara , Nobuaki Mori , 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}