{"title":"Isolated unilateral absence of the pulmonary artery (IUAPA) presenting with progressive interstitial shadows: A case report.","authors":"Tomohito Takeshige, Ryo Koyama, Makiko Kohmaru, Takashi Akimoto, Junko Watanabe, Toshifumi Yae, Kenji Kido, Kazuhisa Takahashi","doi":"10.1002/rcr2.70048","DOIUrl":"https://doi.org/10.1002/rcr2.70048","url":null,"abstract":"<p><p>Unilateral absence of the pulmonary artery (UAPA) is a rare congenital condition. When UAPA occurs without associated congenital heart disease, it is referred to as isolated unilateral absence of the pulmonary artery (IUAPA). IUAPA is frequently not diagnosed until adulthood. A 61-year-old female patient presented with right chest pain and a worsening interstitial shadow over 9 years. Contrast-enhanced computed tomography revealed the absence of blood flow from the pulmonary artery in the right lung, leading to the diagnosis of IUAPA. In this case, the diagnosis of UAPA was established after approximately 40 years. This case underscores the importance of identifying vascular abnormalities to differentiate this condition in patients presenting with nonspecific symptoms and interstitial shadows.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11480744/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477284","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}
Respirology Case ReportsPub Date : 2024-10-16eCollection Date: 2024-10-01DOI: 10.1002/rcr2.70050
Mohammad Hadi Tajik Jalayeri, Mehrdad Aghaei, Mahdi Mazandarani, Narges Lashkarbolouk, Ali Sharifpour
{"title":"Diagnosis of pulmonary lophomoniasis infection in patient with systemic lupus erythematosus; A case report and literature review.","authors":"Mohammad Hadi Tajik Jalayeri, Mehrdad Aghaei, Mahdi Mazandarani, Narges Lashkarbolouk, Ali Sharifpour","doi":"10.1002/rcr2.70050","DOIUrl":"https://doi.org/10.1002/rcr2.70050","url":null,"abstract":"<p><p>Over the past 30 years, there has been an increasing number of documented human infections associated with the protozoan Lophomonas, specifically <i>Lophomonas blattarum</i>, which is considered a relatively rare infection. These infections are primarily associated with states of immune suppression, including those resulting from corticosteroid therapy. We report a 61-year-old female patient with a 20-year medical history of Systemic lupus erythematosus (SLE) who was admitted due to persistent respiratory symptoms that were unresponsive to treatment. The patient was receiving immunosuppressive therapy for SLE. Upon hospitalization, computed tomography of the lungs revealed the presence of centrilobular nodules exhibiting tree-in-bud patterns, as well as bronchiectasis, predominantly in the middle and lower lobes. Subsequently, the patient underwent bronchoscopy, during which a BAL sample was obtained. Microscopic analysis of the sample indicated the presence of <i>L. blattarum</i>. Clinicians often focus on the primary symptoms of SLE, but they must also consider the risk of severe respiratory complications like lophomoniasis. This condition is critical to address in the management of SLE patients, who are immunosuppressed due to the disease's nature and its treatment.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11483512/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477282","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}
Respirology Case ReportsPub Date : 2024-10-15eCollection Date: 2024-10-01DOI: 10.1002/rcr2.70049
Keitaro Nakamoto, Keiji Fujiwara, Kozo Morimoto, Ken Ohta
{"title":"Nontuberculous mycobacterial pulmonary disease caused by <i>Mycobacterium seoulense</i>.","authors":"Keitaro Nakamoto, Keiji Fujiwara, Kozo Morimoto, Ken Ohta","doi":"10.1002/rcr2.70049","DOIUrl":"https://doi.org/10.1002/rcr2.70049","url":null,"abstract":"<p><p><i>Mycobacterium seoulense</i> was first isolated and reported in Korea in 2007. We report a rare case of nontuberculous mycobacterial (NTM) pulmonary disease caused by <i>M. seoulense</i>. A woman in her mid-90s was referred to our hospital due to bloody sputum. She had medical histories of chronic heart failure and chemoradiotherapy for right small cell lung carcinoma. Chest computed tomography showed a consolidation on the mediastinal side of the right lung following radiotherapy and nodules with bronchiectasis of the right upper and middle lobes. Acid-fast culture tests of sputum were positive for <i>M. seoulense</i>. We are currently treating her with tranexamic acid and carbazochrome sodium sulfonate and conducting careful follow-up. <i>M. seoulense</i> infection may need to be recognized as one of the notable NTM pulmonary diseases presenting with respiratory symptoms and radiological findings.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11479681/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477286","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}
Respirology Case ReportsPub Date : 2024-10-14eCollection Date: 2024-10-01DOI: 10.1002/rcr2.70041
Eugene MingJin Gan, Carrie Kah-Lai Leong
{"title":"A novel case of simultaneous tracheal stenting and endobronchial valve (EBV) deployment for tracheal stenosis and persistent air leak guided by an endobronchial collateral ventilation assessment system and digital chest drain in malignant esophageal carcinoma.","authors":"Eugene MingJin Gan, Carrie Kah-Lai Leong","doi":"10.1002/rcr2.70041","DOIUrl":"https://doi.org/10.1002/rcr2.70041","url":null,"abstract":"<p><p>Persistent air leak may complicate malignant disease of the thorax, causing significant morbidity and mortality. A 51-year-old male with a 30-pack-year history of smoking was diagnosed with metastatic esophageal carcinoma with invasion into the right upper lobe of the lung. He developed a large right hydropneumothorax complicated by empyema leading to persistent air leak despite the insertion of two chest drains. Computed Tomography imaging with 3-dimensional reconstruction showed severe tracheal compression from the enlarging esophageal tumour as well as bronchopleural fistulas in the right upper lobe. Rigid bronchoscopy with metallic tracheal stenting was performed. This was followed by localization and quantification of air leak with an endobronchial collateral ventilation assessment system and a digital chest drain to guide endobronchial valve placement, in a single operative procedure. This novel combination of interventional bronchoscopy techniques enabled successful transition to an ambulatory chest drain and subsequent hospital discharge.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11471976/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477269","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":"Successful treatment of large hemoptysis and pseudoaneurysm of the pulmonary artery associated to oesophagomediastinal fistula with amphotericin B cholesterol sulfate complex: A case report.","authors":"Zhujun Chen, Jian He, Qin Huang, Peiqiang Liang, Liang Gong, Qiangzhong Pi","doi":"10.1002/rcr2.70047","DOIUrl":"https://doi.org/10.1002/rcr2.70047","url":null,"abstract":"<p><p>Oesophagomediastinal fistula is uncommon. Oesophageal fistulas, may manifest as recurrent pneumonias. While pulmonary infections can lead to pulmonary artery pseudoaneurysms (PAPs), particularly in fungal infections. PAPs pose a rupture risk, potentially causing life-threatening hemoptysis. We report a unique case of a 45-year-old male who presented with sudden cough, dyspnea, and hemoptysis. Bronchoscopy triggered massive hemoptysis, necessitating emergency embolization. Persistent hemoptysis prompted further imaging, revealing an aneurysmal dilation located next to the spine and infectious lesions, suggesting an oesophagomediastinal fistula. After initiating therapy with Amphotericin B Cholesterol Sulfate Complex and fistula closure, the patient's hemoptysis resolved, with imaging resolution of the PAP. Long-term Voriconazole therapy ensured continued improvement. This case highlights the rarity and severity of such fistulas may be associated with fungal infections and PAPs, emphasizing the importance of prompt recognition, aggressive treatment for favourable outcomes.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11473154/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477289","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":"Ramucirumab-induced ascites with endothelial growth factor receptor mutation-positive non-small cell lung cancer: Two case reports.","authors":"Keisuke Shiraha, Tomoki Tamura, Taisaku Koyanagi, Takahiro Umeno, Kazuya Nishii, Shoichi Kuyama","doi":"10.1002/rcr2.70046","DOIUrl":"https://doi.org/10.1002/rcr2.70046","url":null,"abstract":"<p><p>Ramucirumab (RAM) has been approved for the treatment of non-small cell lung cancer (NSCLC). Here, we report two cases of RAM-induced ascites with epidermal growth factor receptor-mutant NSCLC. Patient 1, a 72-year-old man, developed ascites 20 months after erlotinib (ERL) and RAM administration, which resolved after their discontinuation and performing paracentesis. Patient 2, an 83-year-old woman, developed ascites 9 months after ERL and RAM administration, which resolved after RAM discontinuation and furosemide administration. Ramucirumab administration can cause ascites due to increased hepatic sinusoidal pressure. Clinicians should be aware of RAM-induced ascites in patients with NSCLC and should appropriately manage it.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11473188/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477288","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}
Respirology Case ReportsPub Date : 2024-10-13eCollection Date: 2024-10-01DOI: 10.1002/rcr2.70042
Belgundi Preeti Vidyasagar, Harikishan Gonuguntla, Sejal B Radia, Suhas Dhulipala
{"title":"A novel technique of airway silicon stent deployment under vision-Dr. Vidyasagar's technique.","authors":"Belgundi Preeti Vidyasagar, Harikishan Gonuguntla, Sejal B Radia, Suhas Dhulipala","doi":"10.1002/rcr2.70042","DOIUrl":"https://doi.org/10.1002/rcr2.70042","url":null,"abstract":"<p><p>The conventional methods of silicon stent insertion recommend usage of external loading devices, where the stent is folded into the loading device and pushed in to the tracheobronchial tree using an external pusher which is blind, and leads to placement of stent either distally or proximally needing repositioning or is done with fluoroscopy that involves radiation exposure. We demonstrate our experience in 16 cases of successful silicon stent placement using this technique, wherein an Ultrathin flexible bronchoscope or Hopkins Rigid telescope is pushed alongside the forceps that hold upper end of the folded silicon stent allowing stent placement under direct vision with control over the stent. The Proximal end of the stent can be pulled under vision before deployment for appropriate positioning while pulling the rigid barrel. The stent is always under the operator's control providing excellent control over placement, simplifies the procedure and is safe with no reported complications.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11471311/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477280","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}
Respirology Case ReportsPub Date : 2024-10-11eCollection Date: 2024-10-01DOI: 10.1002/rcr2.70045
Po-Keng Su, Chen-Chieh Lin, Szu-Yen Hu, Ming-Hsien Lin, Chun-Yu Wu, Shun-Mao Yang, Takahiro Oto
{"title":"Neoadjuvant immunochemotherapy followed by ex situ lung auto-transplant (Oto procedure) for central lung cancer: A case report with literature review.","authors":"Po-Keng Su, Chen-Chieh Lin, Szu-Yen Hu, Ming-Hsien Lin, Chun-Yu Wu, Shun-Mao Yang, Takahiro Oto","doi":"10.1002/rcr2.70045","DOIUrl":"https://doi.org/10.1002/rcr2.70045","url":null,"abstract":"<p><p>Sleeve and double-sleeve lobectomies are lung-sparing techniques for treating central lung cancers. However, if the tumour extends to involve the bronchi and vessels, lung auto-transplantation may be an alternative to pneumonectomy. Neoadjuvant therapy after surgery is the most common strategy for patients with extensive central lung cancer. Herein, we report a case of central lung cancer in a patient who underwent immunochemotherapy as neoadjuvant therapy following lung auto-transplantation. A 68-year-old man with stage IIIA non-small cell lung cancer and left upper lobe squamous cell carcinoma underwent neoadjuvant immunochemotherapy. Following partial regression, a multidisciplinary team decided on a back-table procedure with auto-lung transplantation after pneumonectomy to preserve pulmonary function. The patient had an uneventful recovery and was discharged after three weeks with no residual tumour or lymph node metastases. Lung auto-transplantation can be successfully performed in non-lung transplantation centres, potentially broadening treatment options for patients with central lung cancer.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11469736/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477285","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}
Respirology Case ReportsPub Date : 2024-10-10eCollection Date: 2024-10-01DOI: 10.1002/rcr2.70037
Jelena Solujic, Phan Nguyen, Peter Bardy, Yao Ly, Emily Lawton
{"title":"A case of VEXAS (vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic) syndrome presenting as progressive multisystem involvement with parenchymal infiltrates following infection with Epstein Barr virus.","authors":"Jelena Solujic, Phan Nguyen, Peter Bardy, Yao Ly, Emily Lawton","doi":"10.1002/rcr2.70037","DOIUrl":"https://doi.org/10.1002/rcr2.70037","url":null,"abstract":"<p><p>VEXAS (vacuoles, E1 enzyme, X-linked, autoinflammatory, and somatic) syndrome is a rare multisystem disease affecting predominantly males over 50 and manifesting as widespread progressive inflammatory sequelae and haematological dysfunction. We describe a patient who presented with systemic symptoms of fevers, night sweats and weight loss, and developed progressive inflammatory sequelae including cutaneous lesions, haematological dysfunction, lymphadenopathy, migratory inflammatory arthropathies, with new pulmonary infiltrates, following infection with Epstein Barr Virus. Laboratory investigations, bronchoscopy, bone marrow biopsy and imaging were consistent with an inflammatory aetiology. The constellation of organ system involvement, laboratory, biopsy, and imaging results were suspicious for VEXAS syndrome, and this diagnosis was confirmed by identification of a somatic mutation in the UBA1 gene following extensive exclusion of infectious and autoimmune causes. Interestingly the onset of the VEXAS syndrome coincided with serological confirmation of Epstein Barr Virus raising the importance of further exploration into the underlying aetiology of VEXAS syndrome.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11466892/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477268","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}
Respirology Case ReportsPub Date : 2024-10-10eCollection Date: 2024-10-01DOI: 10.1002/rcr2.70039
Coskun Ardan Sener, Aylin Ozgen Alpaydın, Oguz Kılınc
{"title":"A rare pathology that mimics lung cancer: IgG4-related vasculitis.","authors":"Coskun Ardan Sener, Aylin Ozgen Alpaydın, Oguz Kılınc","doi":"10.1002/rcr2.70039","DOIUrl":"https://doi.org/10.1002/rcr2.70039","url":null,"abstract":"<p><p>Immunoglobulin-G4 (IgG4)-related disease is essentially a fibro-inflammatory disease that can affect any organ simultaneously or at different times. The disease usually presents with organ growth that mimics a tumour and can affect the lacrimal glands, major salivary glands, pancreas, bile ducts, retroperitoneal area, lungs, kidneys, aorta, meninges and thyroid gland. The immunopathogenesis behind this new disease has not yet been elucidated. Histopathological distinguishing features of the disease include dense lymphoplasmocytic infiltrates dominated by IgG4 positive plasma cells, storiform fibrosis and obliterative phlebitis. The likelihood of developing with immunoglobulin G4 (IgG4-RD) is a recently identified rare systemic fibroinflammatory disease with an estimated incidence of less than 1 in 100,000 people worldwide. We present our case, which was diagnosed with IGG4-related vasculitis by lung fine needle aspiration biopsy, which is very rare in the literature.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":null,"pages":null},"PeriodicalIF":0.8,"publicationDate":"2024-10-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11466831/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142477281","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}