Respiratory Medicine Case Reports最新文献

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Nodular pulmonary amyloidosis presenting as a localized manifestation of lymphoproliferative disease: A three-case series 结节性肺淀粉样变表现为淋巴细胞增生性疾病的局部表现:三例系列报告
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2025-12-26 DOI: 10.1016/j.rmcr.2025.102353
Sanya Chandna , Rutvik Raval , Tanya Rangwala , Atul C. Mehta
{"title":"Nodular pulmonary amyloidosis presenting as a localized manifestation of lymphoproliferative disease: A three-case series","authors":"Sanya Chandna ,&nbsp;Rutvik Raval ,&nbsp;Tanya Rangwala ,&nbsp;Atul C. Mehta","doi":"10.1016/j.rmcr.2025.102353","DOIUrl":"10.1016/j.rmcr.2025.102353","url":null,"abstract":"<div><div>Pulmonary amyloidosis presents in nodular, tracheobronchial, and diffuse alveolar-septal forms. Nodular pulmonary amyloidosis (NPA) is usually localized and asymptomatic. It is often linked to indolent B-cell lymphoproliferative disorders (LPD). The National Comprehensive Cancer Network recommends long-term follow-up and evaluation for low-grade B-cell lymphoproliferative disorders when specific clinical features are present. However, there are no established screening guidelines specifically for detecting lymphoma in patients with nodular pulmonary amyloidosis. We describe three patients with NPA who later developed LPD. A 53-year-old woman with biopsy-proven κ-type localized AL amyloidosis was later found to have monoclonal gammopathy of undetermined significance. A 63-year-old man with prior hairy cell leukemia had λ-type NPA and subsequently Epstein–Barr virus–positive Hodgkin lymphoma. A 74-year-old man undergoing pneumothorax surgery had NPA with bronchus-associated lymphoid tissue lymphoma and later gastric mucosa-associated lymphoid tissue lymphoma. None of them had systemic amyloidosis at diagnosis. These cases suggest NPA may coexist with or precede LPD, underscoring the importance of longitudinal monitoring. Larger studies are needed to help standardize screening, as early identification of an underlying LPD could potentially improve hematologic outcomes.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"59 ","pages":"Article 102353"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145939250","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}
引用次数: 0
Successful Re-challenge of zongertinib following erythema multiforme-like drug eruption in a patient with HER2-mutant non-small cell lung cancer her2突变型非小细胞肺癌患者多形红斑样药疹后,成功再次使用宗厄替尼
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2026-06-02 DOI: 10.1016/j.rmcr.2026.102444
Kodai Kawamura, Kaori Kubota, Kazuya Ichikado
{"title":"Successful Re-challenge of zongertinib following erythema multiforme-like drug eruption in a patient with HER2-mutant non-small cell lung cancer","authors":"Kodai Kawamura,&nbsp;Kaori Kubota,&nbsp;Kazuya Ichikado","doi":"10.1016/j.rmcr.2026.102444","DOIUrl":"10.1016/j.rmcr.2026.102444","url":null,"abstract":"<div><h3>Background</h3><div>Zongertinib is a novel, oral, HER2-selective tyrosine kinase inhibitor (TKI) that spares wild-type EGFR. While it has a favorable safety profile, severe cutaneous adverse events (scAEs) can occur, especially in the post-immunotherapy setting.</div></div><div><h3>Case presentation</h3><div>A 55-year-old female with HER2-mutant non-small cell lung cancer (NSCLC) was treated with zongertinib as second-line therapy following chemo-immunotherapy with pembrolizumab. By day 14 of zongertinib treatment, she developed a high fever (39.4 °C), generalized targetoid eruptions, and mucosal involvement, leading to a diagnosis of erythema multiforme (EM)-like drug eruption. Although zongertinib was initially discontinued and a reduced-dose re-challenge failed, a subsequent step-up protocol—starting with every-other-day (QOD) followed by daily (QD) dosing—was successful. Serial chest radiographs confirmed significant tumor shrinkage in the left lung, achieving a partial response without recurrence of severe scAEs.</div></div><div><h3>Conclusion</h3><div>A cautious dose-escalation strategy can allow for the continuation of zongertinib even after severe EM-like eruptions, preserving a potent treatment option.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"62 ","pages":"Article 102444"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148168928","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}
引用次数: 0
Light Chain Deposition Disease – presenting with Cystic Pulmonary Disorder as primary manifestation - A case report 轻链沉积病-以囊性肺疾病为主要表现- 1例报告
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2025-12-24 DOI: 10.1016/j.rmcr.2025.102347
M. Krauss-Schilling , E.H. Fyllingen , A. Jørgensen , M. Raki , S. Spetalen , S.G.F. Wahl , T.S. Slørdahl , H. Moaddab Hønnås , Ø. Rasch-Halvorsen
{"title":"Light Chain Deposition Disease – presenting with Cystic Pulmonary Disorder as primary manifestation - A case report","authors":"M. Krauss-Schilling ,&nbsp;E.H. Fyllingen ,&nbsp;A. Jørgensen ,&nbsp;M. Raki ,&nbsp;S. Spetalen ,&nbsp;S.G.F. Wahl ,&nbsp;T.S. Slørdahl ,&nbsp;H. Moaddab Hønnås ,&nbsp;Ø. Rasch-Halvorsen","doi":"10.1016/j.rmcr.2025.102347","DOIUrl":"10.1016/j.rmcr.2025.102347","url":null,"abstract":"<div><div>Light Chain Deposition Disease (LCDD) is a rare disease characterized by deposition of abnormal, non-amyloid, monoclonal light chains in tissues, potentially leading to organ dysfunction or failure. In most cases, LCDD occurs secondary to lymphoproliferative conditions, such as underlying B-cell disease. LCDD mainly affects the kidneys, liver and heart.</div><div>Pulmonary LCDD (pLCDD) is less common, usually asymptomatic and acknowledged post-mortem. Furthermore, pLCDD exists in three forms i.e. an airway dominant, a nodular and a cystic form, which can all arise from a dominant B-cell clone restricted to the lung without multisystemic involvement.</div><div>Herein we present a young man with isolated pLCDD manifested by cystic pulmonary involvement. Surgical lung wedge resection provided histological diagnostic confirmation showing extracellular deposits, which were negative by Congo red staining but positive for κ-light chain by immunohistochemistry and mass spectrometry. There was no sign of underlying hematological disease.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"61 ","pages":"Article 102347"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147613971","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}
引用次数: 0
Extracorporeal membrane oxygenation as a bridge to cardiac catheterization in a pediatric patient with pulmonary arteriovenous malformation: A case report and literature review 体外膜氧合作为儿科肺动静脉畸形患者心导管置入术的桥梁:1例报告并文献复习
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2026-04-22 DOI: 10.1016/j.rmcr.2026.102417
Mohammed Alzaid , Nada Aljassim , Turki Alshahrani , Abubaker Alkalifa , Anfal Alsalhi , Mohammed AlGhoshimi , Omar Altamimi
{"title":"Extracorporeal membrane oxygenation as a bridge to cardiac catheterization in a pediatric patient with pulmonary arteriovenous malformation: A case report and literature review","authors":"Mohammed Alzaid ,&nbsp;Nada Aljassim ,&nbsp;Turki Alshahrani ,&nbsp;Abubaker Alkalifa ,&nbsp;Anfal Alsalhi ,&nbsp;Mohammed AlGhoshimi ,&nbsp;Omar Altamimi","doi":"10.1016/j.rmcr.2026.102417","DOIUrl":"10.1016/j.rmcr.2026.102417","url":null,"abstract":"<div><h3>Introduction</h3><div>Pulmonary arteriovenous malformations (PAVMs) are rare vascular anomalies that can present with profound hypoxemia in children. While transcatheter embolization is the treatment of choice, extreme hypoxemia may preclude safe intervention. Extracorporeal membrane oxygenation (ECMO) can provide temporary stabilization in such scenarios, though its use as a bridge in pediatric PAVM is seldom reported.</div></div><div><h3>Case presentation</h3><div>We describe a 12-year-old girl with no prior comorbidities who presented with severe hypoxemia refractory to maximal ventilatory support. Imaging revealed multiple bilateral PAVMs. Due to worsening desaturation to 60–70% despite high-flow oxygen and intubation, venovenous ECMO was initiated, resulting in improved oxygenation and hemodynamic stability. Once stabilized, she underwent successful staged transcatheter embolization using multiple vascular plugs. ECMO was weaned within 12 hours post-procedure, and the patient was extubated the following day. At follow-up, she remained stable with oxygen saturations of 88–90% on room air and no sequelae from ECMO.</div></div><div><h3>Discussion</h3><div>This case highlights ECMO as a life-saving bridge to intervention in pediatric PAVM with refractory hypoxemia. Multidisciplinary coordination enabled safe transport, comprehensive imaging, and definitive embolization. Literature review confirms that pediatric reports of ECMO use in PAVM are scarce, with most confined to infancy. Our case underscores the role of ECMO in expanding therapeutic options for older children with severe shunt physiology.</div></div><div><h3>Conclusion</h3><div>In children with life-threatening PAVM and critical hypoxemia, ECMO should be considered as a bridging strategy to transcatheter embolization. Early recognition and coordinated multidisciplinary management are essential for favorable outcomes.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"61 ","pages":"Article 102417"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147744591","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}
引用次数: 0
Fogarty balloon–assisted extraction of an impacted friable foreign body causing complete left main bronchus obstruction: A case report 福格蒂球囊辅助取出致左主支气管完全阻塞的易碎异物1例
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2026-06-11 DOI: 10.1016/j.rmcr.2026.102449
Yael Shapira-Galitz , Evgeni Gershman
{"title":"Fogarty balloon–assisted extraction of an impacted friable foreign body causing complete left main bronchus obstruction: A case report","authors":"Yael Shapira-Galitz ,&nbsp;Evgeni Gershman","doi":"10.1016/j.rmcr.2026.102449","DOIUrl":"10.1016/j.rmcr.2026.102449","url":null,"abstract":"<div><div>Foreign body aspiration (FBA) in adults is uncommon but can lead to significant morbidity due to delayed or atypical presentation. Flexible bronchoscopy is the preferred diagnostic and therapeutic modality, although extraction may be challenging depending on the foreign body's characteristics. We report a case of severe airway obstruction caused by a soft organic foreign body successfully managed using a Fogarty balloon catheter technique.</div><div>A 73-year-old man with schizophrenia presented with severe hypoxemia and complete left lung atelectasis. Imaging demonstrated complete obstruction of the left main bronchus by an ovoid foreign body. Following endotracheal intubation, flexible bronchoscopy revealed a whole grape tightly impacted in the bronchus. Conventional forceps extraction attempts failed because of the grape's smooth and friable surface. A longitudinal channel was created through the grape, enabling insertion of a 5-French Fogarty balloon catheter. Balloon inflation and gentle traction allowed successful removal.</div><div>This technique may represent an effective rescue strategy for extracting large, soft organic airway foreign bodies.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"62 ","pages":"Article 102449"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148231667","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}
引用次数: 0
Activin signaling inhibitor Sotatercept in pulmonary veno-occlusive disease: building a bridge to transplantation? 激活素信号抑制剂索特塞普在肺静脉闭塞疾病中的作用:为移植搭建桥梁?
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2026-06-10 DOI: 10.1016/j.rmcr.2026.102448
Marcel Mietzner, Matthias Held, Elena Pfeuffer-Jovic
{"title":"Activin signaling inhibitor Sotatercept in pulmonary veno-occlusive disease: building a bridge to transplantation?","authors":"Marcel Mietzner,&nbsp;Matthias Held,&nbsp;Elena Pfeuffer-Jovic","doi":"10.1016/j.rmcr.2026.102448","DOIUrl":"10.1016/j.rmcr.2026.102448","url":null,"abstract":"<div><h3>Background</h3><div>Pulmonary veno-occlusive disease (PVOD) is a rare form of pulmonary arterial hypertension (PAH). Its prognosis is considerably worse and no medical treatment has a proven efficacy. Vasodilating drugs for PAH may contribute to a pulmonary edema in PVOD. Sotatercept is the latest approved medical treatment for PAH with a unique mode of action and promising treatment effects. However, none of the clinical trials of sotatercept have included patients with PVOD, so the effect of sotatercept is unknown.</div></div><div><h3>Case description</h3><div>We report two cases of hereditary PVOD that were treated with sotatercept in addition to established pulmonary arterial hypertension therapy. Under treatment with sotatercept, both patients showed stabilization of clinical condition and gas exchange, accompanied by slight improvements in DLCOc and KCOc.</div></div><div><h3>Conclusions</h3><div>These two cases suggest that sotatercept may have a beneficial effect on disease stabilization in hereditary PVOD, as reflected by improvements in DLCOc and KCOc as well as stabilization of clinical status. Therefore, it may be a therapeutic option to stabilize patients with PVOD and potentially prolong the bridging time to lung transplantation.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"62 ","pages":"Article 102448"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148231668","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}
引用次数: 0
Temporary airway stenting as a bridge to chemotherapy in primary mediastinal Ewing's sarcoma with critical tracheal compression: A case report 临时气道支架置入术作为原发性纵隔尤文氏肉瘤伴严重气管压迫化疗的桥梁:1例报告
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2026-07-14 DOI: 10.1016/j.rmcr.2026.102469
Maryam Mazraehei Farahani, Sara Mohammadi, Negar Golkarami, Mitra Rezaei, Arda Kiani, Sharareh Seifi
{"title":"Temporary airway stenting as a bridge to chemotherapy in primary mediastinal Ewing's sarcoma with critical tracheal compression: A case report","authors":"Maryam Mazraehei Farahani,&nbsp;Sara Mohammadi,&nbsp;Negar Golkarami,&nbsp;Mitra Rezaei,&nbsp;Arda Kiani,&nbsp;Sharareh Seifi","doi":"10.1016/j.rmcr.2026.102469","DOIUrl":"10.1016/j.rmcr.2026.102469","url":null,"abstract":"<div><h3>Background</h3><div>Primary mediastinal Ewing sarcoma is an exceptionally rare and aggressive malignancy that may rapidly lead to life-threatening compression of mediastinal structures. Management is particularly challenging when critical airway obstruction is present.</div></div><div><h3>Case presentation</h3><div>A 38-year-old woman presented with progressive dyspnea and cough. Chest imaging revealed a large right paratracheal mediastinal mass causing severe extrinsic tracheal compression with near-complete airway narrowing. Diagnosis was established by EBUS-TBNA, and histopathological and immunohistochemical findings were consistent with Ewing sarcoma. Given the imminent risk of respiratory failure, diagnostic EBUS-TBNA and airway stenting were performed during the same procedural session under general anesthesia. A fully covered self-expanding metallic stent was placed to secure the airway and enable safe initiation of systemic chemotherapy. The patient subsequently received VAC/IE chemotherapy. Follow-up imaging demonstrated marked tumor regression, and the stent was successfully removed after three months. At 6-month follow-up, the patient remained asymptomatic without clinical or radiological evidence of recurrence.</div></div><div><h3>Conclusion</h3><div>Temporary tracheal stenting may serve as an effective bridge to definitive therapy in selected patients with mediastinal Ewing's sarcoma and critical airway obstruction. Multidisciplinary management is essential in such high-risk cases.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"63 ","pages":"Article 102469"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148503882","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}
引用次数: 0
A case report of pregnancy complicated by diffuse alveolar hemorrhage: An unexpected link to untreated Graves' disease 妊娠合并弥漫性肺泡出血1例:与未经治疗的格雷夫斯病的意外联系
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2026-07-16 DOI: 10.1016/j.rmcr.2026.102471
Sara Soltanmohammadi, Hesam Amini
{"title":"A case report of pregnancy complicated by diffuse alveolar hemorrhage: An unexpected link to untreated Graves' disease","authors":"Sara Soltanmohammadi,&nbsp;Hesam Amini","doi":"10.1016/j.rmcr.2026.102471","DOIUrl":"10.1016/j.rmcr.2026.102471","url":null,"abstract":"<div><h3>Background</h3><div>Diffuse Alveolar Hemorrhage (DAH) is a life-threatening condition characterized by widespread bleeding within the alveoli, often resulting from pulmonary capillaritis, bland pulmonary hemorrhage, or diffuse alveolar damage. It presents with hemoptysis, dyspnea, hypoxemia, and anemia, and can lead to respiratory failure. This case report highlights a rare instance of DAH in a pregnant woman with untreated Graves' disease, emphasizing the importance of considering uncommon etiologies in the absence of typical triggers.</div></div><div><h3>Case presentation</h3><div>A 36-year-old pregnant woman at 37 weeks of gestation presented with hemoptysis and dyspnea. Initial evaluations, including pulmonary CT angiography, ruled out pulmonary embolism but revealed bilateral diffuse alveolar involvement. Bronchoscopy confirmed DAH, with serologic tests indicating hyperthyroidism due to Graves' disease. The patient was treated with pulse methylprednisolone and later methimazole, leading to symptom resolution. She delivered a healthy baby at 40 weeks via uncomplicated vaginal delivery.</div></div><div><h3>Discussion</h3><div>DAH during pregnancy is rare, and its association with untreated Graves' disease is exceptionally uncommon. In this case, untreated Graves' disease may have contributed to left ventricular diastolic dysfunction, exerting shear stress on pulmonary vasculature and potentially resulting in DAH. The absence of coagulopathy, environmental exposures, or autoantibodies supported this conclusion. This case underscores the need to consider thyroid disorders in the differential diagnosis of bland pulmonary hemorrhage, particularly when other causes are excluded. Early identification and management of underlying causes can significantly improve maternal and fetal outcomes. This report highlights the importance of exploring rare etiologies in cases of DAH, especially in pregnant patients, to ensure timely and effective intervention.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"63 ","pages":"Article 102471"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148503810","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}
引用次数: 0
Isavuconazole as an adjunctive antifungal in the multimodal management of pulmonary aspergilloma and chronic cavitary pulmonary aspergillosis: a single-centre case series Isavuconazole作为辅助抗真菌药物在肺曲菌肿和慢性空腔性肺曲菌病的多模式治疗:单中心病例系列
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2026-08-14 DOI: 10.1016/j.rmcr.2026.102486
Meagan Deviaene, Elaine Dumoulin, Ranjani Somayaji, Christopher H. Mody, Christina S. Thornton
{"title":"Isavuconazole as an adjunctive antifungal in the multimodal management of pulmonary aspergilloma and chronic cavitary pulmonary aspergillosis: a single-centre case series","authors":"Meagan Deviaene,&nbsp;Elaine Dumoulin,&nbsp;Ranjani Somayaji,&nbsp;Christopher H. Mody,&nbsp;Christina S. Thornton","doi":"10.1016/j.rmcr.2026.102486","DOIUrl":"10.1016/j.rmcr.2026.102486","url":null,"abstract":"<div><div>Symptomatic pulmonary aspergilloma and chronic cavitary pulmonary aspergillosis (CCPA) are typically managed with surgical resection or prolonged triazole therapy, but many patients are poor surgical candidates, and many cannot tolerate voriconazole. Our centre has previously described bronchoscopic debridement combined with antifungal therapy as a minimally invasive alternative for patients with accessible intracavitary fungal balls. Isavuconazole, a newer triazole with a more favourable adverse-event and drug–drug interaction profile, has not been well characterised in this setting. We performed a retrospective single-centre review of nine adults with pulmonary aspergilloma or CCPA referred to our interventional pulmonology service who received isavuconazole between September 2022 and September 2025. Of these, six underwent attempted bronchoscopic debridement (four successful, two with limited or unsuccessful access), one required surgical resection after bronchoscopy failed, one was managed medically because the cavity was inaccessible, and one declined further procedure. Three patients had previously received voriconazole; two were switched to isavuconazole because of intolerance and one was re-treated with isavuconazole at recurrence. One patient developed isavuconazole-associated drug-induced liver injury requiring discontinuation. Three patients died during follow-up: two from causes unrelated to aspergillosis (acute coronary event during dialysis; progression of metastatic colorectal cancer) and one from refractory hemoptysis after isavuconazole was stopped for hepatotoxicity. In this small heterogeneous cohort, isavuconazole appeared well tolerated and was a feasible alternative to voriconazole when prolonged adjunctive therapy was required alongside bronchoscopic or medical management of pulmonary aspergilloma and CCPA. Larger comparative studies are needed to define its place in multimodal protocols.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"63 ","pages":"Article 102486"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148731471","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}
引用次数: 0
Symptomatic diffuse pulmonary meningotheliomatosis with persistent diffusing-capacity impairment 症状性弥漫性肺脑膜瘤病伴持续性弥漫性损伤
IF 0.7
Respiratory Medicine Case Reports Pub Date : 2026-01-01 Epub Date: 2026-08-10 DOI: 10.1016/j.rmcr.2026.102483
Thomas Christian Landry, Youjin Kim, Pascale Packia Raj, Sylvia Riley, Nicholas G. Wysham
{"title":"Symptomatic diffuse pulmonary meningotheliomatosis with persistent diffusing-capacity impairment","authors":"Thomas Christian Landry,&nbsp;Youjin Kim,&nbsp;Pascale Packia Raj,&nbsp;Sylvia Riley,&nbsp;Nicholas G. Wysham","doi":"10.1016/j.rmcr.2026.102483","DOIUrl":"10.1016/j.rmcr.2026.102483","url":null,"abstract":"<div><div>Diffuse pulmonary meningotheliomatosis (DPM) is an ultrarare diffuse parenchymal lung disease in which pulmonary meningothelial-like nodules produce a miliary radiographic pattern. Most reported cases are asymptomatic and incidentally detected, and longitudinal physiologic data in symptomatic patients remain scarce. A 52-year-old postmenopausal never-smoker presented with three months of progressive exertional dyspnea. High-resolution computed tomography showed innumerable small bilateral nodules in a diffuse, random miliary distribution. A non-dedicated chest CT obtained approximately three months earlier had not described nodules. Serologic and microbiologic workup was unrevealing, and bronchoscopy was nondiagnostic. Video-assisted thoracoscopic wedge resection showed bland spindle-cell nests, and immunohistochemistry established DPM. Serial pulmonary function testing showed moderate restriction with reduced diffusing capacity and alveolar volume. During follow-up, forced vital capacity and six-minute walk distance improved, while static volumes and the diffusing-capacity deficit persisted. Severe obesity likely contributed to the restrictive defect. At reduced alveolar volume, the transfer coefficient (KCO) remained lower than expected for pure extrapulmonary restriction, raising the possibility of an intrinsic pulmonary component. However, obesity, possible pulmonary vascular disease, and other factors precluded attribution of these abnormalities to DPM alone. Management was conservative, without corticosteroids, and the disease burden remained radiographically stable. Symptomatic DPM may show functional improvement despite persistent restriction and impaired diffusing capacity. DPM should be considered among the causes of diffuse miliary opacities when imaging and serologic evaluation are nondiagnostic.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"63 ","pages":"Article 102483"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148731536","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}
引用次数: 0
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