{"title":"Face-mask CPAP as palliative support for diffuse tracheobronchomalacia from ring agenesis in an infant: When sleep becomes therapeutic","authors":"Abir Bouhamdi , Yassin Chefchaou , Nahla En-nejjari , Meryem Benjelloun , Sanae Labyad , Lamiyae Senhaji , Meryem Karhate , Ilham Tadmouri , Badr Alami , Zouhayr Souirti , Mounia Serraj , Bouchra Amara , Mohamed Chakib Benjelloun , Mohammed Elbiaze","doi":"10.1016/j.rmcr.2025.102351","DOIUrl":"10.1016/j.rmcr.2025.102351","url":null,"abstract":"<div><div>Congenital absence or deficiency of tracheal rings is exceptionally rare and may cause diffuse tracheobronchomalacia (TBM) with life-threatening airway collapse. A 6-month-old girl had persistent stridor from birth, cyanotic spells during feeding, and nocturnal desaturation. Cervicothoracic CT suggested severe airway collapse, and rigid bronchoscopy showed a flattened trachea and main bronchi without visible cartilaginous rings, consistent with ring agenesis and diffuse TBM. Curative surgery was not feasible. After in-hospital titration during a nap, face-mask CPAP with heated humidification was prescribed during sleep and postprandial windows. A minimal effective pressure of 6 cmH<sub>2</sub>O stabilized oxygenation and reduced cyanotic episodes, with good tolerance and device-recorded adherence. Despite transient clinical benefit, the infant died at one and a half years of age (18 months) due to disease progression. In extensive congenital TBM from ring agenesis not amenable to reconstruction, targeted face-mask CPAP can provide palliative/bridging support by counteracting dynamic airway collapse at times of highest vulnerability (sleep and after feeding). This case highlights practical aspects of titration, interface choice, and monitoring in infants.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"59 ","pages":"Article 102351"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145927120","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 near-fatal asthma requiring VV-ECMO","authors":"Elisa Bentivegna , Greta Insalata , Luca Paita , Alessia Catalisano , Chiara Allegrini , Giovanni Cianchi , Manuela Bonizzoli , Federico Lavorini , Gianna Camiciottoli","doi":"10.1016/j.rmcr.2025.102348","DOIUrl":"10.1016/j.rmcr.2025.102348","url":null,"abstract":"<div><div>Phase III studies have demonstrated that the anti-TSLP biologic tezepelumab significantly reduces asthma exacerbations in severe asthma, regardless of endotype. However, evidence on its use in near-fatal asthma is lacking. Tezepelumab's ability to reduce bronchial hyperresponsiveness and ILC2, eosinophil, and mast cell activation may contribute to attenuating severe exacerbations.</div><div>We report the case of a 44-year-old female active smoker (25 pack-years) with a six-month history of asthma, admitted to the ICU with near-fatal asthma triggered by influenza A infection. Orotracheal intubation and invasive ventilation were required. Due to persistent ventilatory failure and acidaemia despite high-pressure ventilation, VV-ECMO was initiated. Treatment included oseltamivir, high-dose intravenous steroids, magnesium, and salbutamol. Attempts to reduce sedation repeatedly induced severe bronchospasm.</div><div>After five days on VV-ECMO, given the critical condition and lack of asthma endotype data, azithromycin 250 mg every other day and tezepelumab 210 mg were administered to achieve a potent anti-inflammatory effect. Within 48 hours of tezepelumab administration, progressive weaning from VV-ECMO and ventilation was possible, leading to successful extubation. The patient was transferred to the pulmonology unit for rehabilitation. Forced oscillation techniques revealed reduced reactance and increased resistance at 5 Hz, both improving one month post-discharge.</div><div>This is the second reported case of near-fatal asthma requiring VV-ECMO treated with tezepelumab, and the first in a patient without prior asthma diagnosis. Biologic therapy in acute settings may improve outcomes in refractory near-fatal asthma, warranting further clinical investigation.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"59 ","pages":"Article 102348"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145927193","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 β-cell failure induced by selpercatinib in RET fusion–positive non-small cell lung cancer: A case report","authors":"Hitokazu Tsukao, Ryosuke Kojima, Shinichi Nakanishi, Yudai Miyanishi, Yuya Fujii, Wataru Yamaguchi, Junya Nakaya, Toru Kojima","doi":"10.1016/j.rmcr.2026.102372","DOIUrl":"10.1016/j.rmcr.2026.102372","url":null,"abstract":"","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"59 ","pages":"Article 102372"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146038127","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}
Ye Ji Jung , Sunjin Ryu , Young Kim , Kye Chul Shin , Seongho Jo , Seong Gyu Yoon , Jung Tae Kim
{"title":"Early VV-ECMO enabling immunosuppressive therapy in severe diffuse alveolar hemorrhage due to ANCA-associated vasculitis","authors":"Ye Ji Jung , Sunjin Ryu , Young Kim , Kye Chul Shin , Seongho Jo , Seong Gyu Yoon , Jung Tae Kim","doi":"10.1016/j.rmcr.2026.102394","DOIUrl":"10.1016/j.rmcr.2026.102394","url":null,"abstract":"<div><h3>Background</h3><div>Diffuse alveolar hemorrhage (DAH) is a catastrophic manifestation of ANCA-associated vasculitis (AAV) that can rapidly progress to severe hypoxemic respiratory failure. The optimal sequencing of immunosuppression and extracorporeal support remains unclear, particularly in the setting of active pulmonary bleeding, where anticoagulation during extracorporeal membrane oxygenation (ECMO) presents a major clinical challenge.</div></div><div><h3>Case presentation</h3><div>A 62-year-old previously healthy woman presented with hemoptysis and rapidly progressive hypoxemia. Despite high-flow oxygen therapy and mechanical ventilation, oxygenation continued to deteriorate, prompting initiation of venovenous ECMO with minimized anticoagulation due to ongoing DAH. High-dose methylprednisolone pulse therapy (1 g/day for 3 days) was initiated immediately, followed by intravenous cyclophosphamide (500 mg) based on chest computed tomography findings consistent with pulmonary capillaritis. Oxygenation improved following initiation of immunosuppressive therapy, allowing discontinuation of ECMO within 11 days and extubation within 3 weeks. Renal function also improved without the need for dialysis, despite a peak serum creatinine level of 5.5 mg/dL and nephritic-range proteinuria (urine protein-to-creatinine ratio 4708 mg/g). MPO-ANCA positivity was confirmed prior to the second cyclophosphamide dose.</div></div><div><h3>Outcome</h3><div>No thrombotic complications occurred despite minimized anticoagulation during ECMO support. Follow-up imaging and inflammatory markers demonstrated sustained improvement. The patient continued recovery with tapering glucocorticoids and azathioprine maintenance therapy.</div></div><div><h3>Conclusion</h3><div>This case suggests that venovenous ECMO with individualized anticoagulation may serve as a bridge to definitive immunosuppression in fulminant AAV-associated DAH. Early cyclophosphamide administration timed to worsening organ involvement may help limit multi-organ injury and avert the need for renal replacement therapy.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"60 ","pages":"Article 102394"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147379599","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}
Jia Shi , Rui Jiang , Zixuan Yang , Shan Yang , Xiaobo Hu
{"title":"Multidisciplinary respiratory rehabilitation after spontaneous pneumothorax in cystic lung disease: a case report with literature review","authors":"Jia Shi , Rui Jiang , Zixuan Yang , Shan Yang , Xiaobo Hu","doi":"10.1016/j.rmcr.2025.102345","DOIUrl":"10.1016/j.rmcr.2025.102345","url":null,"abstract":"<div><h3>Background</h3><div>Spontaneous pneumothorax (SP) is a recurrent and debilitating complication in cystic lung diseases (CLD), often requiring invasive interventions and multidisciplinary respiratory rehabilitation (MRR). However, evidence regarding the role of MRR in post-pneumothorax recovery remains limited.</div></div><div><h3>Case report</h3><div>We report a 51-year-old male with bullous CLD and a 30-pack-year smoking history who presented with acute dyspnea and right-sided pneumothorax. Chest CT revealed extensive emphysematous bullae with 60–70 % lung collapse. After initial chest drainage failed to resolve persistent air leakage, the patient underwent video-assisted thoracoscopic surgery (VATS) for bullectomy and pleurodesis. A MRR protocol was initiated on postoperative day (POD) 2.</div></div><div><h3>Outcomes</h3><div>Pulmonary function improved markedly, with FEV1 rising from 27.5 % predicted preoperatively to 55.4 % at the time of POD 48 (Discharge). Activities of daily living scores doubled from 40 to 80 points, reflecting substantial recovery of functional capacity. The patient ultimately returned to independent community living.</div></div><div><h3>Conclusion</h3><div>This case highlights the critical role of MRR in optimizing outcomes after spontaneous pneumothorax in CLD patients. Integration of IMT, airway clearance, and early mobilization contributed to functional restoration despite significant postoperative complications. Furthermore, this finding underscores the value of personalized rehabilitation strategies in complex thoracic conditions.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"59 ","pages":"Article 102345"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145712495","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":"Fatal multi-organ failure following delayed diagnosis of ruptured pulmonary hydatid cyst in an 11-year-Old boy: The airway management dilemma (‘intubate or not intubate')","authors":"Zohreh Shalchi, Nasibeh Ghalandari, Bahman Shams Shafigh","doi":"10.1016/j.rmcr.2026.102498","DOIUrl":"10.1016/j.rmcr.2026.102498","url":null,"abstract":"<div><div>Hydatid disease (cystic echinococcosis), caused by Echinococcus granulosus, is a zoonotic infection that can involve the lungs, particularly in endemic regions. We report an 11-year-old boy with a 4-month history of chronic cough following an upper respiratory infection. He presented acutely with recurrent vomiting, hemoptysis, severe respiratory distress, and hypoxemia (SpO2 82% on oxygen). Blood pressure was preserved and there were no clinical features of anaphylaxis or sepsis at arrival. Imaging confirmed a ruptured left pulmonary hydatid cyst with bronchial fistula; right-sided radiographic opacities were also present and likely reflected aspiration of cyst contents contributing to the initial severity. Emergency thoracotomy, cyst resection, and chest-tube drainage of an associated pneumothorax (reflecting bronchopleural fistula) were performed Imaging revealed a ruptured pulmonary hydatid cyst. He underwent emergency left thoracotomy with cyst resection and chest tube placement. Postoperatively, he developed acute kidney injury with oliguria, managed conservatively, along with ongoing respiratory complications. Albendazole therapy was initiated. This case highlights the diagnostic delays, airway management challenges, and high morbidity of complicated pulmonary hydatid disease in children.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"64 ","pages":"Article 102498"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148949766","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":"Pulmonary dirofilariasis mimicking malignant pulmonary lesions: Two cases presenting as solitary and multiple pulmonary nodules","authors":"Takashi Ishihara, Tomoki Nakagawa, Haruka Kishi, Shota Fujino, Masaya Ohara, Kei Nakano, Rurika Hamanaka, Yoichiro Ikoma, Syunsuke Yamada, Ryota Masuda","doi":"10.1016/j.rmcr.2026.102523","DOIUrl":"10.1016/j.rmcr.2026.102523","url":null,"abstract":"<div><div>Pulmonary dirofilariasis is a relatively rare zoonotic infection caused by <em>Dirofilaria immitis</em>, which often presents as pulmonary nodules mimicking primary lung cancer or metastatic disease on imaging. Herein, we report two cases of pulmonary dirofilariasis diagnosed using video-assisted thoracoscopic wedge resection. A man in his 60s with an 8-mm solitary pulmonary nodule and a woman in her 70s with multiple pulmonary nodules detected during follow-up after endoscopic submucosal dissection for intramucosal colorectal cancer underwent thoracoscopic wedge resection because malignancy could not be excluded. <sup>18</sup>F-fluorodeoxyglucose positron emission tomography/computed tomography (FDG-PET/CT) demonstrated only mild or absent fluorodeoxyglucose uptake in the lesions. Histopathological examination revealed cross sections of <em>D. immitis</em> within the pulmonary arteries, confirming the diagnosis of pulmonary dirofilariasis in both cases. As pulmonary dirofilariasis closely resembles malignant pulmonary lesions on imaging, preoperative diagnosis remains difficult. Accordingly, this disease should be included in the differential diagnosis of solitary and multiple pulmonary nodules. Video-assisted thoracoscopic wedge resection provides a valuable approach as it provides definitive diagnosis and curative treatment.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"64 ","pages":"Article 102523"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149161312","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":"Interstitial lung disease exacerbations following initiation of nerandomilast: A case series","authors":"Traci N. Adams","doi":"10.1016/j.rmcr.2026.102512","DOIUrl":"10.1016/j.rmcr.2026.102512","url":null,"abstract":"<div><div>Nerandomilast is a novel PDE4 inhibitor approved for the treatment of idiopathic pulmonary fibrosis (IPF) and progressive pulmonary fibrosis (PPF). This case series reports 5 patients with an acute exacerbation of IPF or PPF within 2 weeks of administration of nerandomilast. One patient was on background nintedanib and none were on pirfenidone at the time of the exacerbation. All exacerbations were confirmed by a change in dyspnea and/or cough, worsening oxygen requirement, and new ground glass infiltrates on CT. All required hospitalization. A suggested mechanism for exacerbations on nerandomilast includes upregulation of cAMP by nerandomilast, which can drive IL-17 production. IL-17 has a known association with IPF exacerbations. The results of this case series need to be evaluated prospectively and ongoing pharmacovigilance is required.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"64 ","pages":"Article 102512"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149161383","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"A case of marginal zone lymphoma presenting with paraneoplastic vasculitis and pulmonary infiltration","authors":"Nazli Zeynep Uslu , Umit Yilmaz , Ebru Bilir , Merih Kalamanoglu Balci","doi":"10.1016/j.rmcr.2025.102362","DOIUrl":"10.1016/j.rmcr.2025.102362","url":null,"abstract":"<div><h3>Background</h3><div>Paraneoplastic vasculitis is a rare but important manifestation of underlying malignancies, most commonly hematological, and often presents with atypical features resistant to standard immunosuppression. Marginal zone lymphoma (MZL), a low-grade B-cell lymphoma, may infrequently present with pulmonary involvement and paraneoplastic immune phenomena.</div></div><div><h3>Case description</h3><div>We report a 73-year-old male with a history of COPD who presented with progressive dyspnoea, weight loss, lower limb purpura, and pancytopenia. Initial suspicion included pneumonia and COPD exacerbation; however, imaging revealed bilateral pulmonary infiltrates and pleural effusion. Laboratory evaluation showed systemic inflammation and worsening cytopenias. Further workup, including PET-CT and biopsies, identified CD20-positive extranodal marginal zone lymphoma with pulmonary infiltration. The patient developed presumed paraneoplastic vasculitis involving the skin and coronary arteries, in the absence of classical autoimmune markers. Rituximab monotherapy was initiated, resulting in both clinical and radiological improvement.</div></div><div><h3>Conclusions</h3><div>This case highlights the importance of considering underlying malignancy in patients with unexplained systemic inflammation, vasculitic skin lesions, or cytopenias, particularly when autoimmune markers are negative. Prompt histological diagnosis and multidisciplinary collaboration enabled timely immunochemotherapy, resulting in the resolution of both lymphoma and associated paraneoplastic complications.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"59 ","pages":"Article 102362"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145977469","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":"Erratum to Acute β-cell failure induced by selpercatinib in RET fusion–positive non-small cell lung cancer: A case report [Respir. Med. Case Rep. 59 (2026) 102372]","authors":"Hitokazu Tsukao, Ryosuke Kojima, Shinichi Nakanishi, Yudai Miyanishi, Yuya Fujii, Wataru Yamaguchi, Junya Nakaya, Toru Kojima","doi":"10.1016/j.rmcr.2026.102384","DOIUrl":"10.1016/j.rmcr.2026.102384","url":null,"abstract":"<div><h3>Background</h3><div>Selpercatinib, a highly selective RET tyrosine kinase inhibitor, has demonstrated remarkable efficacy in patients with RET fusion–positive non-small cell lung cancer (NSCLC). The underlying mechanisms of rare metabolic adverse events such as hyperglycemia remain poorly understood.</div></div><div><h3>Case presentation</h3><div>A 66-year-old man with type 2 diabetes mellitus (baseline HbA1c: 6.7%) and stage IVB, KIF5B–RET fusion–positive lung adenocarcinoma commenced selpercatinib 320 mg once daily. On day 30, he developed grade 3 hepatotoxicity, prompting a 24-day treatment interruption; therapy was restarted at 160 mg/day. Twenty days after reinitiation, he experienced polydipsia and polyuria. Laboratory evaluation revealed random plasma glucose of 556 mg/dL, HbA1c 11.1 %, and serum C-peptide 0.5 ng/mL. Adrenocorticotropic hormone and cortisol levels were within reference ranges, and he tested negative for anti–glutamic acid decarboxylase antibodies. A glucagon stimulation test demonstrated a blunted C-peptide response, confirming profound insulin secretory failure. Intensive insulin therapy rapidly normalized glycaemia, allowing selpercatinib to be resumed at 240 mg/day with ongoing tumor control.</div></div><div><h3>Conclusion</h3><div>This case suggests that selpercatinib may precipitate acute β-cell failure, potentially through off-target inhibition of fibroblast growth factor receptors. Regular monitoring of plasma glucose and C-peptide concentrations should be considered, particularly in patients with preexisting diabetes treated with selpercatinib.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"60 ","pages":"Article 102384"},"PeriodicalIF":0.7,"publicationDate":"2026-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147539617","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}