Respiratory Medicine Case Reports最新文献

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Combined extracorporeal membrane oxygenation support and patent ductus arteriosus ligation following surgical correction for congenital diaphragmatic hernia, a case report and literature review 先天性膈疝手术矫治后联合体外膜氧合支持及动脉导管未闭结扎1例报告并文献复习
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102215
Kun-Yao Hong , Zhi Zheng , Yi-Rong Zheng , Hong Liang , Liang Gao , Yu-Cong Lin , Jin-Xi Huang , Qiang Chen , Xin-Zhu Lin
{"title":"Combined extracorporeal membrane oxygenation support and patent ductus arteriosus ligation following surgical correction for congenital diaphragmatic hernia, a case report and literature review","authors":"Kun-Yao Hong ,&nbsp;Zhi Zheng ,&nbsp;Yi-Rong Zheng ,&nbsp;Hong Liang ,&nbsp;Liang Gao ,&nbsp;Yu-Cong Lin ,&nbsp;Jin-Xi Huang ,&nbsp;Qiang Chen ,&nbsp;Xin-Zhu Lin","doi":"10.1016/j.rmcr.2025.102215","DOIUrl":"10.1016/j.rmcr.2025.102215","url":null,"abstract":"<div><div>Our department recently achieved a successful outcome in a case of right-sided CDH. The patient required emergent surgery for right-sided diaphragmatic hernia repair due to severe hypoxemia, followed by urgent bedside venous-arterial ECMO and simultaneous bedside ECMO-assisted patent ductus arteriosus ligation, necessitated by severe hypoxemia and pulmonary hypertension. During the second day on ECMO, the patient developed intracranial hemorrhage, leading to the performance of a lateral ventriculostomy. Fortunately, the patient was successfully weaned off ECMO. This report presents an analysis of the clinical data from this case and shares insights from our experience.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102215"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143895730","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
Chylothorax complicating extrathoracic sarcoidosis 乳糜胸并发胸外结节病
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102230
Tarik Al-Bermani , Ralph Sabang , Oleg Epelbaum
{"title":"Chylothorax complicating extrathoracic sarcoidosis","authors":"Tarik Al-Bermani ,&nbsp;Ralph Sabang ,&nbsp;Oleg Epelbaum","doi":"10.1016/j.rmcr.2025.102230","DOIUrl":"10.1016/j.rmcr.2025.102230","url":null,"abstract":"<div><div>Clinically significant pleural effusion is an extremely rare manifestation of sarcoidosis. Chylothorax associated with sarcoidosis is an even rarer phenomenon reported in the literature only as individual cases. According to the available literature, whenever chylothorax has occurred in the setting of sarcoidosis, this has almost invariably been in the setting of lymphadenopathy or at least in the setting of prior or concurrent evidence of thoracic sarcoidosis. We describe a patient with biopsy-proven abdominal sarcoidosis who developed chylothorax in the absence of lymphadenopathy or thoracic involvement and whose chylothorax rapidly and durably resolved after resumption of corticosteroid therapy.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"56 ","pages":"Article 102230"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143907660","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
Clot in transit in a patient with massive pulmonary embolism: A case report of successful catheter directed thrombectomy 大量肺栓塞患者的运输血栓:导管定向取栓成功一例报告
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102205
Patrick C. Magnus , Stephanie Cull
{"title":"Clot in transit in a patient with massive pulmonary embolism: A case report of successful catheter directed thrombectomy","authors":"Patrick C. Magnus ,&nbsp;Stephanie Cull","doi":"10.1016/j.rmcr.2025.102205","DOIUrl":"10.1016/j.rmcr.2025.102205","url":null,"abstract":"<div><div>Clot in transit is a potentially catastrophic entity in the setting of a massive pulmonary embolism. There have been previous reports of various therapeutic strategies however the optimal treatment is unknown. We present a case of massive pulmonary embolism complicated by clot in transit which was quickly recognized and expeditiously treated with catheter directed thrombectomy with a good clinical outcome.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102205"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143892236","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
Response of EGFR-mutated pulmonary pleomorphic carcinoma to pembrolizumab egfr突变的肺多形性癌对派姆单抗的反应
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102234
Yuki Hatakeyama , Hidenori Mizugaki , Noriyuki Yamada , Yasushi Mizukami , Ken Kuwahara , Hajime Asahina , Hirofumi Adachi , Hiroshi Yokouchi , Yoshihiro Matsuno , Satoshi Oizumi
{"title":"Response of EGFR-mutated pulmonary pleomorphic carcinoma to pembrolizumab","authors":"Yuki Hatakeyama ,&nbsp;Hidenori Mizugaki ,&nbsp;Noriyuki Yamada ,&nbsp;Yasushi Mizukami ,&nbsp;Ken Kuwahara ,&nbsp;Hajime Asahina ,&nbsp;Hirofumi Adachi ,&nbsp;Hiroshi Yokouchi ,&nbsp;Yoshihiro Matsuno ,&nbsp;Satoshi Oizumi","doi":"10.1016/j.rmcr.2025.102234","DOIUrl":"10.1016/j.rmcr.2025.102234","url":null,"abstract":"<div><div>Pulmonary pleomorphic carcinoma (PPC) is a rare and aggressive lung malignancy with limited treatment options. While immune checkpoint inhibitors (ICIs) have shown promise in treating PPC, evidence regarding their efficacy in epidermal growth factor receptor (<em>EGFR</em>)-mutated cases remains scarce. We report a case of a woman in her 70s diagnosed with PPC harboring <em>EGFR</em> L858R + E709K mutations and high expression (95 %) of programmed death-ligand 1 (PD-L1). After relapsing from concurrent chemoradiotherapy for a Pancoast tumor, the patient received osimertinib as second-line therapy. Despite an initial response, rapid disease progression necessitated left lower lobectomy, confirming PPC diagnosis. Subsequent treatment with pembrolizumab achieved notably tumor response. Although Grade 3 immune-related colitis developed, it was successfully managed with prednisolone, allowing completion of six courses of pembrolizumab. This case demonstrates the potential efficacy of ICIs in <em>EGFR</em>-mutated PPC, even after epidermal growth factor receptor -tyrosine kinase inhibitor (EGFR-TKI) failure and highlights the importance of appropriate adverse event management. Our findings suggest that ICIs may be a viable treatment option for <em>EGFR</em>-mutated PPC patients, particularly those with high PD-L1 expression.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"56 ","pages":"Article 102234"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144099429","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
Effectiveness of afatinib after long-term gefitinib treatment for EGFR L858R and S768I compound mutation-positive lung adenocarcinoma: A case report 长期吉非替尼治疗EGFR L858R和S768I复合突变阳性肺腺癌后阿法替尼的疗效:1例报告
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102249
Ayuka Dai-Shinozaki , Jun Sakakibara-Konishi , Kanako C. Hatanaka , Kento Wakabayashi , Naofumi Shinagawa , Yoshihiro Matsuno , Yutaka Hatanaka , Satoshi Konno
{"title":"Effectiveness of afatinib after long-term gefitinib treatment for EGFR L858R and S768I compound mutation-positive lung adenocarcinoma: A case report","authors":"Ayuka Dai-Shinozaki ,&nbsp;Jun Sakakibara-Konishi ,&nbsp;Kanako C. Hatanaka ,&nbsp;Kento Wakabayashi ,&nbsp;Naofumi Shinagawa ,&nbsp;Yoshihiro Matsuno ,&nbsp;Yutaka Hatanaka ,&nbsp;Satoshi Konno","doi":"10.1016/j.rmcr.2025.102249","DOIUrl":"10.1016/j.rmcr.2025.102249","url":null,"abstract":"<div><div>Afatinib, a second-generation epidermal growth factor receptor tyrosine kinase inhibitor (EGFR-TKI), irreversibly inhibits the pan-human epidermal growth factor receptor (HER) family. It is effective in patients with lung cancer with various <em>EGFR</em> mutations; however, its efficacy in overcoming resistance following first-line EGFR-TKI treatment remains unclear. Here, we report the case of a 68-year-old woman with lung adenocarcinoma (pStage IA, pT1bN0M0) who underwent surgical resection in March 2012 following several previous lung cancer resections. In November 2012, postoperative recurrence with pleural dissemination led to the detection of the <em>EGFR</em> L858R mutation in the malignant pleural effusion specimen using peptide nucleic acid-locked nucleic acid polymerase chain reaction clamping (PNA-LNA PCR clamp). The patient was treated with gefitinib for 8 years, after disease progression with multiple lung metastases. The metastatic lesions harbored compound <em>EGFR</em> S768I and <em>EGFR</em> L858R mutations, as identified using the Oncomine Dx Target Test Multi-CDx System (ODxTT). Treatment with afatinib resulted in rapid metastatic lesion regression. Reanalysis of the previously resected surgical specimens confirmed the presence of the <em>EGFR</em> S768I and L858R compound mutations in all samples, which suggests that S768I was not an acquired resistance mutation. Furthermore, immunohistochemical analysis revealed an increase in HER2 and HER3 protein expression in the gefitinib-resistant specimens. These findings suggest that HER2 and HER3 upregulation may contribute to gefitinib resistance, and that afatinib may effectively target these resistance mechanisms.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102249"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144572133","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
Empyema associated with Actinomyces georgiae successfully treated with multimodal therapy including surgical intervention: A first case report 包括手术干预在内的多模式治疗成功治疗了与放线菌相关的脓胸:首例报告
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102254
Senichi Fukuda , Yuya Homma , Hiroki Kawakami , Soichiro Yamaji , Yuki Sato , Nao Sato , Reina Idemitsu , Taiki Kawai , Naoki Inoshima , Jun Hayashi , Norihiko Kubota , Tatsuya Nagai , Ayumu Otsuki , Hiroyuki Ito , Hiroshi Sugimura , Kei Nakashima
{"title":"Empyema associated with Actinomyces georgiae successfully treated with multimodal therapy including surgical intervention: A first case report","authors":"Senichi Fukuda ,&nbsp;Yuya Homma ,&nbsp;Hiroki Kawakami ,&nbsp;Soichiro Yamaji ,&nbsp;Yuki Sato ,&nbsp;Nao Sato ,&nbsp;Reina Idemitsu ,&nbsp;Taiki Kawai ,&nbsp;Naoki Inoshima ,&nbsp;Jun Hayashi ,&nbsp;Norihiko Kubota ,&nbsp;Tatsuya Nagai ,&nbsp;Ayumu Otsuki ,&nbsp;Hiroyuki Ito ,&nbsp;Hiroshi Sugimura ,&nbsp;Kei Nakashima","doi":"10.1016/j.rmcr.2025.102254","DOIUrl":"10.1016/j.rmcr.2025.102254","url":null,"abstract":"<div><div>A 77-year-old man presented with right-sided chest pain and dyspnea. Computed tomography revealed a loculated pleural effusion, and thoracentesis yielded purulent fluid. <em>Actinomyces georgiae</em> was identified using pleural fluid culture. Despite treatment with ampicillin-sulbactam and thoracic drainage, the patient's condition worsened, requiring video-assisted thoracoscopic surgery. The patient recovered completely after completing an 11-month course of antibiotics. This is the first reported case of empyema associated with <em>A. georgiae</em>. <em>Actinomyces</em> should be considered as a potential cause of empyema, and a comprehensive approach, including surgical intervention, is necessary for optimal management.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102254"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144569889","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
Using delafloxacin, a 4th generation fluoroquinolone, in combination with nebulised tobramycin to eradicate Pseudomonas aeruginosa in cystic fibrosis 第四代氟喹诺酮类药物德拉沙星联合雾化妥布霉素治疗囊性纤维化患者铜绿假单胞菌
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102267
Michael Kevin Dooney, Tarek Saba, Natasha Pickering, Mohamed Etumi
{"title":"Using delafloxacin, a 4th generation fluoroquinolone, in combination with nebulised tobramycin to eradicate Pseudomonas aeruginosa in cystic fibrosis","authors":"Michael Kevin Dooney,&nbsp;Tarek Saba,&nbsp;Natasha Pickering,&nbsp;Mohamed Etumi","doi":"10.1016/j.rmcr.2025.102267","DOIUrl":"10.1016/j.rmcr.2025.102267","url":null,"abstract":"<div><div>Prompt eradication of <em>Pseudomonas aeruginosa</em> following isolation in sputum samples is a fundamental part of therapy in people with cystic fibrosis in order to prevent chronic infection. Whilst multiple eradication regimens exist, none have been proven to be more efficacious than any other. Eradication treatment is effective but not always successful. Ciprofloxacin has been the preferred choice of oral antimicrobial in these eradication regimens due to its superior in-vitro activity against <em>Pseudomonas aeruginosa</em> compared to other fluoroquinolones. Delafloxacin, a fourth-generation fluoroquinolone, has been shown to have superior activity against <em>Pseudomonas aeruginosa</em> compared to ciprofloxacin in-vitro. We show herein, what we believe is the first documented successful eradication of <em>Pseudomonas aeruginosa</em> in a person with CF following a new isolation of the pathogen using oral delafloxacin in combination with nebulised tobramycin, instead of ciprofloxacin after the failure of first line treatment and the emergence of ciprofloxacin-resistance on antimicrobial sensitivity testing.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102267"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144713329","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
Swyer James MacLeod Syndrome: A rare cause of recurrent respiratory infections case report 斯威尔詹姆斯麦克劳德综合征:一个罕见的原因反复呼吸道感染病例报告
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102261
Mustafa Sahid Mahamoud , Morten Hornemann Borg , Ole Hilberg , Anders Løkke Ottesen
{"title":"Swyer James MacLeod Syndrome: A rare cause of recurrent respiratory infections case report","authors":"Mustafa Sahid Mahamoud ,&nbsp;Morten Hornemann Borg ,&nbsp;Ole Hilberg ,&nbsp;Anders Løkke Ottesen","doi":"10.1016/j.rmcr.2025.102261","DOIUrl":"10.1016/j.rmcr.2025.102261","url":null,"abstract":"<div><div>Swyer-James-MacLeod Syndrome (SJMS) is a rare pulmonary disorder that is often underdiagnosed or misdiagnosed, leading to delays in appropriate management. We present a case of a woman in her fifties with an unusual presentation of COVID-19 with recurrent respiratory infections and unilateral hyperlucent lung who was diagnosed with SJMS. This case report highlights the importance of considering SJMS in patients with recurrent respiratory infections and unilateral hyperlucent lung, shedding light on the treatment and management for this rare condition.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102261"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144604408","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
A rare case of pulmonary pleomorphic carcinoma presenting as a nearly pure ground-glass nodule on computed tomography: A case report 罕见的肺多形性癌在计算机断层扫描上表现为几乎纯粹的磨玻璃结节:1例报告
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102268
Masao Kobayashi , Tomohiro Maniwa , Hisaya Chikaraishi , Hironobu Samejima , Ryu Kanzaki , Hidetoshi Satomi , Keiichiro Honma , Jiro Okami
{"title":"A rare case of pulmonary pleomorphic carcinoma presenting as a nearly pure ground-glass nodule on computed tomography: A case report","authors":"Masao Kobayashi ,&nbsp;Tomohiro Maniwa ,&nbsp;Hisaya Chikaraishi ,&nbsp;Hironobu Samejima ,&nbsp;Ryu Kanzaki ,&nbsp;Hidetoshi Satomi ,&nbsp;Keiichiro Honma ,&nbsp;Jiro Okami","doi":"10.1016/j.rmcr.2025.102268","DOIUrl":"10.1016/j.rmcr.2025.102268","url":null,"abstract":"<div><div>Pleomorphic carcinoma is a rare subtype of non-small cell cancer, accounting for 0.1 %–0.4 % of all pulmonary malignancies, and is associated with a poor prognosis. Peritumoral ground-glass nodules (GGNs) are commonly observed and have been attributed to various causes, including inflammation, alveolar hemorrhage, and adenocarcinoma. Here, we describe the case of a female patient with a nearly pure GGN in the right upper lobe. Bronchoscopic biopsy confirmed a diagnosis of adenocarcinoma, which was consistent with the low fluorodeoxyglucose uptake observed on imaging. No lymph node involvement or distant metastases were detected, and the patient was subsequently referred for surgical resection. Lobectomy with mediastinal lymph node dissection was performed. Pathological examination of the surgical specimen revealed a lepidic-dominant adenocarcinoma with areas of acinar adenocarcinoma and a small sarcomatoid component, leading to a diagnosis of pleomorphic carcinoma. Immunohistological analysis demonstrated thyroid transcription factor-1 and AE1/AE3 positivity in both the adenocarcinoma and sarcomatoid components, whereas E-cadherin expression was absent in the sarcomatoid component. These findings suggest that the adenocarcinoma underwent epithelial–mesenchymal transition, eventually transforming into a sarcomatoid component. The patient received uracil and tegafur as adjuvant chemotherapy and has survived for 10 years postoperatively without recurrence. Pleomorphic carcinoma presenting as a nearly pure GGN is exceedingly rare, and to the best of our knowledge, this is the first reported case. Our findings provide insights into the role of EMT in pulmonary malignancies.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102268"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144687219","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
Clinical characteristics and outcomes in patients with Anti-MDA5 positive interstitial lung disease: A case series from a lung transplant center 抗mda5阳性间质性肺病患者的临床特征和预后:来自肺移植中心的病例系列
IF 0.8
Respiratory Medicine Case Reports Pub Date : 2025-01-01 DOI: 10.1016/j.rmcr.2025.102265
René Hage , Eirini Chatzidaki , Christian F. Clarenbach , Macé M. Schuurmans
{"title":"Clinical characteristics and outcomes in patients with Anti-MDA5 positive interstitial lung disease: A case series from a lung transplant center","authors":"René Hage ,&nbsp;Eirini Chatzidaki ,&nbsp;Christian F. Clarenbach ,&nbsp;Macé M. Schuurmans","doi":"10.1016/j.rmcr.2025.102265","DOIUrl":"10.1016/j.rmcr.2025.102265","url":null,"abstract":"<div><h3>Background</h3><div>Anti-Melanoma Differentiation-5 (MDA5)-positive dermatomyositis (DM) is a rare autoimmune disorder frequently complicated by rapidly progressive interstitial lung disease (RP-ILD), often with poor outcomes. Data from European tertiary care and transplant centers remain limited.</div></div><div><h3>Objective</h3><div>To describe the clinical presentation, treatment course, and outcomes of five patients with <em>anti</em>-MDA5+DM-associated ILD at a lung transplant center.</div></div><div><h3>Methods</h3><div>We retrospectively reviewed five patients with confirmed <em>anti</em>-MDA5 seropositivity and interstitial lung involvement between 2023 and 2025. Demographic data, clinical features, lung function, imaging, antibody levels, immunosuppressive treatment, and outcomes were analyzed.</div></div><div><h3>Results</h3><div>Of the five patients 3 were female and two were male with a median age of 51 years (range 22–69). Clinical severity varied from mild cutaneous and pulmonary disease to fulminant RP-ILD. One patient required extracorporeal membrane oxygenation (ECMO) and eventually underwent lung transplantation. Treatment strategies included high-dose corticosteroids, rituximab, mycophenolate mofetil, tacrolimus, and intravenous immunoglobulins (IVIG). Patients with early initiation of rituximab or IVIG and declining <em>anti</em>-MDA5 antibody titers generally showed pulmonary stabilization. Serial lung function tests correlated with clinical improvement or deterioration. Cutaneous flare-ups persisted in some patients despite pulmonary stability.</div></div><div><h3>Conclusion</h3><div>This case series underscores the heterogeneous trajectory of <em>anti</em>-MDA5+DM-ILD and highlights the importance of timely diagnosis, aggressive immunosuppressive treatment, and early transplant evaluation in severe cases. Serial monitoring of <em>anti</em>-MDA5 antibody titers and lung function testing may help guide therapeutic decisions and prognostication.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"57 ","pages":"Article 102265"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144696961","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
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