{"title":"Idiopathic multicentric Castleman disease developing after a diagnosis of sarcoidosis: A case report and literature review","authors":"Takumi Muramatsu , Masato Kono , Masaki Ishige , Takahiko Saito , Misato Higasa , Fumiya Nihashi , Yuya Aono , Mineo Katsumata , Hideki Miwa , Yoshihiro Miki , Taro Misaki , Yoshiro Otsuki , Dai Hashimoto , Noriyuki Enomoto , Takafumi Suda , Hidenori Nakamura","doi":"10.1016/j.rmcr.2025.102188","DOIUrl":"10.1016/j.rmcr.2025.102188","url":null,"abstract":"<div><div>A 72-year-old woman presented with an abnormal shadow on chest radiograph. She was histologically diagnosed with sarcoidosis 20 years previously, and prednisolone was initiated 8 years previously. Computed tomography revealed centrilobular micronodules and bronchovascular bundle thickening in both lungs with multicentric lymphadenopathies; multiple pulmonary nodular lesions appeared during prednisolone tapering. Laboratory findings included polyclonal hypergammaglobulinemia and elevated interleukin-6 levels. Surgical lung biopsy revealed marked lymphoplasmacytic infiltration with lymphoid aggregates. The patient tested negative for human herpesvirus-8 and clinically diagnosed with idiopathic multicentric Castleman disease. The coexistence of sarcoidosis and Castleman disease is rare; this case improved with additional tocilizumab treatment.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"54 ","pages":"Article 102188"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143528787","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":"Acute fibrinous and organizing pneumonia after lung transplantation: A case report of treatment with infliximab and tocilizumab and literature review","authors":"Christophe Abellan , Foteini Ioakeim , Alessio Casutt , Benoit Lechartier , Zisis Balmpouzis , Samuel Rotman , Leslie Noirez , Isabelle Rochat , John-David Aubert , Angela Koutsokera","doi":"10.1016/j.rmcr.2024.102159","DOIUrl":"10.1016/j.rmcr.2024.102159","url":null,"abstract":"<div><h3>Introduction</h3><div>Acute fibrinous and organizing pneumonia (AFOP) is a severe form of acute lung injury which can occur after lung transplantation. Treatment is empiric, based on immunosuppressive regimens and the mortality rate is very high.</div></div><div><h3>Case presentation</h3><div>We report the case of a young lung transplant (LT) recipient who developed AFOP following a respiratory viral infection while on suboptimal maintenance immunosuppression due to adherence issues. Diagnosis was confirmed by cryobiopsies showing intra-alveolar fibrin balls. Despite high dose systemic corticosteroids, the patient developed severe respiratory failure requiring mechanical ventilation. IV infliximab and tocilizumab were administered. The patient was extubated 11 days later and discharged to home 42 days after intubation with 1L/min O2. She developed severe pleuritic pain needing opioid treatment and died 4 months later.</div></div><div><h3>Conclusion</h3><div>While high-dose systemic corticosteroids remain the first line of treatment, the use of anti TNF-α has shown promising results in case reports. Furthermore, we propose prompt realization of a cytokine panel analysis in both blood and bronchoalveolar lavage to better guide the adjuvant administration of a targeted anti-inflammatory therapy.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"53 ","pages":"Article 102159"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11743898/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143016109","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}
Sebastian Schregenberger , Vera Graup , Adrian Schibli , Benjamin Preiswerk , Irène Laube , Lars C. Huber , Melina Stüssi-Helbling
{"title":"Immune reconstitution inflammatory syndrome (IRIS): Case series and review of the literature","authors":"Sebastian Schregenberger , Vera Graup , Adrian Schibli , Benjamin Preiswerk , Irène Laube , Lars C. Huber , Melina Stüssi-Helbling","doi":"10.1016/j.rmcr.2025.102213","DOIUrl":"10.1016/j.rmcr.2025.102213","url":null,"abstract":"<div><h3>Background</h3><div>Immune-reconstitution inflammatory syndrome (IRIS) is a dysregulated host inflammatory response following the initiation of appropriate therapy targeting an infectious disease. It is most commonly reported in human immunodeficiency virus patients following the initiation of antiretroviral therapy; however, IRIS can also be seen in immunocompromised patients without HIV, when the immune system is recovering. The diagnosis is confirmed using clinical and laboratory data after excluding differential diagnoses and concomitant infections.</div></div><div><h3>Case series</h3><div>Here, we describe three cases of patients with IRIS that were treated at our tertiary care center. The first case involves a paradoxical IRIS in an HIV-positive patient with TB, where the re-initiation of ART led to an inflammatory response despite effective anti-tuberculous treatment (ATT). The second case highlights unmasking IRIS in an HIV-positive patient, where the initiation of ART revealed an underlying Epstein-Barr virus (EBV)-associated B-cell lymphoma. The third case describes paradoxical worsening of pulmonary TB in an HIV-negative patient, expanding the scope of IRIS beyond its conventional association with HIV infection.</div></div><div><h3>Conclusion</h3><div>These cases illustrate the various manifestations of IRIS and emphasize the need for timely diagnosis and appropriate management strategies to mitigate the potentially severe outcomes associated with this syndrome. Our report highlights the challenges faced in the diagnosis of IRIS which impede prompt onset of therapy.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102213"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143829292","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Endobronchial mucormycosis successfully treated with pharmacologic therapy: A rare case report","authors":"Soo Kyun Jung, Keum-Ju Choi","doi":"10.1016/j.rmcr.2025.102232","DOIUrl":"10.1016/j.rmcr.2025.102232","url":null,"abstract":"<div><div>A 36-year-old female patient who did not have a previous history of any disease visited our institution because of shortness of breath. The patient was first diagnosed with diabetes upon admission. Chest computed tomography (CT) scan showed a lesion that completely obstructed the left main bronchus and pneumonia. Due to hypoxia, the patient was placed on mechanical ventilation and admitted to the intensive care unit. Bronchoscopy was performed, and results revealed complete atelectasis with a soft tissue mass at the left main bronchus. Bronchoscopic biopsy confirmed that the patient's condition was endobronchial mucormycosis. Treatment with liposomal amphotericin B was started. After 1 week, atelectasis improved based on the chest radiography result. The patient was successfully extubated. However, 4 weeks after treatment, bronchoscopy still showed complete atelectasis with purulent secretion and a soft tissue mass. After 6 weeks, liposomal amphotericin B was switched to isavuconazole, with a duration of 2 months. After completing the treatment, bronchoscopy showed no endobronchial lesion. Herein, we present a rare case of rapidly progressing endobronchial mucormycosis in a young patient who did not have a previous history of any disease and who was successfully managed with pharmacological therapy.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"56 ","pages":"Article 102232"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144115795","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":"Pulmonary arteriovenous malformation diagnosed by recurrent hemothorax – A case report","authors":"Toshiki Sakuma , Hajime Kasai , Toshihiko Sugiura , Kenichiro Takeda , Minori Watanabe , Kenichi Suzuki , Takashi Urushibara , Takuji Suzuki","doi":"10.1016/j.rmcr.2025.102204","DOIUrl":"10.1016/j.rmcr.2025.102204","url":null,"abstract":"<div><h3>Background</h3><div>A pulmonary arteriovenous malformation (PAVM) is an abnormal connection between a pulmonary artery and vein that causes various complications owing to a right-to-left shunt. However, hemorrhagic complications developing from a PAVM are rare.</div></div><div><h3>Case report</h3><div>We report the case of a 61-year-old woman with a PAVM complicated by hemothorax. She had a history of right hemothorax with no identifiable cause during pregnancy 35 years prior to presentation. Although the PAVM had been identified on chest computed tomography two months previously, the patient was observed. She visited a nearby hospital with back pain and dyspnea without any inciting factor nor history of trauma. Contrast-enhanced chest CT showed a PAVM in the posterior segment of the right upper lobe and another in the apical-posterior segment of the left upper lobe. Detailed examinations suggested both past and present hemothorax due to rupture of the PAVM close to the pleura of the right upper lobe. As there was no progression of the presumed hemothorax during the index hospitalization, the patient was managed conservatively. After the hemothorax resolved, the patient underwent elective coil embolization at our facility. No recurrence of hemothorax was observed over a 10-month follow-up period.</div></div><div><h3>Conclusion</h3><div>A PAVM can cause recurrent hemothorax, and should be considered when evaluating a patient with hemothorax. If the cause of the hemothorax cannot be identified, a PAVM may become apparent after a long period of time.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102204"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143785669","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":"Asymptomatic sigmoid diverticulum perforation-induced subcutaneous, mediastinal, and retroperitoneal emphysema: A case report and literature review","authors":"Daichi Setoguchi , Naoki Iwanaga , Kotaro Nema , Tomoya Hagiwara , Kotaro Hayashida , Koki Yamashita , Tatsuro Hirayama , Masataka Yoshida , Kazuaki Takeda , Shotaro Ide , Masato Tashiro , Takahiro Takazono , Masachika Kitajima , Noriho Sakamoto , Koichi Izumikawa , Katsunori Yanagihara , Keiji Inoue , Hiroshi Mukae","doi":"10.1016/j.rmcr.2025.102229","DOIUrl":"10.1016/j.rmcr.2025.102229","url":null,"abstract":"<div><h3>Background</h3><div>Mediastinal and subcutaneous emphysema usually result from spontaneous rupture of the alveolar wall. We present an educational case of subcutaneous, mediastinal, and retroperitoneal emphysema discovered during a routine medical check-up resulting from an asymptomatic perforation of the sigmoid diverticulum.</div></div><div><h3>Case presentation</h3><div>A 66-year-old man presented to our hospital for his health check-up. A chest X-ray revealed mediastinal emphysema during a physical examination. The patient had no subjective symptoms, fever, or hemodynamic instability. Physical examination revealed a snow grip sensation in the anterior neck but no abdominal tenderness. Blood tests showed an elevated inflammatory response, and a plain chest computed tomography scan revealed subcutaneous emphysema around the neck, as well as mediastinal and retroperitoneal emphysema. The patient was then admitted to the hospital and the patient was treated conservatively. On Day 7 post-admission, the emphysema was mildly relieved. However, on Day 10, the patient developed intestinal obstruction caused by barium. Colonoscopy revealed sigmoid colon perforation. On Day 11, partial resection of the sigmoid colon via laparotomy and colostomy (Hartmann operation) was performed. Postoperative pathology revealed a perforation of the sigmoid colon, which was confirmed to be induced by diverticulitis, as multiple diverticula were simultaneously found in the sigmoid colon.</div></div><div><h3>Conclusions</h3><div>Even in the absence of abdominal symptoms, retroperitoneal emphysema may develop due to perforation of the sigmoid colon. Therefore, if retroperitoneal emphysema is combined with mediastinal emphysema, evaluation, including abdominal CT, should be performed to identify the cause of emphysema.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"56 ","pages":"Article 102229"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143912591","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}
Jinhong Chen , Hongxiang Huang , Peiyuan Zhong , Sujuan Peng , Xie Zhu , Xinjing Ding , Fen Wang , Ping Kong , Tiantian Song , Zhihui Lu , Li Chen
{"title":"EGFR-mutant lung adenocarcinoma transformed into small cell Lung cancer: A case report and literatures review","authors":"Jinhong Chen , Hongxiang Huang , Peiyuan Zhong , Sujuan Peng , Xie Zhu , Xinjing Ding , Fen Wang , Ping Kong , Tiantian Song , Zhihui Lu , Li Chen","doi":"10.1016/j.rmcr.2025.102219","DOIUrl":"10.1016/j.rmcr.2025.102219","url":null,"abstract":"<div><div>Advances in molecular biology have positioned epidermal growth factor receptor tyrosine kinase inhibitors (EGFR-TKIs) as highly effective therapies for patients with EGFR-mutant carcinomas. However, the inevitable emergence of acquired resistance significantly limits their long-term efficacy. Among resistance mechanisms, the transformation of lung adenocarcinoma to small cell lung cancer (SCLC) following EGFR-TKIs therapy is an uncommon but clinically important phenomenon contributing to treatment failure.</div><div>We present a case of SCLC transformation in a patient with EGFR-mutant lung adenocarcinoma after 8 months of first-line osimertinib therapy. Following 4 cycles of etoposide combined with lobaplatin chemotherapy, adenocarcinoma cells regained predominance, illustrating a dynamic histological shift between adenocarcinoma and SCLC phenotypes. Subsequent treatment with 2 cycles of chemotherapy plus osimertinib resulted in disease stabilization. However, multiple brain metastases were identified 3 months after completing 6 cycles of chemotherapy.</div><div>This case underscores the bidirectional histological plasticity between lung adenocarcinoma and SCLC during treatment and highlights the critical importance of repeated biopsies for guiding management strategies in the context of resistance. We also provide a comprehensive review of the clinical manifestations, underlying mechanisms, predictive biomarkers, and therapeutic approaches for SCLC transformation.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102219"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143887839","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}
Mourad Elghali , Mariem Mhiri , Imen Chaabene , Ines Bayar , Bilel Ben Amor , Imen Jaziri , Maha Changuel , Rihab Ben Dhia , Narjes Gouta , Jamel Saad , Rym Hadhri , Ahmed Zrig , Soumaya Boudokhane , Hela Marmouche , Mahbouba Jguirim , Nabil Sakly
{"title":"From infection to autoimmunity: can COVID-19 spark new auto-immune conditions?","authors":"Mourad Elghali , Mariem Mhiri , Imen Chaabene , Ines Bayar , Bilel Ben Amor , Imen Jaziri , Maha Changuel , Rihab Ben Dhia , Narjes Gouta , Jamel Saad , Rym Hadhri , Ahmed Zrig , Soumaya Boudokhane , Hela Marmouche , Mahbouba Jguirim , Nabil Sakly","doi":"10.1016/j.rmcr.2025.102216","DOIUrl":"10.1016/j.rmcr.2025.102216","url":null,"abstract":"<div><h3>Background</h3><div>Some studies have described a probable relationship between SARS-CoV-2 infection and autoimmunity.</div></div><div><h3>Aim</h3><div>to present a case series of autoimmune manifestations (AIM) following COVID-19 infection.</div></div><div><h3>Methods</h3><div>A consecutive series of patients from January 2020 to December 2023 was collected from the various departments of Fattouma Bourguiba University Hospital, Monastir, where all clinical assessments were performed. Anti-nuclear antibody (ANA) screening was performed using indirect immunofluorescence on HEp-2 cells (Euroimmun, Germany) with a positivity titer of ≥1/180. Typing was performed using ELISA (Biosystems, Spain) or line blot (Euroimmun, Germany). The assessment of other autoantibodies was performed using various techniques (indirect immunofluorescence, ELISA, and line blot).</div></div><div><h3>Results</h3><div>Sixteen patients presented with AIM after the COVID-19 infection. Their ages ranged from 12 to 67 years (44.6 ± 15.5 years). The sex ratio was 1 (eight men to eight women). Clinical manifestations began between one week and three months after infection. The clinical presentation was polymorphic (general, cutaneous, neurological, ophthalmic, muscular, articular, and abdominal features). Biological, radiological, and histopathological investigations revealed principal abnormalities in endocrine, articular, muscular, or neurological functions in the presence of ANA and/or specific autoantibodies. Fiveteen patients were diagnosed with autoimmune diseases (AID) and treated with specific treatments. The diagnosis retained for the other case was non-specific autoimmune stimulation with spontaneous recovery.</div></div><div><h3>Conclusion</h3><div>These cases suggest that AID or AIM can be triggered or unmasked by SARS-CoV-2 infection.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102216"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143895731","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":"Hemothorax resulting from hemorrhagic degeneration of giant uterine leiomyomata: A rare variant of pseudo-Meigs’ syndrome","authors":"Lawrence Huang , Tana Pradhan , Oleg Epelbaum","doi":"10.1016/j.rmcr.2025.102207","DOIUrl":"10.1016/j.rmcr.2025.102207","url":null,"abstract":"<div><div>Though rare, hemothorax can occur without trauma, coagulopathy, or apparent vascular injury. Similar to the proposed mechanism of Meigs’ syndrome or hepatic hydrothorax, small diaphragmatic defects may allow peritoneal fluid to traverse the diaphragm into the pleural space. While typically this is associated with transudative pleural effusions, there have been case reports of hemoperitoneum crossing the diaphragm into the pleural space. Here, we present a case in which multiple giant leiomyomata with hemorrhagic necrosis led to hemoperitoneum that translocated across the diaphragm into the pleural space, causing a hemothorax.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102207"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143899094","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":"Idiopathic dendriform pulmonary ossification diagnosed only after a surgical biopsy: A case report","authors":"Tomohito Takeshige , Ryo Koyama , Eisuke Goto , Tatsuo Maeyashiki , Akifumi Okajima , Toshihiko Nishioki , Junko Watanabe , Toshifumi Yae , Takamitsu Banno , Kenji Kido , Kazuhisa Takahashi","doi":"10.1016/j.rmcr.2025.102227","DOIUrl":"10.1016/j.rmcr.2025.102227","url":null,"abstract":"<div><div>Idiopathic dendriform pulmonary ossification (IDPO) presents as dendritic ossification of the lungs without a particular cause. IDPO is often diagnosed using surgical biopsy, transbronchial lung biopsy (TBLB), or transbronchial lung cryobiopsy (TBLC). In this case report, we described a 36-year-old male who presented with abnormal chest findings radiographically. TBLB was performed but no diagnosis was made. Instead, IDPO was diagnosed following a surgical biopsy. CT tomography and respiratory function tests showed no progression over the past 3.5 years. However, a nationwide survey in Japan revealed several cases of disease progression. Considering this, careful monitoring of the disease is recommended.</div></div>","PeriodicalId":51565,"journal":{"name":"Respiratory Medicine Case Reports","volume":"55 ","pages":"Article 102227"},"PeriodicalIF":0.8,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143902254","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}