Pub Date : 2026-01-05eCollection Date: 2026-01-01DOI: 10.1002/rcr2.70459
Sze Kye Teoh, Yen Shen Wong, Yiet Fai Ng, Yu Wei Cheah, Suhashini Ganapaty, Saiful Safuan Md Sani
Sarcomatoid carcinoma is a rare, aggressive lung cancer subtype. It typically presents as a pulmonary or pleural mass. Effusion-dominant disease without an identifiable primary mass is exceptionally uncommon. We report a 59-year-old man presenting with dyspnoea, chest pain, and fever. Imaging showed right pleural effusion without a lung mass. Thoracoscopy revealed necrotic pleural nodules. Histology demonstrated spindle-shaped atypical cells, positive for cytokeratin AE1/AE3 and weakly positive for TTF-1, but negative for mesothelial markers, consistent with sarcomatoid carcinoma of probable pulmonary origin. Staging CT revealed contralateral lung nodules, mediastinal lymphadenopathy, and distant metastases, but no dominant primary lesion. The patient declined systemic therapy and received palliative care. Sarcomatoid carcinoma presenting as malignant pleural effusion without a mass is rare. Early thoracoscopy and histological confirmation are crucial. Prognosis remains poor, though emerging data suggest a role for immunotherapy.
{"title":"Effusion-Dominant Pulmonary Sarcomatoid Carcinoma Without a Primary Mass: A Case Report.","authors":"Sze Kye Teoh, Yen Shen Wong, Yiet Fai Ng, Yu Wei Cheah, Suhashini Ganapaty, Saiful Safuan Md Sani","doi":"10.1002/rcr2.70459","DOIUrl":"10.1002/rcr2.70459","url":null,"abstract":"<p><p>Sarcomatoid carcinoma is a rare, aggressive lung cancer subtype. It typically presents as a pulmonary or pleural mass. Effusion-dominant disease without an identifiable primary mass is exceptionally uncommon. We report a 59-year-old man presenting with dyspnoea, chest pain, and fever. Imaging showed right pleural effusion without a lung mass. Thoracoscopy revealed necrotic pleural nodules. Histology demonstrated spindle-shaped atypical cells, positive for cytokeratin AE1/AE3 and weakly positive for TTF-1, but negative for mesothelial markers, consistent with sarcomatoid carcinoma of probable pulmonary origin. Staging CT revealed contralateral lung nodules, mediastinal lymphadenopathy, and distant metastases, but no dominant primary lesion. The patient declined systemic therapy and received palliative care. Sarcomatoid carcinoma presenting as malignant pleural effusion without a mass is rare. Early thoracoscopy and histological confirmation are crucial. Prognosis remains poor, though emerging data suggest a role for immunotherapy.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70459"},"PeriodicalIF":0.8,"publicationDate":"2026-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12771639/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918869","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}
A 37-year-old woman presented to the hospital with a history of a small amount of hemoptysis. Bronchoscopy revealed a polypoid lesion in the left main bronchus. Due to oozing bleeding from the lesion, biopsy was deferred to avoid fatal haemorrhage risk. Bronchial angiography revealed an abnormal tortuous branch with pulmonary artery shunting. Bronchial artery embolisation with coils was successfully performed without complications. Bronchial Dieulafoy's disease was diagnosed. Follow-up bronchoscopy approximately 4 months post-embolisation showed regression of the polypoid lesion, with residual scar formation. Subsequently, the hemoptysis resolved completely. Dieulafoy's disease is characterised by a large-calibre, submucosal artery lying just beneath the mucosa, predisposing to abrupt haemorrhage despite minimal or no mucosal defect. Although this disease has been classically described in the gastrointestinal tract, analogous lesions also occur in the bronchial tree. This case highlights the visual confirmation of lesion regression by bronchoscopy after bronchial artery embolisation.
{"title":"A Case of Bronchial Dieulafoy's Disease Presenting as an Endobronchial Polypoid Lesion That Regressed After Bronchial Artery Embolisation.","authors":"Masayoshi Higashiguchi, Yu Yamaguchi, Satoshi Tetsumoto, Hideo Ishikawa","doi":"10.1002/rcr2.70450","DOIUrl":"10.1002/rcr2.70450","url":null,"abstract":"<p><p>A 37-year-old woman presented to the hospital with a history of a small amount of hemoptysis. Bronchoscopy revealed a polypoid lesion in the left main bronchus. Due to oozing bleeding from the lesion, biopsy was deferred to avoid fatal haemorrhage risk. Bronchial angiography revealed an abnormal tortuous branch with pulmonary artery shunting. Bronchial artery embolisation with coils was successfully performed without complications. Bronchial Dieulafoy's disease was diagnosed. Follow-up bronchoscopy approximately 4 months post-embolisation showed regression of the polypoid lesion, with residual scar formation. Subsequently, the hemoptysis resolved completely. Dieulafoy's disease is characterised by a large-calibre, submucosal artery lying just beneath the mucosa, predisposing to abrupt haemorrhage despite minimal or no mucosal defect. Although this disease has been classically described in the gastrointestinal tract, analogous lesions also occur in the bronchial tree. This case highlights the visual confirmation of lesion regression by bronchoscopy after bronchial artery embolisation.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70450"},"PeriodicalIF":0.8,"publicationDate":"2026-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12771591/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918761","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}
Bordetella bronchiseptica is a zoonotic pathogen that can infect immunocompromised humans. We present a 59-year-old man on immunotherapy for inflammatory bowel disease, with persistent expectorant cough for 18 months. Computed tomography (CT) demonstrated migratory lung infiltrates. Bronchoalveolar lavage cultured B. bronchiseptica resistant to initial antimicrobial therapy, highlighting the diagnostic challenges in immunosuppressed patients.
{"title":"Chronic Productive Cough in an Immunosuppressed Patient due to Biologics: A Rare Case of <i>Bordetella bronchiseptica</i> Pulmonary Infection.","authors":"Debashree Banerjee, Eman Ayaz, Abby Hudson, Eamonn Trainor, Bavithra Vijayakumar, Harshana Bandara","doi":"10.1002/rcr2.70453","DOIUrl":"10.1002/rcr2.70453","url":null,"abstract":"<p><p><i>Bordetella bronchiseptica</i> is a zoonotic pathogen that can infect immunocompromised humans. We present a 59-year-old man on immunotherapy for inflammatory bowel disease, with persistent expectorant cough for 18 months. Computed tomography (CT) demonstrated migratory lung infiltrates. Bronchoalveolar lavage cultured <i>B. bronchiseptica</i> resistant to initial antimicrobial therapy, highlighting the diagnostic challenges in immunosuppressed patients.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70453"},"PeriodicalIF":0.8,"publicationDate":"2026-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12771581/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918911","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}
Endometrial carcinoma is the most common gynecologic malignancy in developed countries and is typically diagnosed at an early stage, with distant metastases uncommon at presentation. Pulmonary involvement occurs in less than 5% of cases and usually manifests as small nodules or interstitial disease; the appearance of multiple, large, round 'cannonball' metastases is exceptionally rare. We report the case of a 60-year-old woman who presented with cough and dyspnea. Imaging revealed numerous bilateral pulmonary 'cannonball' nodules, mediastinal lymphadenopathy, hepatic lesions, and an enlarged uterus. Endometrial biopsy confirmed endometrioid adenocarcinoma, staged as FIGO IV-B. The patient underwent chemotherapy with carboplatin and paclitaxel, resulting in significant regression of pulmonary and hepatic metastases and symptomatic improvement, followed by maintenance therapy with anastrozole that achieved stable disease on surveillance. This case highlights the importance of recognising cannonball pulmonary metastases as a potential, albeit rare, initial presentation of endometrial carcinoma, thereby guiding timely diagnosis and treatment.
{"title":"Cannonball Metastases of the Lung: An Unusual Initial Manifestation of Endometrial Carcinoma.","authors":"Midila Bapineni, Shivendra Tangutoori, Naga Vamsi Krishna Machineni, Kamlesh Sajnani, Maneesh Gaddam","doi":"10.1002/rcr2.70455","DOIUrl":"10.1002/rcr2.70455","url":null,"abstract":"<p><p>Endometrial carcinoma is the most common gynecologic malignancy in developed countries and is typically diagnosed at an early stage, with distant metastases uncommon at presentation. Pulmonary involvement occurs in less than 5% of cases and usually manifests as small nodules or interstitial disease; the appearance of multiple, large, round 'cannonball' metastases is exceptionally rare. We report the case of a 60-year-old woman who presented with cough and dyspnea. Imaging revealed numerous bilateral pulmonary 'cannonball' nodules, mediastinal lymphadenopathy, hepatic lesions, and an enlarged uterus. Endometrial biopsy confirmed endometrioid adenocarcinoma, staged as FIGO IV-B. The patient underwent chemotherapy with carboplatin and paclitaxel, resulting in significant regression of pulmonary and hepatic metastases and symptomatic improvement, followed by maintenance therapy with anastrozole that achieved stable disease on surveillance. This case highlights the importance of recognising cannonball pulmonary metastases as a potential, albeit rare, initial presentation of endometrial carcinoma, thereby guiding timely diagnosis and treatment.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70455"},"PeriodicalIF":0.8,"publicationDate":"2026-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12771667/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918936","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}
Community-acquired pneumonia (CAP) remains a leading cause of morbidity and mortality. Streptococcus pneumoniae is the most common causative pathogen, with urinary antigen testing aiding the diagnosis thereof; however, blood culture yields are low, and guideline recommendations vary. We herein report a case of a 77-year-old man with pneumococcal pneumonia confirmed by urinary antigen testing whose blood cultures also revealed Klebsiella variicola. The detection of this co-infection would have been missed without blood cultures. Moreover, this combination of pathogens is first reported. This case underscores the importance of routine blood cultures in CAP diagnosis and supports the Japanese Respiratory Society's recommendation for their use in hospitalised patients.
{"title":"Reconsidering the Role of Blood Cultures in Community-Acquired Pneumonia: A Case of Co-Infection With <i>Streptococcus pneumoniae</i> and <i>Klebsiella variicola</i>.","authors":"Naoki Fujimoto, Issei Oi, Kohei Fujita, Saiki Yoshimura, Takanori Ito, Takuma Imakita, Osamu Kanai, Kiminobu Tanizawa","doi":"10.1002/rcr2.70457","DOIUrl":"10.1002/rcr2.70457","url":null,"abstract":"<p><p>Community-acquired pneumonia (CAP) remains a leading cause of morbidity and mortality. <i>Streptococcus pneumoniae</i> is the most common causative pathogen, with urinary antigen testing aiding the diagnosis thereof; however, blood culture yields are low, and guideline recommendations vary. We herein report a case of a 77-year-old man with pneumococcal pneumonia confirmed by urinary antigen testing whose blood cultures also revealed <i>Klebsiella variicola</i>. The detection of this co-infection would have been missed without blood cultures. Moreover, this combination of pathogens is first reported. This case underscores the importance of routine blood cultures in CAP diagnosis and supports the Japanese Respiratory Society's recommendation for their use in hospitalised patients.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70457"},"PeriodicalIF":0.8,"publicationDate":"2026-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12771669/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918921","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}
Acute empyema typically occurs in elderly or immunocompromised individuals, and its development in healthy, non-elderly adults is uncommon. We report a case of acute empyema caused by Gemella morbillorum following COVID-19 infection in a previously healthy man in his mid-40s. He developed persistent fever and cough after COVID-19, and left pleural effusion was detected 3 weeks later. Despite meropenem therapy, his symptoms persisted, prompting referral to our institution. Imaging showed multiloculated pleural effusion compatible with stage II empyema, and video-assisted thoracoscopic surgery was performed. G. morbillorum was isolated from intraoperative specimens, and he recovered uneventfully. This case highlights that even healthy, non-elderly individuals may develop significant bacterial superinfection after COVID-19. Clinicians should therefore consider secondary bacterial infections, including empyema, when persistent or recurrent symptoms occur following COVID-19.
{"title":"Acute Empyema due to <i>Gemella morbillorum</i> Following COVID-19 in an Otherwise Healthy Middle-Aged Man.","authors":"Kazumasa Nanjo, Shoji Sakiyama, Naoki Furukawa, Hiroyuki Hino","doi":"10.1002/rcr2.70458","DOIUrl":"10.1002/rcr2.70458","url":null,"abstract":"<p><p>Acute empyema typically occurs in elderly or immunocompromised individuals, and its development in healthy, non-elderly adults is uncommon. We report a case of acute empyema caused by <i>Gemella morbillorum</i> following COVID-19 infection in a previously healthy man in his mid-40s. He developed persistent fever and cough after COVID-19, and left pleural effusion was detected 3 weeks later. Despite meropenem therapy, his symptoms persisted, prompting referral to our institution. Imaging showed multiloculated pleural effusion compatible with stage II empyema, and video-assisted thoracoscopic surgery was performed. <i>G. morbillorum</i> was isolated from intraoperative specimens, and he recovered uneventfully. This case highlights that even healthy, non-elderly individuals may develop significant bacterial superinfection after COVID-19. Clinicians should therefore consider secondary bacterial infections, including empyema, when persistent or recurrent symptoms occur following COVID-19.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70458"},"PeriodicalIF":0.8,"publicationDate":"2026-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12771600/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918774","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}
Pub Date : 2026-01-05eCollection Date: 2026-01-01DOI: 10.1002/rcr2.70456
Guo Li, Ming-Yan Jing, Min Pang
Pulmonary embolism may occur even in young adults with subtle risk factors such as prolonged immobility from mobile gaming. Hampton's hump on chest CT, though classically associated with infarction, remains a critical clue for diagnosis. Early recognition enables prompt anticoagulation and favorable outcomes, even in low-risk individuals.
{"title":"Hampton's Hump as a Radiologic Clue to Pulmonary Embolism in a Young Patient.","authors":"Guo Li, Ming-Yan Jing, Min Pang","doi":"10.1002/rcr2.70456","DOIUrl":"10.1002/rcr2.70456","url":null,"abstract":"<p><p>Pulmonary embolism may occur even in young adults with subtle risk factors such as prolonged immobility from mobile gaming. Hampton's hump on chest CT, though classically associated with infarction, remains a critical clue for diagnosis. Early recognition enables prompt anticoagulation and favorable outcomes, even in low-risk individuals.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70456"},"PeriodicalIF":0.8,"publicationDate":"2026-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12771589/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918917","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}
Pub Date : 2026-01-05eCollection Date: 2026-01-01DOI: 10.1002/rcr2.70449
Jacqueline Chung, Rory O'Donnell, Ayan Sabih
Necrotising sarcoid granulomatosis (NSG) is a rare granulomatous lung disease characterised by non-caseating granulomas, necrosis, and vasculitis. We describe a 50-year-old man who presented with constitutional symptoms and bilateral pulmonary masses initially suggestive of metastatic malignancy. Extensive infectious, malignant, and autoimmune workup was negative. Histopathology from lung biopsies and a video-assisted thoracoscopic surgery (VATS) resection revealed granulomatous inflammation with necrosis and no evidence of malignancy or infection. A trial of corticosteroids resulted in clinical, biochemical, and radiological improvement. This case highlights the diagnostic challenges of NSG and the importance of multidisciplinary evaluation to avoid misdiagnosis.
{"title":"A Rare Case of Necrotising Sarcoid Granulomatosis Presenting as Bilateral Pulmonary Masses Mimicking Malignancy.","authors":"Jacqueline Chung, Rory O'Donnell, Ayan Sabih","doi":"10.1002/rcr2.70449","DOIUrl":"10.1002/rcr2.70449","url":null,"abstract":"<p><p>Necrotising sarcoid granulomatosis (NSG) is a rare granulomatous lung disease characterised by non-caseating granulomas, necrosis, and vasculitis. We describe a 50-year-old man who presented with constitutional symptoms and bilateral pulmonary masses initially suggestive of metastatic malignancy. Extensive infectious, malignant, and autoimmune workup was negative. Histopathology from lung biopsies and a video-assisted thoracoscopic surgery (VATS) resection revealed granulomatous inflammation with necrosis and no evidence of malignancy or infection. A trial of corticosteroids resulted in clinical, biochemical, and radiological improvement. This case highlights the diagnostic challenges of NSG and the importance of multidisciplinary evaluation to avoid misdiagnosis.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70449"},"PeriodicalIF":0.8,"publicationDate":"2026-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12771671/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918843","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}
Pulmonary pleomorphic carcinoma (PPC) is a rare subtype of lung cancer that is characterised by rapid progression and poor prognosis. Hemothorax as the initial clinical presentation of PPC is exceptionally rare and, to the best of our knowledge, has not been previously reported. Here, we report a rare case of a KRAS G12D-mutated PPC penetrating the visceral pleura, leading to rapid tumour growth and uncontrolled hemothorax. In addition to the high proliferative and invasive potential of the tumour, the oncological properties associated with the KRAS G12D mutation likely precipitated both the abrupt onset and recurrence of massive hemothorax. Early recognition, complete macroscopic resection to achieve definitive haemostasis, and prompt initiation of postoperative therapy are essential to improve clinical outcomes.
{"title":"Hemothorax as the Initial Manifestation of <i>KRAS G12D</i> Positive Pulmonary Pleomorphic Carcinoma: A Case Report.","authors":"Takuma Ikeda, Hirotaka Matsumoto, Shigenari Iwagaki, Ryo Ogawa, Yushi Shimamura, Emiko Saito, Takehisa Fukada, Hiroaki Sakai","doi":"10.1002/rcr2.70454","DOIUrl":"10.1002/rcr2.70454","url":null,"abstract":"<p><p>Pulmonary pleomorphic carcinoma (PPC) is a rare subtype of lung cancer that is characterised by rapid progression and poor prognosis. Hemothorax as the initial clinical presentation of PPC is exceptionally rare and, to the best of our knowledge, has not been previously reported. Here, we report a rare case of a KRAS G12D-mutated PPC penetrating the visceral pleura, leading to rapid tumour growth and uncontrolled hemothorax. In addition to the high proliferative and invasive potential of the tumour, the oncological properties associated with the KRAS G12D mutation likely precipitated both the abrupt onset and recurrence of massive hemothorax. Early recognition, complete macroscopic resection to achieve definitive haemostasis, and prompt initiation of postoperative therapy are essential to improve clinical outcomes.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70454"},"PeriodicalIF":0.8,"publicationDate":"2026-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12771678/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918882","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}
Pub Date : 2026-01-04eCollection Date: 2026-01-01DOI: 10.1002/rcr2.70423
Michael Xie, Ronan O'Donnabhain, Zemin Cao, Jazmin Eckhaus, Phillip Parente, Alan Wong, David Holcdorf, Victor Duong
Doege-Potter Syndrome (DPS) is a rare paraneoplastic cause of hypoglycaemia, arising from solitary fibrous tumours (SFTs) that ectopically secrete insulin-like growth factor II (IGF-II). We report a case of DPS in a 76-year-old man presenting with recurrent severe hypoglycaemia. Biochemistry investigations revealed hypoglycaemia with suppressed insulin, C-peptide and beta-hydroxybutyrate levels. Magnetic resonance imaging (MRI) confirmed a giant (205 × 190 × 169 mm) right pleurally-based SFT. Hypoglycaemia was managed with intravenous dextrose infusion and glucagon until surgical resection via thoracotomy. A massive transfusion protocol was activated for intra-operative bleeding. Histopathology confirmed a spindle cell SFT without malignant features. Post-operative blood glucose levels normalised, and the patient was discharged without complication. This case highlights the diagnostic challenges of DPS, the potential to maintain pre-operative normoglycaemia without glucocorticoid therapy and the importance of recognising tumour vascularity to guide pre-operative planning and reduce bleeding risk.
{"title":"From Hypoglycaemia to Haemorrhage: A Case of Giant Pleural Solitary Fibrous Tumour With Doege-Potter Syndrome.","authors":"Michael Xie, Ronan O'Donnabhain, Zemin Cao, Jazmin Eckhaus, Phillip Parente, Alan Wong, David Holcdorf, Victor Duong","doi":"10.1002/rcr2.70423","DOIUrl":"10.1002/rcr2.70423","url":null,"abstract":"<p><p>Doege-Potter Syndrome (DPS) is a rare paraneoplastic cause of hypoglycaemia, arising from solitary fibrous tumours (SFTs) that ectopically secrete insulin-like growth factor II (IGF-II). We report a case of DPS in a 76-year-old man presenting with recurrent severe hypoglycaemia. Biochemistry investigations revealed hypoglycaemia with suppressed insulin, C-peptide and beta-hydroxybutyrate levels. Magnetic resonance imaging (MRI) confirmed a giant (205 × 190 × 169 mm) right pleurally-based SFT. Hypoglycaemia was managed with intravenous dextrose infusion and glucagon until surgical resection via thoracotomy. A massive transfusion protocol was activated for intra-operative bleeding. Histopathology confirmed a spindle cell SFT without malignant features. Post-operative blood glucose levels normalised, and the patient was discharged without complication. This case highlights the diagnostic challenges of DPS, the potential to maintain pre-operative normoglycaemia without glucocorticoid therapy and the importance of recognising tumour vascularity to guide pre-operative planning and reduce bleeding risk.</p>","PeriodicalId":45846,"journal":{"name":"Respirology Case Reports","volume":"14 1","pages":"e70423"},"PeriodicalIF":0.8,"publicationDate":"2026-01-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12765591/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145906788","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}