Clara Long, Abdulrahman Al-Abdulmalek, Jonathan Lai, David G. Haegert, S. Isnard, Denis Cournoyer, J. Routy
Background: Autoimmune diseases are not contraindications for immune checkpoint inhibitors (ICI) therapy in patients with cancer. However, immune-related adverse events (irAEs) are frequently observed in patients receiving ICIs including dermatitis, thyroiditis, colitis, and pneumonitis. Thrombocytopenic purpura, aplasia, and haemophagocytic lymphohistiocytosis (HLH) are rarely observed during ICIs. Case description: We report the case of a male patient with pre-existing untreated HLA B27 and ankylosing spondylitis with gastric cancer and liver metastases. The 79-year-old man was treated with anti-HER2 trastuzumab and anti-PD-1 nivolumab. Seventeen days after the seventh cycle of treatment, he presented at the emergency department with acute fever, confusion, and hypotension. Laboratory results showed pancytopenia, and elevation of ferritin and triglyceride. No infections were detected. Although not seen in a bone marrow biopsy, clinical presentation, and absence of infection, together with an H-score of 263, indicated HLH. The patient was treated with dexamethasone for four days and discharged on a tapering dose of steroids. At the two-month follow-up, clinical presentation was normal and blood test almost normalised. At 8 months, no liver metastases were observed. Conclusions: In a patient with a pre-existing autoimmune condition, immunotherapy led to the development of HLH, which was controlled by glucocorticoid. Absence of the feature of haemophagocytosis in the bone marrow biopsy did not exclude the diagnosis, as HLH can occur in the spleen or in the liver. Glucocorticoid therapy did not prevent the anti-cancer effect of ICIs, and liver metastases disappeared 8 months post-HLH. This case warrants further research on the interplay between autoimmunity and ICI response, as well as ICI-induced irAEs.
{"title":"Haemophagocytic lymphohistiocytosis following the anti-PD-1 nivolumab in a patient with gastric cancer and ankylosing spondylitis","authors":"Clara Long, Abdulrahman Al-Abdulmalek, Jonathan Lai, David G. Haegert, S. Isnard, Denis Cournoyer, J. Routy","doi":"10.12890/2024_004370","DOIUrl":"https://doi.org/10.12890/2024_004370","url":null,"abstract":"Background: Autoimmune diseases are not contraindications for immune checkpoint inhibitors (ICI) therapy in patients with cancer. However, immune-related adverse events (irAEs) are frequently observed in patients receiving ICIs including dermatitis, thyroiditis, colitis, and pneumonitis. Thrombocytopenic purpura, aplasia, and haemophagocytic lymphohistiocytosis (HLH) are rarely observed during ICIs. Case description: We report the case of a male patient with pre-existing untreated HLA B27 and ankylosing spondylitis with gastric cancer and liver metastases. The 79-year-old man was treated with anti-HER2 trastuzumab and anti-PD-1 nivolumab. Seventeen days after the seventh cycle of treatment, he presented at the emergency department with acute fever, confusion, and hypotension. Laboratory results showed pancytopenia, and elevation of ferritin and triglyceride. No infections were detected. Although not seen in a bone marrow biopsy, clinical presentation, and absence of infection, together with an H-score of 263, indicated HLH. The patient was treated with dexamethasone for four days and discharged on a tapering dose of steroids. At the two-month follow-up, clinical presentation was normal and blood test almost normalised. At 8 months, no liver metastases were observed. Conclusions: In a patient with a pre-existing autoimmune condition, immunotherapy led to the development of HLH, which was controlled by glucocorticoid. Absence of the feature of haemophagocytosis in the bone marrow biopsy did not exclude the diagnosis, as HLH can occur in the spleen or in the liver. Glucocorticoid therapy did not prevent the anti-cancer effect of ICIs, and liver metastases disappeared 8 months post-HLH. This case warrants further research on the interplay between autoimmunity and ICI response, as well as ICI-induced irAEs.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":"9 7","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140372253","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}
Pepijn van Gastel, Jildou N. Dijkstra, Mina A. Jacob, Jeroen A. Schouten, Bram Kok
Introduction: Sudden onset of reduced consciousness, psychomotor agitation and mydriasis are all indicative of an anticholinergic toxidrome. It is important to note that numerous drugs, as well as certain herbs and plants, possess anticholinergic properties. Case description: An 84-year-old female patient had sudden nocturnal onset of uncoordinated hand movements and altered mental status. Shortly after, the patient’s 83-year-old husband developed symptoms of dysarthria, gait ataxia, vertigo, and delirium. Conclusion: Anticholinergic syndrome consists of a combination of central and peripheral anticholinergic symptoms. Physostigmine given intravenously resulted in rapid reversal of symptoms. Thorn apple seeds had been accidentally ingested and were identified as the cause.
{"title":"Anticholinergic toxidrome due to thorn apple seed ingestion in an elderly couple","authors":"Pepijn van Gastel, Jildou N. Dijkstra, Mina A. Jacob, Jeroen A. Schouten, Bram Kok","doi":"10.12890/2024_004381","DOIUrl":"https://doi.org/10.12890/2024_004381","url":null,"abstract":"Introduction: Sudden onset of reduced consciousness, psychomotor agitation and mydriasis are all indicative of an anticholinergic toxidrome. It is important to note that numerous drugs, as well as certain herbs and plants, possess anticholinergic properties. Case description: An 84-year-old female patient had sudden nocturnal onset of uncoordinated hand movements and altered mental status. Shortly after, the patient’s 83-year-old husband developed symptoms of dysarthria, gait ataxia, vertigo, and delirium. Conclusion: Anticholinergic syndrome consists of a combination of central and peripheral anticholinergic symptoms. Physostigmine given intravenously resulted in rapid reversal of symptoms. Thorn apple seeds had been accidentally ingested and were identified as the cause.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":"18 17","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140373086","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}
Novena Lin Sing Cheng, Chung Wai Wong, Wen Ming Yu, Kim Hung Tsang
Introduction: Rectus sheath haematoma (RSH) has become increasingly common but is often underdiagnosed. Prompt diagnosis will avoid unnecessary investigations and procedures, resulting in early treatment and a better outcome. Case description: We described a case of a spontaneous RSH with intraperitoneal extension and formation of a vesico-haematoma fistula, which was initially misdiagnosed as a urinary tract infection. The diagnosis was made ten days after admission, when a CT scan showed an over-16 cm RSH with intraperitoneal extension, bladder perforation and a vesico-haematoma fistula. The patient was managed conservatively. Discussion: RSH accounts for less than 2% of acute abdomen cases and is often unrecognised. Its presentation can mimic other intra-abdominal pathologies, and the diagnosis is often delayed or missed. Complications can arise from an RSH although it is generally viewed as a self-limiting condition. Conclusion: RSH has become increasingly common, and we would like to highlight the need to include abdominal wall pathologies in the initial differential diagnoses of acute abdomen to avoid delay in diagnosis.
{"title":"Bladder perforation and vesico-haematoma fistula: an uncommon complication of rectus sheath haematoma","authors":"Novena Lin Sing Cheng, Chung Wai Wong, Wen Ming Yu, Kim Hung Tsang","doi":"10.12890/2024_004362","DOIUrl":"https://doi.org/10.12890/2024_004362","url":null,"abstract":"Introduction: Rectus sheath haematoma (RSH) has become increasingly common but is often underdiagnosed. Prompt diagnosis will avoid unnecessary investigations and procedures, resulting in early treatment and a better outcome. Case description: We described a case of a spontaneous RSH with intraperitoneal extension and formation of a vesico-haematoma fistula, which was initially misdiagnosed as a urinary tract infection. The diagnosis was made ten days after admission, when a CT scan showed an over-16 cm RSH with intraperitoneal extension, bladder perforation and a vesico-haematoma fistula. The patient was managed conservatively. Discussion: RSH accounts for less than 2% of acute abdomen cases and is often unrecognised. Its presentation can mimic other intra-abdominal pathologies, and the diagnosis is often delayed or missed. Complications can arise from an RSH although it is generally viewed as a self-limiting condition. Conclusion: RSH has become increasingly common, and we would like to highlight the need to include abdominal wall pathologies in the initial differential diagnoses of acute abdomen to avoid delay in diagnosis.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":" 11","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-03-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140382109","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}
Background: This study presents a patient diagnosed with tricuspid valvular stenosis due to right ventricular lymphoma, who was treated successfully. Case presentation: A 66-year-old man with a history of worsening shortness of breath during activity for the last three weeks sought medical attention. The patient later experienced swelling in the extremities, fluid build-up around the lungs and abdominal fluid accumulation, with no reported chest pain, fever, or weight loss. An echocardiogram found a mass in the lateral wall near the tricuspid valve of the right ventricle, leading to moderate tricuspid stenosis. The cardiac magnetic resonance imaging (MRI) revealed a lumpy, poorly defined mass that invaded the heart muscle and displayed varied enhancement after contrast administration. Suspicion arose for a malignant tumour or metastatic lesion due to its features and contrast uptake capability. A percutaneous biopsy was carried out on the mass in the right ventricle to confirm the diagnosis. The pathology report indicated a diagnosis of non-Hodgkin’s lymphoma. After being diagnosed, the patient underwent chemotherapy using the R-CHOP regimen. Over time the symptoms improved, and echocardiograms revealed a decrease in the size of the tumour. After undergoing six rounds of chemotherapy, a cardiac MRI four months later showed no signs of a tumour. After that, the patient resumed their regular activities. Conclusion: Right ventricular tumours are mostly malignant lesions and often have an inferior prognosis. Early diagnosis with imaging techniques and myocardial biopsy is necessary to deliver life-saving treatment quickly.
{"title":"Successful chemotherapy treatment for a tricuspid valvular stenosis patient due to right ventricular lymphoma","authors":"Do Van Chien, Tran Quoc Quy","doi":"10.12890/2024_004451","DOIUrl":"https://doi.org/10.12890/2024_004451","url":null,"abstract":"Background: This study presents a patient diagnosed with tricuspid valvular stenosis due to right ventricular lymphoma, who was treated successfully. Case presentation: A 66-year-old man with a history of worsening shortness of breath during activity for the last three weeks sought medical attention. The patient later experienced swelling in the extremities, fluid build-up around the lungs and abdominal fluid accumulation, with no reported chest pain, fever, or weight loss. An echocardiogram found a mass in the lateral wall near the tricuspid valve of the right ventricle, leading to moderate tricuspid stenosis. The cardiac magnetic resonance imaging (MRI) revealed a lumpy, poorly defined mass that invaded the heart muscle and displayed varied enhancement after contrast administration. Suspicion arose for a malignant tumour or metastatic lesion due to its features and contrast uptake capability. A percutaneous biopsy was carried out on the mass in the right ventricle to confirm the diagnosis. The pathology report indicated a diagnosis of non-Hodgkin’s lymphoma. After being diagnosed, the patient underwent chemotherapy using the R-CHOP regimen. Over time the symptoms improved, and echocardiograms revealed a decrease in the size of the tumour. After undergoing six rounds of chemotherapy, a cardiac MRI four months later showed no signs of a tumour. After that, the patient resumed their regular activities. Conclusion: Right ventricular tumours are mostly malignant lesions and often have an inferior prognosis. Early diagnosis with imaging techniques and myocardial biopsy is necessary to deliver life-saving treatment quickly.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":"3 ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-03-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140383607","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}
Tariq Siddiqui, G. Strandvik, A. El-Menyar, Sandro Rizoli, H. Al-Thani
We present a 30-year-old male who sustained a mild traumatic brain injury and then was intubated due to deterioration of consciousness. A head CT scan revealed mild brain oedema, a fractured nasal bone and mild left thoracic wall haematoma. Despite complete clinical and radiological normalisation within 36 hours, he failed to wean off the ventilator. The patient was found to have subtle bulbar manifestations including dysphonia, dysarthria, and dysphagia, with recurrent left lung collapse. He responded to an empirical pyridostigmine trial despite negative biochemical tests for myasthenia gravis (MG). The patient was weaned successfully from the ventilator, transferred to a long-term care facility, and then discharged home. Classic symptoms and signs of a disease may be absent, but the presence of dysarthria, dysphagia, transient vocal cord palsy, nasal speech, absent gag reflex and respiratory failure in difficult-to-wean patients, with no definitive diagnosis, may warrant an empirical trial of therapy for suspected MG and for the benefit of any doubt.
{"title":"Empirical pyridostigmine in a patient with difficult weaning from mechanical ventilation after traumatic brain injury","authors":"Tariq Siddiqui, G. Strandvik, A. El-Menyar, Sandro Rizoli, H. Al-Thani","doi":"10.12890/2024_004363","DOIUrl":"https://doi.org/10.12890/2024_004363","url":null,"abstract":"We present a 30-year-old male who sustained a mild traumatic brain injury and then was intubated due to deterioration of consciousness. A head CT scan revealed mild brain oedema, a fractured nasal bone and mild left thoracic wall haematoma. Despite complete clinical and radiological normalisation within 36 hours, he failed to wean off the ventilator. The patient was found to have subtle bulbar manifestations including dysphonia, dysarthria, and dysphagia, with recurrent left lung collapse. He responded to an empirical pyridostigmine trial despite negative biochemical tests for myasthenia gravis (MG). The patient was weaned successfully from the ventilator, transferred to a long-term care facility, and then discharged home. Classic symptoms and signs of a disease may be absent, but the presence of dysarthria, dysphagia, transient vocal cord palsy, nasal speech, absent gag reflex and respiratory failure in difficult-to-wean patients, with no definitive diagnosis, may warrant an empirical trial of therapy for suspected MG and for the benefit of any doubt.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":" January","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-03-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140383397","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}
Pleuroperitoneal leak as a cause of pleural effusions in peritoneal dialysis is a rare but important complication to consider in continuous ambulatory peritoneal dialysis (CAPD) patients presenting with recurrent progressive dyspnoea. Generally, these effusions are unilateral and right-sided, resulting in shortness of breath and reduced ultrafiltration volume, which are initially managed by peritoneal rest. We describe a case of bilateral pleural effusions in a 57-year-old female on chronic CAPD who developed recurrent progressive dyspnoea but maintained adequate dialysis output. A chest radiograph revealed bilateral pleural effusions with high glucose content, and scintigraphy confirmed the existence of a definite pleuroperitoneal communication. She was managed by temporary substitution to haemodialysis, followed by suturing of the shunt and successful video-assisted thoracoscopic surgery (VATS) pleurodesis with an aldehyde-based surgical glue. Unexplained recurring dyspnoea in chronic CAPD should raise the suspicion of a possible pleuroperitoneal leak, even in patients without an apparent loss of ultrafiltration. Pleurodesis using an aldehyde-based adhesive was effective and tolerated well by our patient and may be considered in managing cases of recurrent pleural effusion.
{"title":"Bilateral pleural effusion in continuous ambulatory peritoneal dialysis managed by VATS pleurodesis","authors":"J. Jonny, Laurencia Violetta","doi":"10.12890/2024_004343","DOIUrl":"https://doi.org/10.12890/2024_004343","url":null,"abstract":"Pleuroperitoneal leak as a cause of pleural effusions in peritoneal dialysis is a rare but important complication to consider in continuous ambulatory peritoneal dialysis (CAPD) patients presenting with recurrent progressive dyspnoea. Generally, these effusions are unilateral and right-sided, resulting in shortness of breath and reduced ultrafiltration volume, which are initially managed by peritoneal rest. We describe a case of bilateral pleural effusions in a 57-year-old female on chronic CAPD who developed recurrent progressive dyspnoea but maintained adequate dialysis output. A chest radiograph revealed bilateral pleural effusions with high glucose content, and scintigraphy confirmed the existence of a definite pleuroperitoneal communication. She was managed by temporary substitution to haemodialysis, followed by suturing of the shunt and successful video-assisted thoracoscopic surgery (VATS) pleurodesis with an aldehyde-based surgical glue. Unexplained recurring dyspnoea in chronic CAPD should raise the suspicion of a possible pleuroperitoneal leak, even in patients without an apparent loss of ultrafiltration. Pleurodesis using an aldehyde-based adhesive was effective and tolerated well by our patient and may be considered in managing cases of recurrent pleural effusion.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":" 26","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-03-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140387907","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}
Muhammad Umer Riaz Gondal, John Lemoine, Jared Segal, Zainab Kiyani, Muhammad Ibraiz Bilal, F. Ansari, Brian McCauley
Introduction: Combination-based adjuvant chemotherapy utilising capecitabine and oxaliplatin is widely used in gastric cancer treatment. Rare but severe cardiac events such as prolonged QT, cardiac arrest and cardiogenic shock can result from their use. Case description: A 45-year-old female with gastric adenocarcinoma was started on capecitabine-oxaliplatin chemotherapy one week before presenting to the emergency department with weakness. Blood pressure was 78/56 mmHg, heart rate 140 bpm and oxygen saturation 85%. She became unresponsive with pulseless ventricular fibrillation; CPR was initiated with immediate intubation. She received two shocks with a return of spontaneous circulation. Laboratory tests revealed serum potassium (3.1 mmol/l), magnesium (1.1 mg/dl) and troponin (0.46 ng/ml). An EKG revealed sinus tachycardia with a prolonged QT interval (556 ms). The combined effects of capecitabine, oxaliplatin and electrolyte abnormalities likely contributed to the QT prolongation. An echocardiogram demonstrated an ejection fraction of 10%–15%. An emergent right-heart catheterisation showed right atrial pressure of 10 mmHg and pulmonary artery pressure of 30/18 mmHg; cardiac output and index were not recorded. An intra-aortic balloon pump was placed, and she was admitted to the ICU for cardiogenic shock requiring norepinephrine, vasopressin and dobutamine. A repeat echocardiogram showed a significantly improved ejection fraction of 65%, and she was discharged. Discussion: Capecitabine and oxaliplatin cardiotoxicity is an exceedingly rare occurrence, with both drugs reported to cause QT prolongation. Conclusion: Healthcare providers must recognise the QT prolongation effects of capecitabine and oxaliplatin, leading to life-threatening cardiac arrhythmias.
{"title":"Cardiotoxicity induced by capecitabine and oxaliplatin in gastric cancer treatment: a rare case of cardiac arrest and cardiogenic shock","authors":"Muhammad Umer Riaz Gondal, John Lemoine, Jared Segal, Zainab Kiyani, Muhammad Ibraiz Bilal, F. Ansari, Brian McCauley","doi":"10.12890/2024_004417","DOIUrl":"https://doi.org/10.12890/2024_004417","url":null,"abstract":"Introduction: Combination-based adjuvant chemotherapy utilising capecitabine and oxaliplatin is widely used in gastric cancer treatment. Rare but severe cardiac events such as prolonged QT, cardiac arrest and cardiogenic shock can result from their use. Case description: A 45-year-old female with gastric adenocarcinoma was started on capecitabine-oxaliplatin chemotherapy one week before presenting to the emergency department with weakness. Blood pressure was 78/56 mmHg, heart rate 140 bpm and oxygen saturation 85%. She became unresponsive with pulseless ventricular fibrillation; CPR was initiated with immediate intubation. She received two shocks with a return of spontaneous circulation. Laboratory tests revealed serum potassium (3.1 mmol/l), magnesium (1.1 mg/dl) and troponin (0.46 ng/ml). An EKG revealed sinus tachycardia with a prolonged QT interval (556 ms). The combined effects of capecitabine, oxaliplatin and electrolyte abnormalities likely contributed to the QT prolongation. An echocardiogram demonstrated an ejection fraction of 10%–15%. An emergent right-heart catheterisation showed right atrial pressure of 10 mmHg and pulmonary artery pressure of 30/18 mmHg; cardiac output and index were not recorded. An intra-aortic balloon pump was placed, and she was admitted to the ICU for cardiogenic shock requiring norepinephrine, vasopressin and dobutamine. A repeat echocardiogram showed a significantly improved ejection fraction of 65%, and she was discharged. Discussion: Capecitabine and oxaliplatin cardiotoxicity is an exceedingly rare occurrence, with both drugs reported to cause QT prolongation. Conclusion: Healthcare providers must recognise the QT prolongation effects of capecitabine and oxaliplatin, leading to life-threatening cardiac arrhythmias.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":" 30","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-03-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140387923","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}
A. Faraone, Alberto Fortini, Vanni Borgioli, C. Cappugi, Aldo Lo Forte, Valeria Maria Bottaro, Augusto Vaglio
Background: eosinophilic granulomatosis with polyangiitis (EGPA) is a rare multisystem inflammatory disease characterized by asthma, eosinophilia and granulomatous or vasculitic involvement of various organs. While the eye is uncommonly affected in patients with EGPA, multiple ophthalmic manifestations have been reported, which can result in serious visual impairment without timely treatment. Case report: we report the case of a 79-year-old woman with a history of asthma and nasal polyps who presented with low-grade fever, mild alteration of mental status, and fatigue. Chest X-ray revealed bilateral interstitial infiltrates. Lab tests showed elevated C-reactive protein level and eosinophilia (eosinophil count, 4.6 x109 cells/l); blood cultures and parasitological examination of stools tested negative. Four days after presentation, the patient reported sudden and severe blurring of vision in her left eye. Ophthalmological examination revealed bilateral swollen optic disc and visual field loss, more severe in the left eye. A diagnosis of EGPA complicated by arteritic anterior ischaemic optic neuropathy (A-AION) was proposed, while an alternative or concurrent diagnosis of giant cell arteritis was ruled out based on clinical picture. Immunosuppressive treatment with high-dose intravenous glucocorticoids was promptly started. The patient’s visual defect did not improve; however, two months later, no worsening was registered on ophthalmic reassessment. Conclusions: A-AION is an infrequent but severe manifestation of EGPA, requiring prompt diagnosis and emergency-level glucocorticoid therapy to prevent any further vision loss. Disease awareness and a multidisciplinary approach are crucial to expedite diagnostic work-up and effective management of EGPA-related ocular complications.
背景：嗜酸性粒细胞肉芽肿伴多血管炎（EGPA）是一种罕见的多系统炎症性疾病，其特征是哮喘、嗜酸性粒细胞增多、肉芽肿或血管炎累及多个器官。虽然眼部受累在 EGPA 患者中并不常见，但也有报道称眼部出现多种表现，如不及时治疗可导致严重的视力损害。病例报告：我们报告了一例 79 岁女性患者的病例，她有哮喘和鼻息肉病史，曾出现低热、轻度精神状态改变和乏力。胸部 X 光片显示双侧间质浸润。实验室检查显示 C 反应蛋白水平升高，嗜酸性粒细胞增多（嗜酸性粒细胞计数为 4.6 x109 cells/l）；血液培养和粪便寄生虫检查均呈阴性。就诊四天后，患者报告左眼视力突然严重模糊。眼科检查发现双侧视盘肿胀，视野缺损，左眼更为严重。诊断结果为 EGPA 并发动脉炎性前部缺血性视神经病变（A-AION），同时根据临床表现排除了巨细胞动脉炎的替代或并发诊断。医生立即开始使用大剂量糖皮质激素进行免疫抑制治疗。患者的视力缺陷没有得到改善；然而，两个月后，眼科复查结果显示患者的视力没有恶化。结论：A-AION是EGPA的一种少见但严重的表现形式，需要及时诊断并给予紧急糖皮质激素治疗，以防止视力进一步下降。疾病意识和多学科方法对于加快诊断工作和有效处理与EGPA相关的眼部并发症至关重要。
{"title":"Sudden visual loss due to arteritic anterior ischaemic optic neuropathy: A rare manifestation of eosinophilic granulomatosis with polyangiitis","authors":"A. Faraone, Alberto Fortini, Vanni Borgioli, C. Cappugi, Aldo Lo Forte, Valeria Maria Bottaro, Augusto Vaglio","doi":"10.12890/2024_004345","DOIUrl":"https://doi.org/10.12890/2024_004345","url":null,"abstract":"Background: eosinophilic granulomatosis with polyangiitis (EGPA) is a rare multisystem inflammatory disease characterized by asthma, eosinophilia and granulomatous or vasculitic involvement of various organs. While the eye is uncommonly affected in patients with EGPA, multiple ophthalmic manifestations have been reported, which can result in serious visual impairment without timely treatment. Case report: we report the case of a 79-year-old woman with a history of asthma and nasal polyps who presented with low-grade fever, mild alteration of mental status, and fatigue. Chest X-ray revealed bilateral interstitial infiltrates. Lab tests showed elevated C-reactive protein level and eosinophilia (eosinophil count, 4.6 x109 cells/l); blood cultures and parasitological examination of stools tested negative. Four days after presentation, the patient reported sudden and severe blurring of vision in her left eye. Ophthalmological examination revealed bilateral swollen optic disc and visual field loss, more severe in the left eye. A diagnosis of EGPA complicated by arteritic anterior ischaemic optic neuropathy (A-AION) was proposed, while an alternative or concurrent diagnosis of giant cell arteritis was ruled out based on clinical picture. Immunosuppressive treatment with high-dose intravenous glucocorticoids was promptly started. The patient’s visual defect did not improve; however, two months later, no worsening was registered on ophthalmic reassessment. Conclusions: A-AION is an infrequent but severe manifestation of EGPA, requiring prompt diagnosis and emergency-level glucocorticoid therapy to prevent any further vision loss. Disease awareness and a multidisciplinary approach are crucial to expedite diagnostic work-up and effective management of EGPA-related ocular complications.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":" 41","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-03-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140388087","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}
A. Nasrullah, Muhammad Ibraiz Bilal, Khalid Malik, Briana E Disilvio, Tariq Cheema
A 52-year-old female with a history of chronic lymphoedema and untreated deep vein thrombosis, presented with non-specific right-sided chest pain. A CT angiogram confirmed bilateral inferior pulmonary vein thromboses (PVT). A comprehensive hypercoagulable workup and age-appropriate cancer screening were unremarkable; the lack of associated risk factors confirmed idiopathic PVT. The management strategy of systemic anticoagulation with apixaban and multidisciplinary follow-up underscores the treatment challenges of rare presentations. This case accentuates the importance of considering PVT in differential diagnoses of atypical chest pain and contributes valuable insights into the diagnosis, understanding and management of this uncommon condition.
{"title":"Idiopathic bilateral pulmonary vein thrombosis","authors":"A. Nasrullah, Muhammad Ibraiz Bilal, Khalid Malik, Briana E Disilvio, Tariq Cheema","doi":"10.12890/2024_004291","DOIUrl":"https://doi.org/10.12890/2024_004291","url":null,"abstract":"A 52-year-old female with a history of chronic lymphoedema and untreated deep vein thrombosis, presented with non-specific right-sided chest pain. A CT angiogram confirmed bilateral inferior pulmonary vein thromboses (PVT). A comprehensive hypercoagulable workup and age-appropriate cancer screening were unremarkable; the lack of associated risk factors confirmed idiopathic PVT. The management strategy of systemic anticoagulation with apixaban and multidisciplinary follow-up underscores the treatment challenges of rare presentations. This case accentuates the importance of considering PVT in differential diagnoses of atypical chest pain and contributes valuable insights into the diagnosis, understanding and management of this uncommon condition.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":"10 7","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-01-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139594315","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}
Mutsuka Kurihara, Yasutaka Yanagita, D. Yokokawa, Yu Li, M. Ikusaka
Kikuchi-Fujimoto disease (KFD), also called histiocytic necrotizing lymphadenitis, is more common in young women and typically presents with small, painful, localized cervical lymphadenopathy that resolves spontaneously within a few weeks. Laboratory findings are variable. As many as 40% of KFD cases are reported to be painless, and up to 22% to be generalized lymphadenopathy. Therefore, malignant lymphoma could be a differential diagnosis of KFD. A histopathologic diagnosis is needed when it is difficult to distinguish KFD from lymphoma. KFD typically shows small, highly accumulated cervical lymph nodes on fluorodeoxyglucose positron emission tomography (FDG-PET). This contrasts with malignant lymphoma, which tends to be associated with massive lymphadenopathy. In our case, a 40-year-old Japanese male presented with painless lumps in the right neck, accompanied by fever, night sweats, and loss of appetite. His symptoms and laboratory results worsened over a month. FDG-PET revealed highly accumulated uptake in cervical, mediastinal, and axillary lymph nodes. The PET imaging showed a small, high FDG uptake and contributed to the correct diagnosis of KFD. This case report highlights the importance of FDG-PET, which is a valuable diagnostic tool for KFD as it typically differentiates large clusters of small lymph nodes typical of KFD from normal lymph nodes.
{"title":"Atypical Kikuchi-Fujimoto disease: FDG-PET contribution to the diagnosis","authors":"Mutsuka Kurihara, Yasutaka Yanagita, D. Yokokawa, Yu Li, M. Ikusaka","doi":"10.12890/2024_004258","DOIUrl":"https://doi.org/10.12890/2024_004258","url":null,"abstract":"Kikuchi-Fujimoto disease (KFD), also called histiocytic necrotizing lymphadenitis, is more common in young women and typically presents with small, painful, localized cervical lymphadenopathy that resolves spontaneously within a few weeks. Laboratory findings are variable. As many as 40% of KFD cases are reported to be painless, and up to 22% to be generalized lymphadenopathy. Therefore, malignant lymphoma could be a differential diagnosis of KFD. A histopathologic diagnosis is needed when it is difficult to distinguish KFD from lymphoma. KFD typically shows small, highly accumulated cervical lymph nodes on fluorodeoxyglucose positron emission tomography (FDG-PET). This contrasts with malignant lymphoma, which tends to be associated with massive lymphadenopathy. In our case, a 40-year-old Japanese male presented with painless lumps in the right neck, accompanied by fever, night sweats, and loss of appetite. His symptoms and laboratory results worsened over a month. FDG-PET revealed highly accumulated uptake in cervical, mediastinal, and axillary lymph nodes. The PET imaging showed a small, high FDG uptake and contributed to the correct diagnosis of KFD. This case report highlights the importance of FDG-PET, which is a valuable diagnostic tool for KFD as it typically differentiates large clusters of small lymph nodes typical of KFD from normal lymph nodes.","PeriodicalId":502981,"journal":{"name":"European Journal of Case Reports in Internal Medicine","volume":"119 24","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-01-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139596523","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}
京公网安备 11010802042870号
京ICP备2023020795号-1
扫码关注我们
求助内容：
应助结果提醒方式：