Acute transverse myelitis is a rare neurological complication of systemic lupus erythematosus (SLE), particularly in sub-Saharan African origin patients. We report a case of acute transverse myelitis in a sub-Saharan African origin patient with SLE. This case involved a 37-year-old female patient with SLE and secondary Sjögren's syndrome, presenting with progressively worsening paraplegia. Examination revealed paraplegia of the lower limbs and paraparesis of the upper limbs. Lumbar puncture yielded clear cerebrospinal fluid with hyperproteinorachia. Spinal magnetic resonance imaging showed transverse myelitis in lumbar level; the 24-hour proteinuria was 4.2 g. The diagnosis of acute transverse myelitis and renal flare of SLE was made. Clinical improvement was achieved with methylprednisolone, cyclophosphamide, and physiotherapy. Acute transverse myelitis remains a rare and poorly understood complication of lupus, characterized by its severity and very poor prognosis.
{"title":"Systemic Lupus Erythematosus and Acute Transverse Myelitis: An Unusual Association-A Case Report.","authors":"Baowêndsôm Armelle Djamilatou Sawadogo, Yannick Laurent Tchenadoyo Bayala, Ahmed Devis Wendyam Zougmoré, Marcellin Bonkoungou, Fulgence Kaboré, Wendlassida Joëlle Stéphanie Zabsonré/Tiendrébéogo, Dieu-Donné Ouédraogo","doi":"10.1155/carm/9243188","DOIUrl":"10.1155/carm/9243188","url":null,"abstract":"<p><p>Acute transverse myelitis is a rare neurological complication of systemic lupus erythematosus (SLE), particularly in sub-Saharan African origin patients. We report a case of acute transverse myelitis in a sub-Saharan African origin patient with SLE. This case involved a 37-year-old female patient with SLE and secondary Sjögren's syndrome, presenting with progressively worsening paraplegia. Examination revealed paraplegia of the lower limbs and paraparesis of the upper limbs. Lumbar puncture yielded clear cerebrospinal fluid with hyperproteinorachia. Spinal magnetic resonance imaging showed transverse myelitis in lumbar level; the 24-hour proteinuria was 4.2 g. The diagnosis of acute transverse myelitis and renal flare of SLE was made. Clinical improvement was achieved with methylprednisolone, cyclophosphamide, and physiotherapy. Acute transverse myelitis remains a rare and poorly understood complication of lupus, characterized by its severity and very poor prognosis.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"9243188"},"PeriodicalIF":0.7,"publicationDate":"2025-12-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12750142/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145877755","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 : 2025-12-29eCollection Date: 2025-01-01DOI: 10.1155/carm/1281603
Henrique Madureira Da Rocha Coutinho, José Xavier López, Juan Luis Muñoz Noblecilla, Esmeralci Ferreira, Joaquim Henrique De Souza Aguiar Coutinho
Background: Cardiovascular syphilis (CVS) is a rare but severe manifestation of tertiary syphilis, often remaining clinically silent until life-threatening complications develop. Syphilitic aortitis can lead to aneurysmal degeneration, most commonly involving the ascending aorta and aortic arch. Despite advances in public health, the global resurgence of syphilis has renewed concern about its cardiovascular sequelae and the need for diagnostic vigilance.
Case report: We describe a 59-year-old male with systemic hypertension and a remote smoking history, who was largely asymptomatic except for mild exertional dyspnea (New York Heart Association Class II). During routine preoperative evaluation for noncardiac surgery, imaging revealed a giant aortic arch aneurysm measuring 114 × 94 × 93 mm, with associated dilatation of the ascending and descending aorta. Serologic testing confirmed syphilitic infection, and syphilitic aortitis was identified as the underlying etiology. After the completion of benzathine penicillin therapy, the patient underwent successful replacement of the ascending aorta and entire aortic arch under deep hypothermic circulatory arrest with selective antegrade cerebral perfusion. The supraaortic trunks were reimplanted en bloc using the island technique. Postoperative recovery was complicated by pleural effusion and transient neurological symptoms, both of which were managed conservatively, with full resolution. At two-month follow-up, the patient had returned to normal daily activities with preserved functional capacity.
Conclusion: This case illustrates an atypical, arch-predominant presentation of CVS diagnosed incidentally in the contemporary era. It emphasizes the need to consider infectious etiologies, particularly syphilis, in patients with nonatherosclerotic or unusual aortic aneurysm patterns. Early recognition, appropriate antimicrobial therapy, and timely surgical intervention are essential to prevent catastrophic outcomes. The ongoing resurgence of syphilis underscores the importance of maintaining clinical awareness of this "forgotten disease," especially in patients with atypical aortic pathology.
{"title":"Silent Threat: Incidental Diagnosis and Surgical Management of a Giant Syphilitic Aortic Aneurysm.","authors":"Henrique Madureira Da Rocha Coutinho, José Xavier López, Juan Luis Muñoz Noblecilla, Esmeralci Ferreira, Joaquim Henrique De Souza Aguiar Coutinho","doi":"10.1155/carm/1281603","DOIUrl":"10.1155/carm/1281603","url":null,"abstract":"<p><strong>Background: </strong>Cardiovascular syphilis (CVS) is a rare but severe manifestation of tertiary syphilis, often remaining clinically silent until life-threatening complications develop. Syphilitic aortitis can lead to aneurysmal degeneration, most commonly involving the ascending aorta and aortic arch. Despite advances in public health, the global resurgence of syphilis has renewed concern about its cardiovascular sequelae and the need for diagnostic vigilance.</p><p><strong>Case report: </strong>We describe a 59-year-old male with systemic hypertension and a remote smoking history, who was largely asymptomatic except for mild exertional dyspnea (New York Heart Association Class II). During routine preoperative evaluation for noncardiac surgery, imaging revealed a giant aortic arch aneurysm measuring 114 × 94 × 93 mm, with associated dilatation of the ascending and descending aorta. Serologic testing confirmed syphilitic infection, and syphilitic aortitis was identified as the underlying etiology. After the completion of benzathine penicillin therapy, the patient underwent successful replacement of the ascending aorta and entire aortic arch under deep hypothermic circulatory arrest with selective antegrade cerebral perfusion. The supraaortic trunks were reimplanted en bloc using the island technique. Postoperative recovery was complicated by pleural effusion and transient neurological symptoms, both of which were managed conservatively, with full resolution. At two-month follow-up, the patient had returned to normal daily activities with preserved functional capacity.</p><p><strong>Conclusion: </strong>This case illustrates an atypical, arch-predominant presentation of CVS diagnosed incidentally in the contemporary era. It emphasizes the need to consider infectious etiologies, particularly syphilis, in patients with nonatherosclerotic or unusual aortic aneurysm patterns. Early recognition, appropriate antimicrobial therapy, and timely surgical intervention are essential to prevent catastrophic outcomes. The ongoing resurgence of syphilis underscores the importance of maintaining clinical awareness of this \"forgotten disease,\" especially in patients with atypical aortic pathology.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"1281603"},"PeriodicalIF":0.7,"publicationDate":"2025-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12745877/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145862274","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 : 2025-12-29eCollection Date: 2025-01-01DOI: 10.1155/carm/4810248
Georgios Lyras, Prokopios Karydis, Sofia Dionysia Tetradi, Aikaterini Maria Lechouriti, Charalampos Potsios, Dimitra Taprantzi, Christos Michailides, Dimitrios Velissaris
Introduction: Immune checkpoint inhibitors (ICIs), such as nivolumab, have made significant advancements in the treatment of several malignancies. However, they have been associated with a new and diverse spectrum of immune-related adverse effects (irAEs). We report the case of a patient with metastatic renal cell carcinoma who developed encephalitis and other potential irAEs, including hypophysitis with isolated adrenocorticotropic hormone deficiency, transient autoimmune thyroiditis, and autoimmune hemolytic anemia, following combination therapy with nivolumab and cabozantinib. The patient ultimately required rituximab administration.
Case presentation: A 55-year-old woman with a history of metastatic renal cell carcinoma and a recent hospitalization for urinary tract infection was admitted to the Department of Internal Medicine due to confusion, disorientation, and hypotension. A brain MRI revealed multiple new T2/FLAIR hyperintense lesions. After an extensive workup to exclude common neurologic ailments, an adverse reaction due to nivolumab therapy was hypothesized. Treatment was initiated with corticosteroids and IVIG, and the patient showed rapid improvement. Given the fast response to corticosteroids, the diagnosis of ICI-induced encephalitis was made. Rituximab was then administered, and the patient showed near complete remission of her neurologic symptoms. The patient's hypotension was attributed to secondary adrenal insufficiency and treated accordingly. Concomitant findings included hyperthyroidism and later-onset autoimmune hemolytic anemia, both of which were attributed to nivolumab, although the diagnosis could not be confirmed with certainty. Significant improvement was achieved with corticosteroid treatment and rituximab.
Conclusion: irAEs can affect any system, individually or simultaneously, and require high clinical suspicion to diagnose. ICI-induced encephalitis is potentially life-threatening but, with prompt treatment, near-complete recovery is possible. Corticosteroids and IVIG constitute the main treatment, although rituximab may be used successfully in more severe cases.
{"title":"Encephalitis and Immune-Related Complications in a Patient Treated With Nivolumab: A Case Report.","authors":"Georgios Lyras, Prokopios Karydis, Sofia Dionysia Tetradi, Aikaterini Maria Lechouriti, Charalampos Potsios, Dimitra Taprantzi, Christos Michailides, Dimitrios Velissaris","doi":"10.1155/carm/4810248","DOIUrl":"10.1155/carm/4810248","url":null,"abstract":"<p><strong>Introduction: </strong>Immune checkpoint inhibitors (ICIs), such as nivolumab, have made significant advancements in the treatment of several malignancies. However, they have been associated with a new and diverse spectrum of immune-related adverse effects (irAEs). We report the case of a patient with metastatic renal cell carcinoma who developed encephalitis and other potential irAEs, including hypophysitis with isolated adrenocorticotropic hormone deficiency, transient autoimmune thyroiditis, and autoimmune hemolytic anemia, following combination therapy with nivolumab and cabozantinib. The patient ultimately required rituximab administration.</p><p><strong>Case presentation: </strong>A 55-year-old woman with a history of metastatic renal cell carcinoma and a recent hospitalization for urinary tract infection was admitted to the Department of Internal Medicine due to confusion, disorientation, and hypotension. A brain MRI revealed multiple new T2/FLAIR hyperintense lesions. After an extensive workup to exclude common neurologic ailments, an adverse reaction due to nivolumab therapy was hypothesized. Treatment was initiated with corticosteroids and IVIG, and the patient showed rapid improvement. Given the fast response to corticosteroids, the diagnosis of ICI-induced encephalitis was made. Rituximab was then administered, and the patient showed near complete remission of her neurologic symptoms. The patient's hypotension was attributed to secondary adrenal insufficiency and treated accordingly. Concomitant findings included hyperthyroidism and later-onset autoimmune hemolytic anemia, both of which were attributed to nivolumab, although the diagnosis could not be confirmed with certainty. Significant improvement was achieved with corticosteroid treatment and rituximab.</p><p><strong>Conclusion: </strong>irAEs can affect any system, individually or simultaneously, and require high clinical suspicion to diagnose. ICI-induced encephalitis is potentially life-threatening but, with prompt treatment, near-complete recovery is possible. Corticosteroids and IVIG constitute the main treatment, although rituximab may be used successfully in more severe cases.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"4810248"},"PeriodicalIF":0.7,"publicationDate":"2025-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12747856/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145877781","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 : 2025-12-25eCollection Date: 2025-01-01DOI: 10.1155/carm/6656955
Ravi Mani, John Dereix, Penelope Beddoes, Tom Lawton
Poisoning from mercury has the potential to affect multiple organ systems and can be fatal in some circumstances. We present a case of a patient who had both ingested and inhaled elemental mercury resulting in deposits in his pulmonary and gastrointestinal systems. The patient was admitted to the intensive care unit (ICU) as a precautionary measure but did not require any organ support. The patient was treated with chelation therapy guided by the UK National Poisons Information Service (NPIS) and was discharged from ICU after having a prophylactic appendicectomy due to mercury deposition. This case presented several challenges logistically as there was a need to follow expert guidance with regards to personal protective equipment (PPE) and waste disposal, alongside the clinical requirements.
{"title":"Mercury Poisoning and the Wider Implications.","authors":"Ravi Mani, John Dereix, Penelope Beddoes, Tom Lawton","doi":"10.1155/carm/6656955","DOIUrl":"10.1155/carm/6656955","url":null,"abstract":"<p><p>Poisoning from mercury has the potential to affect multiple organ systems and can be fatal in some circumstances. We present a case of a patient who had both ingested and inhaled elemental mercury resulting in deposits in his pulmonary and gastrointestinal systems. The patient was admitted to the intensive care unit (ICU) as a precautionary measure but did not require any organ support. The patient was treated with chelation therapy guided by the UK National Poisons Information Service (NPIS) and was discharged from ICU after having a prophylactic appendicectomy due to mercury deposition. This case presented several challenges logistically as there was a need to follow expert guidance with regards to personal protective equipment (PPE) and waste disposal, alongside the clinical requirements.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"6656955"},"PeriodicalIF":0.7,"publicationDate":"2025-12-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12740589/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145848936","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 : 2025-12-22eCollection Date: 2025-01-01DOI: 10.1155/carm/2450436
Sadhvi Batra, Mario Dervishi, Mohamed Al-Natour, Tyler Katz
Introduction: Diagnosing the cause of delayed postpregnancy hemorrhage is imperative for effective management. The most common factor to lead to this is retained products of conception (RPOC); however, it is often difficult to distinguish RPOC from uterine arteriovenous malformations (AVMs). AVMs are abnormal vascular connections that can lead to sudden and persistent uterine bleeding that can be life-threatening. Though rare, there are many challenges to establishing a true diagnosis of an AVM versus RPOC and planning for treatment when a diagnosis of AVM is suspected.
Methods: In this study, authors present a case series of five patients who had the potential diagnosis of an AVM and how each was managed. The authors extrapolated best practices from these five cases and created a new algorithm to tailor management for patients with concern for AVMs.
Results: This algorithm relies on utilizing transvaginal ultrasound as the first diagnostic technique, facilitating an early conversation with radiology if suspicion for AVM exists, and then determining if further imaging is needed or if the clinician can proceed to diagnostic hysteroscopy. In addition, if the decision is made for hysteroscopy, consultation with interventional radiology is recommended in case of urgent intervention to prevent further morbidity with hysterectomy.
Conclusion: Though much is left to be explored regarding conservative management for uterine AVM, this paper is an initial attempt at proposing a practice algorithm for multidisciplinary management of this potentially life-threatening condition.
{"title":"A Practice Algorithm for Distinguishing Uterine Arteriovenous Malformation in Postpregnancy Hemorrhage.","authors":"Sadhvi Batra, Mario Dervishi, Mohamed Al-Natour, Tyler Katz","doi":"10.1155/carm/2450436","DOIUrl":"10.1155/carm/2450436","url":null,"abstract":"<p><strong>Introduction: </strong>Diagnosing the cause of delayed postpregnancy hemorrhage is imperative for effective management. The most common factor to lead to this is retained products of conception (RPOC); however, it is often difficult to distinguish RPOC from uterine arteriovenous malformations (AVMs). AVMs are abnormal vascular connections that can lead to sudden and persistent uterine bleeding that can be life-threatening. Though rare, there are many challenges to establishing a true diagnosis of an AVM versus RPOC and planning for treatment when a diagnosis of AVM is suspected.</p><p><strong>Methods: </strong>In this study, authors present a case series of five patients who had the potential diagnosis of an AVM and how each was managed. The authors extrapolated best practices from these five cases and created a new algorithm to tailor management for patients with concern for AVMs.</p><p><strong>Results: </strong>This algorithm relies on utilizing transvaginal ultrasound as the first diagnostic technique, facilitating an early conversation with radiology if suspicion for AVM exists, and then determining if further imaging is needed or if the clinician can proceed to diagnostic hysteroscopy. In addition, if the decision is made for hysteroscopy, consultation with interventional radiology is recommended in case of urgent intervention to prevent further morbidity with hysterectomy.</p><p><strong>Conclusion: </strong>Though much is left to be explored regarding conservative management for uterine AVM, this paper is an initial attempt at proposing a practice algorithm for multidisciplinary management of this potentially life-threatening condition.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"2450436"},"PeriodicalIF":0.7,"publicationDate":"2025-12-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12723177/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145827063","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 : 2025-12-22eCollection Date: 2025-01-01DOI: 10.1155/carm/7243012
Veysel Erol, Osman Parça, Nevin Alayvaz Aslan, Nilay Şen Türk
The recent inclusion of immune-privileged site lymphoma in the World Health Organization classification signifies a distinctive entity encompassing the central nervous system and testicles. This classification is rooted in the unique challenges posed by the blood-brain barrier and blood-testis barrier, along with the distinct cancer microenvironment compared to standard lymphomas. These intricacies contribute to the complexity of treatment strategies, and as of now, a standardized protocol remains elusive. Our presented case underscores the critical need for a comprehensive treatment plan in immune-privileged site lymphomas, where surgery alone, in the form of indolent intervention, may fall short in addressing the underlying aggressive nature of the disease. Failure to administer systemic treatment, where indicated, heightens the risk of aggressive recurrence and substantially elevates mortality rates. Through a detailed examination of our case, we aim to contribute to the evolving body of knowledge surrounding these unique lymphomas, offering valuable insights that may guide future treatment strategies and improve patient outcomes.
{"title":"Choroidal Recurrence of Testicular Diffuse Large B-Cell Lymphoma: A Diagnostic and Therapeutic Challenge in an Immune-Privileged Site.","authors":"Veysel Erol, Osman Parça, Nevin Alayvaz Aslan, Nilay Şen Türk","doi":"10.1155/carm/7243012","DOIUrl":"10.1155/carm/7243012","url":null,"abstract":"<p><p>The recent inclusion of immune-privileged site lymphoma in the World Health Organization classification signifies a distinctive entity encompassing the central nervous system and testicles. This classification is rooted in the unique challenges posed by the blood-brain barrier and blood-testis barrier, along with the distinct cancer microenvironment compared to standard lymphomas. These intricacies contribute to the complexity of treatment strategies, and as of now, a standardized protocol remains elusive. Our presented case underscores the critical need for a comprehensive treatment plan in immune-privileged site lymphomas, where surgery alone, in the form of indolent intervention, may fall short in addressing the underlying aggressive nature of the disease. Failure to administer systemic treatment, where indicated, heightens the risk of aggressive recurrence and substantially elevates mortality rates. Through a detailed examination of our case, we aim to contribute to the evolving body of knowledge surrounding these unique lymphomas, offering valuable insights that may guide future treatment strategies and improve patient outcomes.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"7243012"},"PeriodicalIF":0.7,"publicationDate":"2025-12-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12723176/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145826983","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 : 2025-12-19eCollection Date: 2025-01-01DOI: 10.1155/carm/1829205
Mohammed Ayyad, Radhika Patel, Kevin Molnar, Jared Walsh
Vanishing bile duct syndrome (VBDS) is an extremely rare disorder characterized by the progressive destruction of intrahepatic bile ducts, leading to cholestasis and liver dysfunction. It is most commonly associated with autoimmune and infectious conditions but can also occur in immunocompromised states such as human immunodeficiency virus (HIV). We report the case of a 58-year-old Hispanic woman with HIV who presented with cyclical fevers, transaminitis, hyperbilirubinemia, and hyponatremia over a 3-month period. Laboratory workup revealed marked cholestasis with severe elevations in alkaline phosphatase. A liver biopsy confirmed VBDS with a significant reduction in intrahepatic bile ducts. The patient continued antiretroviral treatment, prophylactic antibiotics, and was started on ursodiol, resulting in improvement in liver tests. An extensive workup was done to investigate for an underlying opportunistic infection or lymphoproliferative disease as a culprit for VBDS. VBDS, while exceedingly rare in HIV-positive patients, is likely immune mediated, involving T-cell dysfunction and cytokine-mediated biliary epithelial cell apoptosis. While drug-induced hepatotoxicity from antiretroviral therapy is a possible contributing factor, it is crucial to consider other underlying etiologies in immunocompromised patients such as infections and lymphoproliferative disorders. VBDS should be considered in HIV-positive patients with unexplained liver dysfunction, particularly those with low CD4 counts. Early recognition and diagnosis are crucial for initiating appropriate treatment and preventing further liver damage. This case highlights the diagnostic complexity and need for ongoing research into the interplay between immunosuppression, cholangiopathy, and liver dysfunction in HIV patients.
{"title":"An Elusive Diagnosis of Vanishing Bile Duct Syndrome in an HIV Patient.","authors":"Mohammed Ayyad, Radhika Patel, Kevin Molnar, Jared Walsh","doi":"10.1155/carm/1829205","DOIUrl":"10.1155/carm/1829205","url":null,"abstract":"<p><p>Vanishing bile duct syndrome (VBDS) is an extremely rare disorder characterized by the progressive destruction of intrahepatic bile ducts, leading to cholestasis and liver dysfunction. It is most commonly associated with autoimmune and infectious conditions but can also occur in immunocompromised states such as human immunodeficiency virus (HIV). We report the case of a 58-year-old Hispanic woman with HIV who presented with cyclical fevers, transaminitis, hyperbilirubinemia, and hyponatremia over a 3-month period. Laboratory workup revealed marked cholestasis with severe elevations in alkaline phosphatase. A liver biopsy confirmed VBDS with a significant reduction in intrahepatic bile ducts. The patient continued antiretroviral treatment, prophylactic antibiotics, and was started on ursodiol, resulting in improvement in liver tests. An extensive workup was done to investigate for an underlying opportunistic infection or lymphoproliferative disease as a culprit for VBDS. VBDS, while exceedingly rare in HIV-positive patients, is likely immune mediated, involving T-cell dysfunction and cytokine-mediated biliary epithelial cell apoptosis. While drug-induced hepatotoxicity from antiretroviral therapy is a possible contributing factor, it is crucial to consider other underlying etiologies in immunocompromised patients such as infections and lymphoproliferative disorders. VBDS should be considered in HIV-positive patients with unexplained liver dysfunction, particularly those with low CD4 counts. Early recognition and diagnosis are crucial for initiating appropriate treatment and preventing further liver damage. This case highlights the diagnostic complexity and need for ongoing research into the interplay between immunosuppression, cholangiopathy, and liver dysfunction in HIV patients.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"1829205"},"PeriodicalIF":0.7,"publicationDate":"2025-12-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12715408/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145803422","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 : 2025-12-18eCollection Date: 2025-01-01DOI: 10.1155/carm/5059027
Jianxin Dun, Qun Hu, Aiguo Liu, Yaqin Wang, Ai Zhang
Purpose: Pediatric Rosai-Dorfman disease (RDD) is extremely rare, and the current treatment plan is not unified. We hope that our study provides new ideas for the clinical treatment of RDD.
Methods: We report a case in which RDD was successfully treated with prednisone and vincristine, resulting in regression of enlarged lymph nodes. In this study, informed consent forms were obtained from participants for their involvement in the research and publication of relevant case details. Moreover, this study aimed to conceal personal patient information.
Results: The child underwent two pathological examinations before a definitive diagnosis was made. Percutaneous fine-needle aspiration biopsy was performed on his first admission, and pathological examination showed a purulent granulomatous reaction under a microscope; however, antibiotic treatment was ineffective. The second pathological biopsy performed at the time of surgery showed RDD. After treatment with vincristine and prednisone, the neck mass significantly shrank and did not increase in size after the treatment was stopped. The patient remains under follow-up.
Conclusions: Sufficient pathological tissue samples were obtained via surgical biopsy to diagnose RDD. Treatment with chemotherapy, including prednisone and vincristine, can induce remission of RDD.
{"title":"Prednisone and Vincristine for the Treatment of Pediatric Rosai-Dorfman Disease: A Case Report.","authors":"Jianxin Dun, Qun Hu, Aiguo Liu, Yaqin Wang, Ai Zhang","doi":"10.1155/carm/5059027","DOIUrl":"10.1155/carm/5059027","url":null,"abstract":"<p><strong>Purpose: </strong>Pediatric Rosai-Dorfman disease (RDD) is extremely rare, and the current treatment plan is not unified. We hope that our study provides new ideas for the clinical treatment of RDD.</p><p><strong>Methods: </strong>We report a case in which RDD was successfully treated with prednisone and vincristine, resulting in regression of enlarged lymph nodes. In this study, informed consent forms were obtained from participants for their involvement in the research and publication of relevant case details. Moreover, this study aimed to conceal personal patient information.</p><p><strong>Results: </strong>The child underwent two pathological examinations before a definitive diagnosis was made. Percutaneous fine-needle aspiration biopsy was performed on his first admission, and pathological examination showed a purulent granulomatous reaction under a microscope; however, antibiotic treatment was ineffective. The second pathological biopsy performed at the time of surgery showed RDD. After treatment with vincristine and prednisone, the neck mass significantly shrank and did not increase in size after the treatment was stopped. The patient remains under follow-up.</p><p><strong>Conclusions: </strong>Sufficient pathological tissue samples were obtained via surgical biopsy to diagnose RDD. Treatment with chemotherapy, including prednisone and vincristine, can induce remission of RDD.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"5059027"},"PeriodicalIF":0.7,"publicationDate":"2025-12-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12715339/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145803333","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 : 2025-12-18eCollection Date: 2025-01-01DOI: 10.1155/carm/8785833
Jovana Ivanovic, Masa Kovacevic, Tamara Svabic Medjedovic, Vanja Radisic Vukomanovic, Branislav Ralic, Aleksandar Ristic, Dejana Jovanovic, Ivana Berisavac
Introduction: Subacute encephalopathy with seizures in alcoholics (SESA) is a rare complication of chronic alcohol abuse manifested as seizures, altered mental state, and focal neurological deficits. Electroencephalography usually shows focal epileptic anomalies, and brain MRI reveals reversible focal cortical or subcortical T2w/FLAIR hyperintense lesions. This syndrome is still underrecognized but a very important entity with a high risk of recurrent seizures requiring long-term antiseizure polytherapy treatment and has an uncertain outcome.
Case series: We report four male patients who presented with focal motor seizures or status epilepticus associated with chronic alcohol intake. They were treated with antiseizure polytherapy combined with other intensive symptomatic therapies. Two patients had a good outcome without new seizure occurrences, while the other two patients, despite intensive care treatment, developed multiorgan dysfunction and had a fatal outcome.
Conclusion: SESA is a life-threatening disease with an uncertain outcome, especially if hospitalization in intensive care units is needed.
{"title":"Patients With SESA Syndrome May Not Have a Good Outcome: A Case Series.","authors":"Jovana Ivanovic, Masa Kovacevic, Tamara Svabic Medjedovic, Vanja Radisic Vukomanovic, Branislav Ralic, Aleksandar Ristic, Dejana Jovanovic, Ivana Berisavac","doi":"10.1155/carm/8785833","DOIUrl":"10.1155/carm/8785833","url":null,"abstract":"<p><strong>Introduction: </strong>Subacute encephalopathy with seizures in alcoholics (SESA) is a rare complication of chronic alcohol abuse manifested as seizures, altered mental state, and focal neurological deficits. Electroencephalography usually shows focal epileptic anomalies, and brain MRI reveals reversible focal cortical or subcortical T2w/FLAIR hyperintense lesions. This syndrome is still underrecognized but a very important entity with a high risk of recurrent seizures requiring long-term antiseizure polytherapy treatment and has an uncertain outcome.</p><p><strong>Case series: </strong>We report four male patients who presented with focal motor seizures or status epilepticus associated with chronic alcohol intake. They were treated with antiseizure polytherapy combined with other intensive symptomatic therapies. Two patients had a good outcome without new seizure occurrences, while the other two patients, despite intensive care treatment, developed multiorgan dysfunction and had a fatal outcome.</p><p><strong>Conclusion: </strong>SESA is a life-threatening disease with an uncertain outcome, especially if hospitalization in intensive care units is needed.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"8785833"},"PeriodicalIF":0.7,"publicationDate":"2025-12-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12714074/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145803336","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}
Mycobacterial spindle cell pseudotumor (MSP) is an uncommon granulomatous inflammation often found in immunocompromised patients, especially those with HIV. This case report discusses an 18-year-old female with congenital HIV, who presented with severe epigastric pain, nausea, and noteworthy lymphadenopathy. Imaging studies identified lobulated masses, and a core needle biopsy confirmed the diagnosis of MSP, revealing spindle-shaped histiocytes and acid-fast bacilli. Additionally, we reviewed nine MSP cases documented since 2017.
{"title":"Mycobacterial Spindle Cell Pseudotumor in an 18-Year-Old Female With Congenital HIV: A Case Report and Literature Review.","authors":"Zahra Aminparast, Zeinab Shakibaee Fard, Amirmohammad Khodaei","doi":"10.1155/carm/1612986","DOIUrl":"10.1155/carm/1612986","url":null,"abstract":"<p><p>Mycobacterial spindle cell pseudotumor (MSP) is an uncommon granulomatous inflammation often found in immunocompromised patients, especially those with HIV. This case report discusses an 18-year-old female with congenital HIV, who presented with severe epigastric pain, nausea, and noteworthy lymphadenopathy. Imaging studies identified lobulated masses, and a core needle biopsy confirmed the diagnosis of MSP, revealing spindle-shaped histiocytes and acid-fast bacilli. Additionally, we reviewed nine MSP cases documented since 2017.</p>","PeriodicalId":9627,"journal":{"name":"Case Reports in Medicine","volume":"2025 ","pages":"1612986"},"PeriodicalIF":0.7,"publicationDate":"2025-12-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12714090/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145803383","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}