{"title":"罕见的老年女性全身性囊性血管瘤最初误诊为血管肿瘤1例报告并文献复习。","authors":"Sandeep Mohan, Rahul Krishnan, Sernam Kuttiyil, S Archa, Hamdi Nizar Ahamed, Rashik Ismail","doi":"10.13107/jocr.2025.v15.i01.5104","DOIUrl":null,"url":null,"abstract":"<p><strong>Introduction: </strong>Systemic cystic angiomatosis is an exceedingly rare condition characterized by widespread cystic vascular lesions involving multiple organs. Its clinical presentation can be non-specific, often leading to diagnostic challenges. This report discusses the case of a 72-year-old female with a long-standing history of diabetes mellitus who presented with non-specific symptoms, ultimately diagnosed with systemic cystic angiomatosis after an initial misdiagnosis of vascular neoplasia. The role of advanced imaging techniques and a multidisciplinary, individualized management approach is emphasized.</p><p><strong>Case report: </strong>A 72-year-old female with long-standing diabetes presented with non-specific symptoms, including fatigue, fever, and rashes on the upper limbs. Initial physical examination revealed anemia and severe thrombocytopenia with a leukoerythroblastic blood picture. Despite unremarkable initial imaging studies, a bone marrow biopsy suggested vascular neoplasia. Further evaluation with a positron emission tomography (PET) scan revealed multiple non-avid lytic skeletal areas and cystic liver lesions, leading to a diagnosis of systemic cystic angiomatosis. A conservative management approach with danazol and eltrombopag was adopted. The patient later developed an acute-on-chronic subdural hematoma, a severe complication of the condition.</p><p><strong>Conclusion: </strong>This case highlights the diagnostic complexity and the necessity for individualized management strategies in systemic cystic angiomatosis. It underscores the importance of considering rare diagnoses when faced with non-specific symptoms and atypical laboratory findings. Advanced imaging techniques, such as PET scans, and a multidisciplinary approach are crucial for accurate diagnosis and effective management of this rare condition.</p>","PeriodicalId":16647,"journal":{"name":"Journal of Orthopaedic Case Reports","volume":"15 1","pages":"8-12"},"PeriodicalIF":0.0000,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11723755/pdf/","citationCount":"0","resultStr":"{\"title\":\"A Rare Case of Systemic Cystic Angiomatosis in an Elderly Female Initially Misdiagnosed as Vascular Neoplasm: A Case Report and Literature Review.\",\"authors\":\"Sandeep Mohan, Rahul Krishnan, Sernam Kuttiyil, S Archa, Hamdi Nizar Ahamed, Rashik Ismail\",\"doi\":\"10.13107/jocr.2025.v15.i01.5104\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Introduction: </strong>Systemic cystic angiomatosis is an exceedingly rare condition characterized by widespread cystic vascular lesions involving multiple organs. Its clinical presentation can be non-specific, often leading to diagnostic challenges. This report discusses the case of a 72-year-old female with a long-standing history of diabetes mellitus who presented with non-specific symptoms, ultimately diagnosed with systemic cystic angiomatosis after an initial misdiagnosis of vascular neoplasia. The role of advanced imaging techniques and a multidisciplinary, individualized management approach is emphasized.</p><p><strong>Case report: </strong>A 72-year-old female with long-standing diabetes presented with non-specific symptoms, including fatigue, fever, and rashes on the upper limbs. Initial physical examination revealed anemia and severe thrombocytopenia with a leukoerythroblastic blood picture. Despite unremarkable initial imaging studies, a bone marrow biopsy suggested vascular neoplasia. Further evaluation with a positron emission tomography (PET) scan revealed multiple non-avid lytic skeletal areas and cystic liver lesions, leading to a diagnosis of systemic cystic angiomatosis. A conservative management approach with danazol and eltrombopag was adopted. The patient later developed an acute-on-chronic subdural hematoma, a severe complication of the condition.</p><p><strong>Conclusion: </strong>This case highlights the diagnostic complexity and the necessity for individualized management strategies in systemic cystic angiomatosis. It underscores the importance of considering rare diagnoses when faced with non-specific symptoms and atypical laboratory findings. Advanced imaging techniques, such as PET scans, and a multidisciplinary approach are crucial for accurate diagnosis and effective management of this rare condition.</p>\",\"PeriodicalId\":16647,\"journal\":{\"name\":\"Journal of Orthopaedic Case Reports\",\"volume\":\"15 1\",\"pages\":\"8-12\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2025-01-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11723755/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of Orthopaedic Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.13107/jocr.2025.v15.i01.5104\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Orthopaedic Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.13107/jocr.2025.v15.i01.5104","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
A Rare Case of Systemic Cystic Angiomatosis in an Elderly Female Initially Misdiagnosed as Vascular Neoplasm: A Case Report and Literature Review.
Introduction: Systemic cystic angiomatosis is an exceedingly rare condition characterized by widespread cystic vascular lesions involving multiple organs. Its clinical presentation can be non-specific, often leading to diagnostic challenges. This report discusses the case of a 72-year-old female with a long-standing history of diabetes mellitus who presented with non-specific symptoms, ultimately diagnosed with systemic cystic angiomatosis after an initial misdiagnosis of vascular neoplasia. The role of advanced imaging techniques and a multidisciplinary, individualized management approach is emphasized.
Case report: A 72-year-old female with long-standing diabetes presented with non-specific symptoms, including fatigue, fever, and rashes on the upper limbs. Initial physical examination revealed anemia and severe thrombocytopenia with a leukoerythroblastic blood picture. Despite unremarkable initial imaging studies, a bone marrow biopsy suggested vascular neoplasia. Further evaluation with a positron emission tomography (PET) scan revealed multiple non-avid lytic skeletal areas and cystic liver lesions, leading to a diagnosis of systemic cystic angiomatosis. A conservative management approach with danazol and eltrombopag was adopted. The patient later developed an acute-on-chronic subdural hematoma, a severe complication of the condition.
Conclusion: This case highlights the diagnostic complexity and the necessity for individualized management strategies in systemic cystic angiomatosis. It underscores the importance of considering rare diagnoses when faced with non-specific symptoms and atypical laboratory findings. Advanced imaging techniques, such as PET scans, and a multidisciplinary approach are crucial for accurate diagnosis and effective management of this rare condition.