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。

Sandeep Mohan, Rahul Krishnan, Sernam Kuttiyil, S Archa, Hamdi Nizar Ahamed, Rashik Ismail
{"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}
引用次数: 0

摘要

简介:全身性囊性血管瘤病是一种非常罕见的疾病,其特征是广泛的囊性血管病变累及多个器官。其临床表现可能是非特异性的,常常导致诊断困难。本报告讨论一例72岁女性糖尿病长期病史,其表现为非特异性症状,最初误诊为血管瘤,最终诊断为全身性囊性血管瘤病。强调了先进的成像技术和多学科、个性化管理方法的作用。病例报告:一名72岁女性,长期患有糖尿病,表现为非特异性症状,包括疲劳、发烧和上肢皮疹。最初的体格检查显示贫血和严重的血小板减少症,并伴有白细胞母细胞血象。尽管最初的影像学检查不明显,骨髓活检提示血管瘤。进一步的正电子发射断层扫描(PET)显示多个非溶解性骨骼区域和囊性肝脏病变,导致系统性囊性血管瘤病的诊断。保守治疗采用达那唑和电子波巴。患者后来发展为急性慢性硬膜下血肿,这是病情的严重并发症。结论:本病例突出了全身性囊性血管瘤病诊断的复杂性和个体化治疗策略的必要性。它强调了在面对非特异性症状和非典型实验室结果时考虑罕见诊断的重要性。先进的成像技术,如PET扫描,以及多学科方法对于这种罕见疾病的准确诊断和有效治疗至关重要。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
查看原文
分享 分享
微信好友 朋友圈 QQ好友 复制链接
本刊更多论文
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.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
自引率
0.00%
发文量
128
审稿时长
30 weeks
期刊最新文献
A Rare Case of Localized Pigmented Villonodular Synovitis of the Wrist presenting as Carpal Tunnel Syndrome. A Rare Case of Gas Gangrene after Upper Limb Fracture. Ochronotic Arthropathy of the Shoulder - A Rare Case Report. Re-arthroscopic Findings 18 Years After Osteochondral Autologous Transplantation for Cartilage Lesion in the Knee: A Case Report. A Rare Case of Staphylococcus Caprae Periprosthetic Hip Infection with Unusual Clinical Presentation.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
现在去查看 取消
×
提示
确定
0
微信
客服QQ
Book学术公众号 扫码关注我们
反馈
×
意见反馈
请填写您的意见或建议
请填写您的手机或邮箱
已复制链接
已复制链接
快去分享给好友吧!
我知道了
×
扫码分享
扫码分享
Book学术官方微信
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术
文献互助 智能选刊 最新文献 互助须知 联系我们:info@booksci.cn
Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。
Copyright © 2023 Book学术 All rights reserved.
ghs 京公网安备 11010802042870号 京ICP备2023020795号-1