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成人斯蒂尔病与肺栓塞:一例报告

Adult-Onset Still's Disease and Pulmonary Embolism: A Case Report.

作者信息

Alotaibie May A, Alqahtani Mohammed, Rajendram Siraj

机构信息

Neurology, National Guard Health Affairs, Riyadh, SAU.

Internal Medicine, National Guard Health Affairs, Riyadh, SAU.

出版信息

Cureus. 2024 Sep 12;16(9):e69245. doi: 10.7759/cureus.69245. eCollection 2024 Sep.

DOI:10.7759/cureus.69245
PMID:39398803
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11470388/
Abstract

Adult-onset Still's disease (AOSD) is a rare systemic inflammatory disorder characterized by fever, rash, arthralgia, and systemic inflammation. Pulmonary embolism (PE), a potentially life-threatening complication, is infrequently associated with AOSD. This report presents a unique case that highlights the importance of considering atypical presentations of PE in this patient population despite the absence of classical risk factors. An 84-year-old male with hypertension, benign prostatic hypertrophy, and AOSD diagnosed six years prior, presented with confusion, fever, and malaise for two days. He denied any recent travel or immobilization. Examination revealed tachycardia and reduced oxygen saturation on room air. D-dimer was elevated, and CT chest angiography (CTCA) confirmed left upper and lower segmental PE without cardiac strain. Investigations for infection were negative. Initial treatment with intravenous heparin was switched to apixaban. The patient was eventually discharged home with stable vital signs. The inflammatory state in AOSD might contribute to hypercoagulability, increasing the risk of PE. This case emphasizes the importance of considering PE in AOSD patients, even in the absence of classical risk factors, to prevent potentially fatal complications. A literature review revealed few cases of AOSD presenting with PE, highlighting the atypical presentation and need for increased awareness. This case underscores the rare, but potentially serious, association between AOSD and unprovoked PE. Clinicians managing AOSD should maintain a high index of suspicion for PE, particularly in patients presenting with unexplained respiratory symptoms.

摘要

成人斯蒂尔病(AOSD)是一种罕见的全身性炎症性疾病,其特征为发热、皮疹、关节痛和全身炎症。肺栓塞(PE)是一种潜在的危及生命的并发症,与AOSD的关联并不常见。本报告介绍了一个独特的病例,强调了尽管缺乏经典危险因素,但在该患者群体中考虑PE非典型表现的重要性。一名84岁男性,有高血压、良性前列腺增生,6年前被诊断为AOSD,出现意识模糊、发热和乏力2天。他否认近期有旅行或制动情况。检查发现心动过速,在室内空气中氧饱和度降低。D-二聚体升高,胸部CT血管造影(CTCA)证实左上和下叶段肺栓塞,无心脏受累。感染相关检查结果为阴性。最初静脉注射肝素治疗改为阿哌沙班治疗。患者最终生命体征稳定后出院回家。AOSD中的炎症状态可能导致高凝状态,增加肺栓塞风险。该病例强调了即使在没有经典危险因素的情况下,AOSD患者也需考虑肺栓塞,以预防潜在的致命并发症。文献综述显示,很少有AOSD合并肺栓塞的病例,突出了其非典型表现以及提高认识的必要性。该病例强调了AOSD与不明原因肺栓塞之间罕见但可能严重的关联。管理AOSD的临床医生应高度怀疑肺栓塞,尤其是在出现不明原因呼吸道症状的患者中。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f85c/11470388/b27362d6bd01/cureus-0016-00000069245-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f85c/11470388/ea802723deb7/cureus-0016-00000069245-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f85c/11470388/5284b483bedc/cureus-0016-00000069245-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f85c/11470388/b27362d6bd01/cureus-0016-00000069245-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f85c/11470388/ea802723deb7/cureus-0016-00000069245-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f85c/11470388/5284b483bedc/cureus-0016-00000069245-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f85c/11470388/b27362d6bd01/cureus-0016-00000069245-i03.jpg

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本文引用的文献

1
An Initially Missed Diagnosis of Venous Thromboembolic Phenomenon in Adult-Onset Still's Disease: A Case Report and Literature Review.成人Still 病中静脉血栓栓塞现象的初始漏诊:病例报告及文献复习。
Am J Case Rep. 2023 Jun 23;24:e939520. doi: 10.12659/AJCR.939520.
2
Increased incidence of adult-onset Still's disease in association with COVID-19 vaccination and SARS-CoV-2 infection.与 COVID-19 疫苗接种和 SARS-CoV-2 感染相关的成人斯蒂尔病发病率增加。
Orphanet J Rare Dis. 2023 Mar 10;18(1):50. doi: 10.1186/s13023-023-02651-3.
3
Recurrent deep venous thromboses in a patient with adult-onset Still's disease.
成人Still 病患者反复发生深静脉血栓形成。
BMJ Case Rep. 2021 Jun 22;14(6):e240986. doi: 10.1136/bcr-2020-240986.
4
Unprovoked Pulmonary Embolism Identified on Initial Presentation of Adult-Onset Still Disease in an Elderly Patient With No Malignancy.在一名无恶性肿瘤的老年患者成人斯蒂尔病初发时发现的无诱因肺栓塞。
J Clin Rheumatol. 2020 Mar;26(2):e40-e42. doi: 10.1097/RHU.0000000000000812.
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[Adult-onset Still's disease complications].[成人斯蒂尔病的并发症]
Rev Med Interne. 2020 Mar;41(3):168-179. doi: 10.1016/j.revmed.2019.12.003. Epub 2020 Jan 7.
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Pathogenesis, disease course, and prognosis of adult-onset Still's disease: an update and review.成人斯蒂尔病的发病机制、疾病过程和预后:更新与综述。
Chin Med J (Engl). 2019 Dec 5;132(23):2856-2864. doi: 10.1097/CM9.0000000000000538.
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Current Management of Acute Pulmonary Embolism.急性肺栓塞的当前管理
Ann Thorac Cardiovasc Surg. 2020 Apr 20;26(2):65-71. doi: 10.5761/atcs.ra.19-00158. Epub 2019 Oct 5.
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Acute pulmonary embolism: a concise review of diagnosis and management.急性肺栓塞:诊断与管理简述。
Intern Med J. 2019 Jan;49(1):15-27. doi: 10.1111/imj.14145.
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Adult-Onset Still's Disease: Still a Serious Health Problem (a Case Report and Literature Review).成人斯蒂尔病:仍然是一个严重的健康问题(病例报告及文献综述)
Am J Case Rep. 2017 Feb 3;18:119-124. doi: 10.12659/ajcr.901846.
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Adult Onset Still's Disease: A Case Report with a Rare Clinical Manifestation and Pathophysiological Correlations.成人斯蒂尔病:一例罕见临床表现及病理生理相关性的病例报告
Case Rep Med. 2013;2013:981232. doi: 10.1155/2013/981232. Epub 2013 Jan 17.