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成人斯蒂尔病合并噬血细胞性淋巴组织细胞增生症及多器官功能衰竭导致的致命性急性肠梗阻:1例罕见病例报告

Fatal Acute Intestinal Obstruction with Hemophagocytic Lymphohistiocytosis and Multiple Organ Failure in Adult-Onset Still's Disease: A Rare Case Report.

作者信息

Li Kun, Pan Xuejia, Guo Hongyu, Jiang Saiping, Fang Xueling

机构信息

Department of Critical Care Medicine, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang Province, People's Republic of China.

Department of Nursing, Hangzhou Xiaoying Community Health Service Center, Hangzhou, Zhejiang Province, People's Republic of China.

出版信息

J Inflamm Res. 2025 Apr 16;18:5161-5171. doi: 10.2147/JIR.S509898. eCollection 2025.

DOI:10.2147/JIR.S509898
PMID:40255663
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12009564/
Abstract

BACKGROUND

Adult-onset Still's disease (AOSD) is a systemic autoinflammatory disorder characterized by unpredictable multi-organ involvement. Although gastrointestinal complications are uncommon in AOSD, they can be life-threatening and present significant diagnostic and management challenges.

CASE SUMMARY

We report the case of a 68-year-old man with AOSD who developed acute intestinal obstruction, a rare and critical complication. Imaging revealed significant colonic wall thickening, with a maximum thickness of 2.6 cm on contrast-enhanced computed tomography. The clinical status of the patient deteriorated, further complicated by the onset of hemophagocytic lymphohistiocytosis (HLH) and multi-organ failure, including acute renal dysfunction. Despite receiving intensive care and aggressive treatment, including supportive measures and immunosuppressive therapy, the patient succumbed to his illness.

CONCLUSION

This case underscores the importance of recognizing rare gastrointestinal and systemic complications in patients with AOSD. Early identification and prompt multidisciplinary management of conditions such as HLH and acute intestinal obstruction are essential for improving outcomes in such critical scenarios.

摘要

背景

成人斯蒂尔病(AOSD)是一种系统性自身炎症性疾病,其特征是多器官受累且不可预测。虽然胃肠道并发症在AOSD中并不常见,但可能危及生命,并带来重大的诊断和管理挑战。

病例摘要

我们报告了一例68岁患有AOSD的男性患者,该患者出现了急性肠梗阻,这是一种罕见且严重的并发症。影像学检查显示结肠壁显著增厚,在增强计算机断层扫描上最大厚度为2.6厘米。患者的临床状况恶化,因噬血细胞性淋巴组织细胞增生症(HLH)和多器官功能衰竭(包括急性肾功能不全)的发生而进一步复杂化。尽管接受了重症监护和积极治疗,包括支持措施和免疫抑制治疗,但患者仍因病死亡。

结论

该病例强调了认识AOSD患者罕见胃肠道和全身并发症的重要性。早期识别和及时对HLH和急性肠梗阻等病症进行多学科管理对于改善此类危急情况下的治疗结果至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d2a/12009564/ed53d39d9642/JIR-18-5161-g0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d2a/12009564/ed53d39d9642/JIR-18-5161-g0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d2a/12009564/ed53d39d9642/JIR-18-5161-g0001.jpg

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本文引用的文献

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EULAR/PReS recommendations for the diagnosis and management of Still's disease, comprising systemic juvenile idiopathic arthritis and adult-onset Still's disease.EULAR/PReS 关于斯蒂尔病(包括全身型幼年特发性关节炎和成人斯蒂尔病)的诊断和治疗建议。
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The 2022 EULAR/ACR points to consider at the early stages of diagnosis and management of suspected haemophagocytic lymphohistiocytosis/macrophage activation syndrome (HLH/MAS).
2022 年 EULAR/ACR 在疑似噬血细胞性淋巴组织细胞增生症/巨噬细胞活化综合征(HLH/MAS)的早期诊断和管理中需要考虑的要点。
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Duodenojejunal inflammation causing chronic vomiting in adult-onset Still's disease.成人Still 病中十二指肠炎引起的慢性呕吐。
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Adult-Onset Still's Disease: Clinical Aspects and Therapeutic Approach.成人起病型斯蒂尔病:临床特征与治疗方法
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Human Cytomegalovirus and Autoimmune Diseases: Where Are We?人巨细胞病毒与自身免疫性疾病:我们处于何处?
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A Rare Case of Cytomegalovirus in the Gut in an Immunocompetent Host With Ischemic Colitis.免疫功能正常的缺血性结肠炎宿主肠道巨细胞病毒感染1例罕见病例
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