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自身免疫性溶血性贫血——沙特女性布鲁氏菌病罕见并发症:病例报告及文献复习。

Autoimmune Hemolytic Anemia - Rare Complication of Brucellosis in a Saudi Woman: A Case Report and Literature Review.

机构信息

Department of Adult Cardiology, King Fahd Armed Forces Hospital, Jeddah, Saudi Arabia.

Department of Internal Medicine, King Fahad Armed Forces Hospital, Jeddah, Saudi Arabia.

出版信息

Am J Case Rep. 2022 Feb 25;23:e935187. doi: 10.12659/AJCR.935187.

DOI:10.12659/AJCR.935187
PMID:35210389
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8886510/
Abstract

BACKGROUND Autoimmune hemolytic anemia and immune thrombocytopenia are rare complications of brucellosis; only a few cases have been reported in the literature. While pancytopenia is common and was reported in Saudi Arabia, the description of autoimmune hemolytic anemia or immune thrombocytopenia has not yet been reported in the kingdom. Hematological complication usually requires supportive treatment, and it is expected to improve with the initiation of antimicrobial therapy for brucellosis. There are few reports on the treatment of patients that fail to improve with conventional therapy. CASE REPORT A 46-year-old previously healthy Saudi woman was admitted to our hospital after multiple visits to the emergency department with chief concerns of fever and fatigability for 30 days. The examination was remarkable only for fever of 38.4°C and tender hepatomegaly. Laboratory tests upon admission were significant of pancytopenia, with a white blood count of 3×10⁹/L, hemoglobin of 8.1 g/dL, platelet of 13×10⁹/L, moderate hyponatremia, hypokalemia, and metabolic acidosis. Tuberculosis was ruled out and pan-sensitive brucellosis was diagnosed. She was started on standard antimicrobial therapy without significant improvement. Further testing revealed Coomb's-positive hemolytic anemia and possible immune-mediated severe thrombocytopenia. She was treated with glucocorticoids and intravenous immunoglobulin, with dramatic response. CONCLUSIONS Autoimmune-mediated destruction of blood lines in brucellosis is rare. It should be sought as a potential diagnosis in case of persistent anemia and/or thrombocytopenia that is severe or fails to improve with proper antimicrobial coverage. Early involvement of hematologists and initiation of glucocorticoid with or without intravenous immunoglobulin is crucial.

摘要

背景

自身免疫性溶血性贫血和免疫性血小板减少症是布鲁氏菌病的罕见并发症;文献中仅报道了少数病例。虽然全血细胞减少症很常见,且在沙特阿拉伯已有报道,但在该国尚未报道自身免疫性溶血性贫血或免疫性血小板减少症的描述。血液学并发症通常需要支持治疗,预计随着开始针对布鲁氏菌病的抗菌治疗,它会得到改善。对于那些常规治疗未能改善的患者,很少有关于其治疗的报告。

病例报告

一名 46 岁的沙特籍既往健康女性,因发热和乏力 30 天多次就诊急诊科后被收入我院。检查仅发现发热(38.4°C)和肝大伴触痛。入院时的实验室检查显著提示全血细胞减少,白细胞计数为 3×10⁹/L,血红蛋白为 8.1 g/dL,血小板为 13×10⁹/L,中度低钠血症、低钾血症和代谢性酸中毒。排除了结核病,诊断为泛敏性布鲁氏菌病。她开始接受标准抗菌治疗,但无明显改善。进一步检查显示 Coomb's 阳性溶血性贫血和可能由免疫介导的严重血小板减少症。她接受了糖皮质激素和静脉免疫球蛋白治疗,反应明显。

结论

布鲁氏菌病中血液系统的自身免疫性破坏较为罕见。在出现持续性贫血和/或血小板减少症且严重或经适当抗菌覆盖治疗后无改善时,应将其作为潜在诊断进行寻找。早期由血液科医生参与并开始使用糖皮质激素联合或不联合静脉免疫球蛋白至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f7b6/8886510/1cecdcaa8930/amjcaserep-23-e935187-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f7b6/8886510/b5828b4bfcb3/amjcaserep-23-e935187-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f7b6/8886510/5862cd71c799/amjcaserep-23-e935187-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f7b6/8886510/1cecdcaa8930/amjcaserep-23-e935187-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f7b6/8886510/b5828b4bfcb3/amjcaserep-23-e935187-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f7b6/8886510/5862cd71c799/amjcaserep-23-e935187-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f7b6/8886510/1cecdcaa8930/amjcaserep-23-e935187-g003.jpg

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本文引用的文献

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Cureus. 2020 May 16;12(5):e8154. doi: 10.7759/cureus.8154.
2
Sero-prevalence and risk factors of brucellosis among suspected febrile patients attending a referral hospital in southern Saudi Arabia (2014-2018).沙特阿拉伯南部转诊医院疑似发热患者布鲁氏菌病的血清流行率及危险因素(2014-2018 年)。
BMC Infect Dis. 2020 Jan 9;20(1):26. doi: 10.1186/s12879-020-4763-z.
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Epidemiology of Brucellosis in Saudi Arabia.沙特阿拉伯布鲁氏菌病的流行病学
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Acute Brucellosis Presenting as an Autoimmune Hemolytic Anemia.表现为自身免疫性溶血性贫血的急性布鲁氏菌病
Case Rep Infect Dis. 2018 Oct 25;2018:1030382. doi: 10.1155/2018/1030382. eCollection 2018.
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Cytopenia in adult brucellosis patients.成人布氏杆菌病患者的血细胞减少症。
Indian J Med Res. 2018 Jan;147(1):73-80. doi: 10.4103/ijmr.IJMR_542_15.
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Brucellosis accompanied by haemophagocytic lymphohistiocytosis and multiple splenic abscesses in a patient with depression.一名抑郁症患者出现布鲁氏菌病伴噬血细胞性淋巴组织细胞增生症和多发性脾脓肿。
BMJ Case Rep. 2018 Mar 27;2018:bcr-2017-224018. doi: 10.1136/bcr-2017-224018.
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