Visceral leishmaniasis complicated with hemophagocytic lymphohistiocytosis and resistant to amphotericin B: a case report

IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Journal of Medical Case Reports Pub Date : 2024-09-10 DOI:10.1186/s13256-024-04760-4
Muhamad Zakaria Brimo Alsaman, Fares Abu Sultan, Yazan Ramadan, Khaled Arnaout, Mohamad Shahrour, Bilal Barakat, Abeer Dayeh
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Abstract

Hemophagocytic lymphohistiocytosis characterized by hemophagocytosis leading to uncontrolled inflammation; the most common etiology in secondary cases of hemophagocytic lymphohistiocytosis is viral infections, especially Epstein–Barr virus. Visceral leishmaniasis is a vectorborne protozoal disease caused by Leishmania donovani complex. It is common in tropical and subtropical regions, with 50,000–90,000 new cases annually. A 15-month-old Arab female was admitted to our hospital with 15 days of fever and decreased weight. On clinical examination, she had a markedly enlarged liver and spleen that were palpable 4 cm and 6 cm below the costal margin, respectively. The peripheral blood smear showed hypochromic microcytic anemia, poikilocytosis, reactive lymphocytosis, and mild thrombocytopenia. Bone marrow aspiration did not show malignancy or any other pathological findings. The patient was put on antibiotic therapy without improvement. Repeated bone marrow aspiration showed erythrophagocytosis; intracellular small round organisms looked like the amastigote form of Leishmania (Donovan bodies) with no evidence of malignancies. Her lab values showed ferritin greater than 500 ug/L, pancytopenia, and hypertriglyceridemia. The patient was diagnosed with hemophagocytic lymphohistiocytosis secondary to visceral leishmaniasis. Hemophagocytic lymphohistiocytosis secondary to visceral leishmaniasis is an extensively rare phenomenon in the medical literature that causes challenges in diagnosis and management. Steroids should be used wisely to not cover the symptoms of infections or malignancy, and amphotericin B resistance should be kept in mind in unresponsive Leishmania cases.
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内脏利什曼病并发嗜血细胞淋巴组织细胞增多症,对两性霉素 B 耐药:病例报告
嗜血细胞淋巴组织细胞增多症的特点是嗜血细胞增多导致炎症失控;继发性嗜血细胞淋巴组织细胞增多症最常见的病因是病毒感染,尤其是 Epstein-Barr 病毒。内脏利什曼病是由多诺万利什曼原虫复合体引起的病媒传播的原虫病。它常见于热带和亚热带地区,每年新增病例 50 000 至 90 000 例。一名 15 个月大的阿拉伯女童因发烧和体重下降 15 天而被送入我院。经临床检查,她的肝脏和脾脏明显肿大,分别可触及肋缘下 4 厘米和 6 厘米处。外周血涂片显示低色素性小细胞性贫血、粒细胞增多、反应性淋巴细胞增多和轻度血小板减少。骨髓穿刺未发现恶性肿瘤或任何其他病理结果。患者接受了抗生素治疗,但病情未见好转。反复骨髓穿刺显示红细胞吞噬症;细胞内的小圆生物看起来像利什曼原虫(多诺万体),但没有恶性肿瘤的迹象。她的化验值显示铁蛋白超过 500 微克/升、泛血细胞减少和高甘油三酯血症。患者被诊断为继发于内脏利什曼病的嗜血细胞淋巴组织细胞增多症。继发于内脏利什曼病的嗜血细胞淋巴组织细胞增多症在医学文献中非常罕见,给诊断和治疗带来了挑战。应明智使用类固醇,以免掩盖感染或恶性肿瘤的症状,对于无反应的利什曼病例,应注意两性霉素 B 的耐药性。
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来源期刊
Journal of Medical Case Reports
Journal of Medical Case Reports Medicine-Medicine (all)
CiteScore
1.50
自引率
0.00%
发文量
436
期刊介绍: JMCR is an open access, peer-reviewed online journal that will consider any original case report that expands the field of general medical knowledge. Reports should show one of the following: 1. Unreported or unusual side effects or adverse interactions involving medications 2. Unexpected or unusual presentations of a disease 3. New associations or variations in disease processes 4. Presentations, diagnoses and/or management of new and emerging diseases 5. An unexpected association between diseases or symptoms 6. An unexpected event in the course of observing or treating a patient 7. Findings that shed new light on the possible pathogenesis of a disease or an adverse effect
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