Belisa Telila K, Haile Asteway M, Mesfin Getinet B, Mengistie Biruk T, Mengistie Chernet T, Haile Bezawit M
School of Medicine, College of Health Sciences, Addis Ababa University, Addis Ababa, Ethiopia.
Department of Pediatrics and Child Health, College of Health Sciences, Addis Ababa University, Addis Ababa, Ethiopia.
IDCases. 2024 Aug 12;37:e02059. doi: 10.1016/j.idcr.2024.e02059. eCollection 2024.
Hemophagocytic lymphohistiocytosis (HLH) is a rare, potentially life-threatening clinical syndrome characterized by hyperactivation of inflammatory mediators and harmful end-organ damage Visceral leishmaniasis (VL)-induced HLH is a rare disease with significant diagnostic and management implications. Herein, we present a case of secondary HLH as a complication of visceral leishmaniasis in a two-year-old toddler. A 2-year-old male toddler was admitted for evaluation of a prolonged 4-week fever. Accompanying the fever, he developed progressive abdominal swelling, intermittent bilateral nasal bleeding, and repeated chest-focus infections of similar duration. The patient was acutely sick, with chronic signs of malnutrition (mid-upper arm circumference of 10.5 cm), fever (39 °C), tachypnea (70 breaths/min), tachycardia (132 beats/min), pallor, and hepatosplenomegaly. Initial investigation revealed leukopenia (2240/μl), anemia (7.3 g/dl), and severe thrombocytopenia (26,000/μl). With consideration of febrile neutropenia, the patient was started on cefepime with further revision to vancomycin and meropenem based on the culture result. After 10 days of persistent fever and poor clinical condition, an immunochromatographic rapid test with the rK39 antigen was conducted, and the patient was found to be positive for spp. Intravenous liposomal amphotericin B (AmBisome) was initiated. On the 6th day of treatment, the patient's clinical and laboratory profiles severely deteriorated, and further laboratory investigation showed elevated triglyceride (524 mg/dl) and ferritin levels (1500 ng/mL). VL-induced secondary HLH was confirmed, and intravenous dexamethasone was initiated. Subsequently, his clinical and laboratory findings significantly improved, and he was discharged with PO dexamethasone. Our case highlights the intricate nature of VL-induced HLH and the need for high index of suspicion and timely management.
噬血细胞性淋巴组织细胞增生症（HLH）是一种罕见的、可能危及生命的临床综合征，其特征为炎症介质过度激活和有害的终末器官损伤。内脏利什曼病（VL）诱发的HLH是一种罕见疾病，对诊断和治疗具有重要意义。在此，我们报告一例两岁幼儿因内脏利什曼病并发继发性HLH的病例。一名2岁男童因持续4周发热入院评估。发热的同时，他出现进行性腹部肿胀、间歇性双侧鼻出血以及持续时间相似的反复胸部感染。该患者病情严重，有慢性营养不良体征（上臂中部周长10.5厘米）、发热（39℃）、呼吸急促（70次/分钟）、心动过速（132次/分钟）、面色苍白以及肝脾肿大。初步检查显示白细胞减少（2240/μl）、贫血（7.3 g/dl）和严重血小板减少（26,000/μl）。考虑到发热性中性粒细胞减少症，患者开始使用头孢吡肟治疗，随后根据培养结果改为万古霉素和美罗培南。在持续发热10天且临床状况不佳后，进行了rK39抗原免疫层析快速检测，结果显示该患者 spp. 呈阳性。开始静脉注射脂质体两性霉素B（安必素）。治疗第6天，患者的临床和实验室检查结果严重恶化，进一步实验室检查显示甘油三酯升高（524 mg/dl）和铁蛋白水平升高（1500 ng/mL）。确诊为VL诱发的继发性HLH，开始静脉注射地塞米松。随后，他的临床和实验室检查结果显著改善，出院时口服地塞米松。我们的病例突出了VL诱发HLH的复杂性，以及高度怀疑和及时治疗的必要性。
