BACKGROUND Infections with multidrug-resistant organisms (MDRO) pose a serious threat to patients with dysregulated immunity such as in hemophagocytic lymphohistiocytosis (HLH), but such infections have rarely been comprehensively characterized. Here, we present a fatal case of HLH secondary to cytomegalovirus (CMV) infection complicated by both anti-viral drug resistance and sepsis from multiple MDROs including pandrug-resistant superbug bacteria. CASE PRESENTATION A previously healthy, six-year-old boy presented with a 45-day history of fever prior to a diagnosis of hemophagocytic lymphohistiocytosis and hemorrhagic colitis, both associated with CMV. On hospital admission, the patient was found to be colonized with multiple, multidrug-resistant (MDR) bacteria including vancomycin-resistant enterococci (VRE) and carbapenamase-producing organisms (CPO). He eventually developed respiratory, urine and bloodstream infections with highly drug-resistant, including pandrug-resistant bacteria, which could not be controlled by antibiotic treatment. Antiviral therapy also failed to contain his CMV infection and the patient succumbed to overwhelming bacterial and viral infection. Whole genome sequencing (WGS) of the MDR bacteria and metagenomic analysis of his blood sample revealed an unusual accumulation of a wide range of antimicrobial resistance mechanisms in a single patient, including antiviral resistance to ganciclovir, and resistance mechanisms to all currently available antibiotics. CONCLUSIONS The case highlights both the risk of acquiring MDR superbugs and the severity of these infections in HLH patients.

中文翻译：

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巨细胞病毒相关的噬血细胞性淋巴组织细胞增生症患者中多种抗生素耐药机制的异常积累：一例。

背景技术具有多重耐药性生物体（MDRO）的感染对免疫功能失调的患者例如在吞噬细胞的淋巴组织细胞增生症（HLH）中构成严重威胁，但是这种感染很少被全面表征。在这里，我们介绍了由巨细胞病毒（CMV）感染继发的HLH致命病例，并伴有抗病毒药物耐药性和来自多个MDRO（包括耐药性超级细菌）的败血症。病例介绍一个先前健康的六岁男孩在诊断为噬血细胞性淋巴组织细胞增生和出血性结肠炎之前均出现了45天的发烧史，两者均与CMV相关。入院时发现该患者被多处定植，耐多药（MDR）细菌，包括耐万古霉素的肠球菌（VRE）和产生碳青霉酶的生物（CPO）。他最终发展出对呼吸道，尿道和血流的感染，并具有高度耐药性，包括耐泛药性细菌，而抗生素治疗无法控制这种细菌。抗病毒治疗也未能遏制他的CMV感染，患者屈服于细菌和病毒感染。MDR细菌的全基因组测序（WGS）和他的血液样本的宏基因组分析显示，单个患者中广泛的抗菌素耐药性机制异常积累，包括对更昔洛韦的抗病毒耐药性以及对目前所有可用抗生素的耐药性机制。