Elliott Ross P, Freeman Brian P, Meier Jeffery L, El-Herte Rima
Internal Medicine Residency Program, MercyOne Medical Center and Clinics, Des Moines, IA 50314, USA.
Mission Cancer and Blood, Des Moines, IA 50309, USA.
Case Rep Infect Dis. 2022 Jul 8;2022:7949471. doi: 10.1155/2022/7949471. eCollection 2022.
Primary cytomegalovirus (CMV) infection of the immunocompetent host usually produces little-to-no illness. Occasionally, the infection results in mononucleosis syndrome, protracted fever, hepatitis, tissue-invasive disease, or Guillain-Barré syndrome. Hemolytic anemia and hemophagocytic lymphohistiocytosis (HLH) are rare complications that have not been reported to co-occur. Having hemolytic anemia in conjunction with more common findings of fever and hepatitis complicates the diagnosis of HLH. . A 34-year-old male with previously good health presented with a prolonged febrile illness, jaundice, and anemia. An extensive work-up during hospitalization revealed intravascular hemolytic anemia, leukopenia, hepatosplenomegaly, and biopsy evidence of extensive lymphohistiocytic infiltration of the liver with microgranulomata and sinusoidal hemophagocytosis. Soluble CD25 level was mildly elevated at 1200.3 pg/mL and the HScore calculation (fever, bicytopenia, hepatosplenomegaly, aspartate aminotransaminase 99 IU/L, ferritin 1570 ng/mL, fibrinogen 488 mg/dL, and triglycerides 173 mg/dL) suggested a moderate probability of reactive HLH. Primary CMV infection was diagnosed based on CMV IgM positivity, low CMV IgG avidity index, and low-grade CMV DNAemia. The CMV antigen was not detected in the liver biopsy, and the bone marrow biopsy was unremarkable. The illness began to improve before he received oral valganciclovir for 5 days, and he was in good health 10 months later.
Acute CMV illness in an immunocompetent adult can present with hemolytic anemia and clinicopathologic abnormalities consistent with a form fruste of HLH. The illness is likely due to an excessive or unbalanced immune response that may self-correct.
免疫功能正常宿主的原发性巨细胞病毒（CMV）感染通常很少或几乎不引起疾病。偶尔，感染会导致单核细胞增多症综合征、持续性发热、肝炎、组织侵袭性疾病或吉兰 - 巴雷综合征。溶血性贫血和噬血细胞性淋巴组织细胞增生症（HLH）是罕见的并发症，尚未有两者同时发生的报道。溶血性贫血与更常见的发热和肝炎表现同时出现会使HLH的诊断复杂化。一名34岁既往健康的男性出现持续性发热性疾病、黄疸和贫血。住院期间的广泛检查发现血管内溶血性贫血、白细胞减少、肝脾肿大，肝脏活检有广泛的淋巴组织细胞浸润伴微肉芽肿和窦状隙噬血细胞现象的证据。可溶性CD25水平轻度升高至1200.3 pg/mL，HScore计算（发热、血细胞减少、肝脾肿大、天冬氨酸转氨酶99 IU/L、铁蛋白1570 ng/mL、纤维蛋白原488 mg/dL和甘油三酯173 mg/dL）提示反应性HLH的可能性为中度。基于CMV IgM阳性、低CMV IgG亲和力指数和低水平CMV血症诊断为原发性CMV感染。肝脏活检未检测到CMV抗原，骨髓活检无异常。在他接受口服缬更昔洛韦5天之前病情开始好转，10个月后他身体健康。
免疫功能正常的成年人急性CMV疾病可表现为溶血性贫血和与HLH不完全型一致的临床病理异常。该疾病可能是由于过度或失衡的免疫反应，可能会自我纠正。
