From the Department of Paediatrics, Imperial College Health Trust, London, United Kingdom.
Centre for Clinical Research in Infectious Diseases & Sexual Health, University College London, London, United Kingdom.
Pediatr Infect Dis J. 2020 Apr;39(4):336-338. doi: 10.1097/INF.0000000000002583.
Congenital cytomegalovirus (CCMV) accounts for high rates of infant morbidity and mortality. Neutropenia is a common finding in CCMV infection, of which the age of presentation overlaps with autoimmune neutropenia (AIN). AIN represents one of the most common forms of chronic neutropenia in childhood.
A literature search exploring biologic associations between CCMV and AIN was conducted: PubMed (MEDLINE), Ovid and Web of Science. We further describe 2 cases of concurrent CCMV and AIN. Both cases were confirmed with the indirect granulocyte immunofluorescence test and alternative etiologies for neutropenia excluded.
Our 2 patients represent confirmed cases of AIN in infants with CCMV. One patient demonstrated neutropenia while undergoing treatment with Valganciclovir, while the other was never treated. With interruption of Valganciclovir in infant A, neutrophil counts (ANC) did not improve and upon resumption of treatment ANC remained static.
Further studies examining a possible biologic link between CCMV and AIN are advocated for. We encourage clinicians to actively consider AIN in the differential diagnosis of all infants with CCMV presenting with neutropenia.
先天性巨细胞病毒(cytomegalovirus,CCMV)可导致较高的婴儿发病率和死亡率。中性粒细胞减少症是 CCMV 感染的常见表现,其发病年龄与自身免疫性中性粒细胞减少症(autoimmune neutropenia,AIN)重叠。AIN 是儿童慢性中性粒细胞减少症最常见的形式之一。
我们进行了一项文献检索,以探讨 CCMV 与 AIN 之间的生物学关联:PubMed(MEDLINE)、Ovid 和 Web of Science。我们进一步描述了 2 例同时存在 CCMV 和 AIN 的病例。这两个病例均通过间接粒细胞免疫荧光试验得到证实,并排除了中性粒细胞减少症的其他病因。
我们的 2 例患者均为患有 CCMV 的婴儿的 AIN 确诊病例。1 例患者在接受缬更昔洛韦治疗时出现中性粒细胞减少症,而另 1 例从未接受过治疗。在婴儿 A 中断缬更昔洛韦治疗后,中性粒细胞计数(ANC)并未改善,且在恢复治疗后 ANC 保持不变。
我们提倡进一步研究以探讨 CCMV 和 AIN 之间可能存在的生物学联系。我们鼓励临床医生在所有患有 CCMV 并伴有中性粒细胞减少症的婴儿的鉴别诊断中积极考虑 AIN。