Eculizumab时代的新生儿非典型溶血性尿毒症综合征。

IF 0.8 Q4 PEDIATRICS
AJP Reports Pub Date : 2021-04-01 Epub Date: 2021-06-23 DOI:10.1055/s-0041-1731057
Sara Madureira Gomes, Rita Pissarra Teixeira, Gustavo Rocha, Paulo Soares, Hercilia Guimaraes, Paulo Santos, Joana Jardim, João Luís Barreira, Helena Pinto
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引用次数: 0

摘要

新生儿非典型溶血性尿毒症综合征(aHUS)是一种罕见的疾病,发病率高。Eculizumab被认为是大龄儿童的一线药物,但未被批准用于新生儿和体重低于5公斤的儿童。我们报告一名5天大的女婴,双胎妊娠36周,因脐带脱垂急诊剖宫产,出生体重为2035 g, Apgar评分为7/7/7,出现微血管病性溶血性贫血、血小板减少和进行性急性肾功能衰竭。在诊断出aHUS后的第5天,开始每天输注新鲜冷冻血浆,血小板减少症得到改善,肾功能有非常轻微的改善。病因学研究(先天性感染、志贺毒素、ADAMTS13活性、定向代谢研究)正常。C3c略有下降。在维持贫血和严重肾衰竭的第16天,她开始使用300mg /剂量的eculizumab。10周后贫血消退,13周后肌酐恢复正常。基因研究是正常的。在这种情况下,eculizumab在控制微血管病变和恢复肾功能方面是有效的。新生儿aHUS的诊断可能具有挑战性,因为其表型异质性和与其他可能混淆其表现的潜在重叠,如围产期窒息或败血症/弥漫性血管内凝血。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Neonatal Atypical Hemolytic Uremic Syndrome in the Eculizumab Era.

Neonatal Atypical Hemolytic Uremic Syndrome in the Eculizumab Era.

The atypical hemolytic uremic syndrome (aHUS) in the newborn is a rare disease, with high morbidity. Eculizumab, considered a first-line drug in older children, is not approved in neonates and in children weighing less than 5 kg. We present a 5-day-old female newborn, born at 36 weeks' twin gestation, by emergency cesarean section due to cord prolapse, with birth weight of 2,035 g and Apgar score of 7/7/7, who develops microangiopathic hemolytic anemia, thrombocytopenia, and progressive acute renal failure. In day 5, after diagnosis of aHUS, a daily infusion of fresh frozen plasma begins, with improvement of thrombocytopenia and very slight improvement in renal function. The etiologic study (congenital infection, Shiga toxin, ADAMTS13 activity, directed metabolic study) was normal. C3c was slightly decreased. On day 16 for maintenance of anemia and severe renal failure, she started 300 mg/dose eculizumab. Anemia resolves in 10 weeks and creatinine has normal values after 13 weeks of treatment. The genetic study was normal. In this case, eculizumab is effective in controlling microangiopathy and in the recovery of renal function. Diagnosis of neonatal aHUS can be challenging because of phenotypic heterogeneity and potential overlap with other manifestations that may confound it, such as perinatal asphyxia or sepsis/disseminated intravascular coagulation.

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来源期刊
AJP Reports
AJP Reports PEDIATRICS-
CiteScore
2.20
自引率
0.00%
发文量
30
审稿时长
12 weeks
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