芬兰型先天性肾病综合征NPSH1致围产期卒中及脑静脉血栓1例

IF 1.2 4区 医学 Q4 CLINICAL NEUROLOGY
Bregje O van Oldenmark, Vivianne E H J Wintjens, Menno J P Toirkens, Roos W G van Rooij-Kouwenhoven, Enrico Lopriore, Linda S de Vries, Sylke J Steggerda
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引用次数: 0

摘要

先天性肾病综合征(CNS)是一种新生儿严重的肾脏疾病,以蛋白尿、低白蛋白血症和高凝血症等并发症为特征。众所周知,随着时间的推移,中枢神经系统易使患者发生血栓形成,但据我们所知,出生后第一周内发生脑静脉血栓形成(CSVT)等脑血管并发症在确诊中枢神经系统的新生儿中以前很少有报道。我们在这里提出一个婴儿,出生正常阴道分娩是复杂的大胎盘潴留。她第一次入院的第三天,口腔周围发绀和红细胞增多症。她出现呼吸暂停,后来经aEEG证实为癫痫发作,并发现多种血栓性并发症,包括广泛的CSVT和双侧丘脑出血。血清白蛋白极低,尿白蛋白水平高,怀疑为芬兰型CNS, NPHS1致病变异p.Cys623Phe和p.Asn870Profs*36证实了这一点。尽管进行了部分交换输血和抗凝治疗,婴儿仍出现了严重的大脑异常。该病例强调了对大胎盘、严重红细胞增多症、蛋白尿和低白蛋白血症的新生儿考虑中枢神经系统的重要性,因为他们可能有发生CSVT的风险。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Perinatal Stroke and Cerebral Sinovenous Thrombosis Caused by Congenital Nephrotic Syndrome NPSH1 (Finnish Type): A Case Report.

Congenital nephrotic syndrome (CNS) is a severe renal disorder in newborns, characterized by complications such as albuminuria, hypoalbuminemia, and hypercoagulability. While CNS is known to predispose patients to thrombosis over time, to our knowledge, cerebrovascular complications such as cerebral sinovenous thrombosis (CSVT) within the first week after birth have rarely been reported before in neonates with confirmed CNS. We present here an infant, born by normal vaginal delivery, which was complicated by the retention of a large placenta. She was first admitted on day 3 with perioral cyanosis and polycythemia. She developed apneas that were later confirmed with amplitude integrated EEG to be seizures and was found to have multiple thrombotic complications, including extensive CSVT and bilateral thalamic hemorrhages. Serum albumin level was very low, with high urinary levels suspicious for Finnish-type CNS, which was confirmed by NPHS1 pathogenic variants p.Cys623Phe and p.Asn870Profs*36. Despite partial exchange transfusions and anticoagulation therapy, the infant developed severe cerebral abnormalities. This case underscores the importance of considering CNS in neonates with a large placenta, severe polycythemia, proteinuria, and hypoalbuminemia, as they may be at risk of developing CSVT.

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来源期刊
Neuropediatrics
Neuropediatrics 医学-临床神经学
CiteScore
2.80
自引率
0.00%
发文量
94
审稿时长
>12 weeks
期刊介绍: For key insights into today''s practice of pediatric neurology, Neuropediatrics is the worldwide journal of choice. Original articles, case reports and panel discussions are the distinctive features of a journal that always keeps abreast of current developments and trends - the reason it has developed into an internationally recognized forum for specialists throughout the world. Pediatricians, neurologists, neurosurgeons, and neurobiologists will find it essential reading.
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