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婴儿甲基丙二酸血症和同型胱氨酸尿症所致溶血尿毒综合征:病例报告及文献复习

Hemolytic Uremic Syndrome Due to Methylmalonic Acidemia and Homocystinuria in an Infant: A Case Report and Literature Review.

作者信息

Karava Vasiliki, Kondou Antonia, Dotis John, Sotiriou Georgia, Gerou Spyridon, Michelakakis Helen, Vargiami Euthymia, Economou Marina, Zafeiriou Dimitrios, Printza Nikoleta

机构信息

1st Department of Pediatrics, Aristotle University of Thessaloniki, Hippokratio General Hospital, 546 42 Thessaloniki, Greece.

Gerou Analysis Medical S.A., Diagnostic-Research Clinics, 546 22 Thessaloniki, Greece.

出版信息

Children (Basel). 2021 Feb 5;8(2):112. doi: 10.3390/children8020112.

DOI:10.3390/children8020112
PMID:33562640
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7915400/
Abstract

Methylmalonic acidemia and homocystinuria cobalamin C (cblC) type is the most common inborn error of the intracellular cobalamin metabolism, associated with multisystem involvement and high mortality rates, especially in the early-onset form of the disease. Hemolytic uremic syndrome (HUS) is a rare manifestation and needs to be distinguished from other causes of renal thrombotic microangiopathy. We describe a case of a 3-month-old infant, with failure to thrive, hypotonia and pallor, who developed HUS in the setting of cblC deficit, along with dilated cardiomyopathy, and presented delayed response to optic stimulation in visual evoked potentials, as well as enlarged bilateral subarachnoid spaces and delayed myelination in brain magnetic resonance imaging. Renal damage was reversed, while neurodevelopmental profile and eye contact improved after supplementation with parenteral hydroxycobalamin, oral folic acid, betaine and levocarnitine. Homozygous mutation of c.271dupA in the MMACHC gene was ultimately detected. In this report, we highlight the diagnostic challenges as well as the significance of early recognition and multidisciplinary management of this unusual condition. A brief review of published case reports of early-onset cblC deficit and related HUS is depicted, pointing out the initial clinical presentation, signs of renal damage and outcome, MMACHC gene type of mutations and accompanying extra-renal manifestations.

摘要

甲基丙二酸血症和同型胱氨酸尿症钴胺素C(cblC)型是细胞内钴胺素代谢最常见的先天性缺陷,与多系统受累和高死亡率相关,尤其是在疾病的早发型中。溶血性尿毒症综合征(HUS)是一种罕见表现,需要与肾血栓性微血管病的其他病因相鉴别。我们描述了一例3个月大的婴儿,有发育不良、肌张力低下和面色苍白,在cblC缺乏的情况下发生了HUS,同时伴有扩张型心肌病,视觉诱发电位对视刺激反应延迟,脑磁共振成像显示双侧蛛网膜下腔增宽和髓鞘形成延迟。补充胃肠外羟钴胺素、口服叶酸、甜菜碱和左卡尼汀后,肾损伤得到逆转,神经发育情况和眼神交流得到改善。最终检测到MMACHC基因中c.271dupA的纯合突变。在本报告中,我们强调了这种罕见疾病的诊断挑战以及早期识别和多学科管理的重要性。简要回顾了已发表的早发型cblC缺乏及相关HUS的病例报告,指出了初始临床表现、肾损伤体征和结局、MMACHC基因突变类型及伴随的肾外表现。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5501/7915400/f21256e17854/children-08-00112-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5501/7915400/86ee6a891754/children-08-00112-g002a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5501/7915400/9a8b8a7dd4d2/children-08-00112-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5501/7915400/f21256e17854/children-08-00112-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5501/7915400/86ee6a891754/children-08-00112-g002a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5501/7915400/9a8b8a7dd4d2/children-08-00112-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5501/7915400/f21256e17854/children-08-00112-g001.jpg

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BMC Nephrol. 2020 Mar 12;21(1):96. doi: 10.1186/s12882-020-01748-2.
2
Do not Miss Rare and Treatable Cause of Early-Onset Hemolytic Uremic Syndrome: Cobalamin C Deficiency.勿漏诊早发性溶血尿毒综合征的罕见且可治疗病因:钴胺素 C 缺乏症。
Nephron. 2019;142(3):258-263. doi: 10.1159/000497822. Epub 2019 May 28.
3
Hemolytic uremic syndrome with dual caution in an infant: cobalamin C defect and complement dysregulation successfully treated with eculizumab.
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Zhejiang Da Xue Xue Bao Yi Xue Ban. 2021 Aug 25;50(4):436-443. doi: 10.3724/zdxbyxb-2021-0276.
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Case Report: Lipoprotein Glomerulopathy Complicated by Atypical Hemolytic Uremic Syndrome.病例报告:脂蛋白肾小球病合并非典型溶血尿毒症综合征
Front Med (Lausanne). 2021 Jun 2;8:679048. doi: 10.3389/fmed.2021.679048. eCollection 2021.
婴儿溶血尿毒症综合征伴双重警示:维生素 B12 缺陷和补体失调经依库珠单抗治疗成功。
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