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Treatment of hypophosphatemic rickets in generalized arterial calcification of infancy (GACI) without worsening of vascular calcification.在不加重血管钙化的情况下治疗婴儿全身性动脉钙化(GACI)中的低磷性佝偻病。
Am J Med Genet A. 2016 May;170A(5):1308-11. doi: 10.1002/ajmg.a.37574. Epub 2016 Feb 9.
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3
[Analysis of two cases with idiopathic infantile arterial calcification].
Zhonghua Er Ke Za Zhi. 2014 Nov;52(11):874-6.
4
Heart transplant and 2-year follow up in a child with generalized arterial calcification of infancy.一名患有婴儿期全身性动脉钙化患儿的心脏移植及2年随访
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5
Idiopathic infantile arterial calcification: a possible cause of refractory cardiopulmonary failure in infancy.特发性婴儿动脉钙化:婴儿期难治性心肺衰竭的一个可能原因。
Case Rep Pathol. 2014;2014:189850. doi: 10.1155/2014/189850. Epub 2014 Feb 9.
6
[Idiopathic arterial calcification of infancy:report of a case].
Zhonghua Bing Li Xue Za Zhi. 2013 Nov;42(11):771-2.
7
Mutations in the ABCC6 gene as a cause of generalized arterial calcification of infancy: genotypic overlap with pseudoxanthoma elasticum.ABCC6 基因突变致婴儿型全身动脉钙化:与弹性假黄瘤的基因型重叠。
J Invest Dermatol. 2014 Mar;134(3):658-665. doi: 10.1038/jid.2013.370. Epub 2013 Sep 5.
8
Idiopathic arterial calcification of infancy-- peritoneal dialysis for treatment of hypertension.
Fetal Pediatr Pathol. 2013 Dec;32(6):443-7. doi: 10.3109/15513815.2013.802399. Epub 2013 Jul 24.
9
The mystery of persistent pulmonary hypertension: an idiopathic infantile arterial calcification.持续性肺动脉高压的奥秘:一种特发性婴儿性动脉钙化。
BMC Pediatr. 2013 Jul 16;13:107. doi: 10.1186/1471-2431-13-107.
10
Severe skeletal toxicity from protracted etidronate therapy for generalized arterial calcification of infancy.长期依替膦酸盐治疗婴儿全身动脉钙化导致严重骨骼毒性。
J Bone Miner Res. 2013 Feb;28(2):419-30. doi: 10.1002/jbmr.1752.

[一名早产儿持续两个月的高血压]

[Persistent hypertension for two months in a preterm infant].

作者信息

Liu Yun-Feng, Han Tong-Yan, Tong Xiao-Mei, Wang Jing, Tang Ya-Nan, Cui Li-Gang, Zhu Xiao-Hui, Piao Mei-Hua, Wang Qing-Qing, Wu Hui

机构信息

Department of Pediatrics, Peking University Third Hospital, Beijing 100191, China.

出版信息

Zhongguo Dang Dai Er Ke Za Zhi. 2018 Nov;20(11):939-943. doi: 10.7499/j.issn.1008-8830.2018.11.012.

DOI:10.7499/j.issn.1008-8830.2018.11.012
PMID:30477627
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7389023/
Abstract

A boy aged 2 months (born at 36 weeks of gestation) was admitted due to cough and dyspnea. After admission, he was found to have persistent hypertension, proteinuria, and persistent convulsion, and imaging examination showed extensive calcification of the aorta and major branches and stenosis of local lumens of the abdominal aorta and the right renal artery with increased blood flow velocity. The boy was admitted during the neonatal period due to wet lung and pulmonary arterial hypertension and was found to have hypertension and proteinuria. High-throughput whole-exome sequencing was performed and found two compound heterozygous mutations in the ENPP1 gene from his parents, c.130C>T (p.Q44X) and c.1112A>T (p.Y371F). c.130C>T was a nonsense mutation, which could cause partial deletion of protein from 44 amino acids, and was defined as a primary pathogenic mutation. c.1112A>T was a missense mutation which had been reported as a pathogenic mutation associated with idiopathic infantile arterial calcification (IIAC). Therefore, he was diagnosed with IIAC. He was given phosphonate drugs, antihypertensive drugs, anticonvulsion treatment, and respiratory support. Blood pressure was maintained at the upper limit of normal value. There was no deterioration of arterial calcification. It is concluded that IIAC should be considered for infants with persistent hypertension and extensive vascular calcification, and imaging and genetic examinations should be performed as early as possible to make a confirmed diagnosis.

摘要

一名2个月大的男孩(孕36周出生)因咳嗽和呼吸困难入院。入院后,发现他患有持续性高血压、蛋白尿和持续性惊厥,影像学检查显示主动脉及主要分支广泛钙化,腹主动脉和右肾动脉局部管腔狭窄,血流速度增加。该男孩在新生儿期因湿肺和肺动脉高压入院,当时就发现有高血压和蛋白尿。进行了高通量全外显子组测序,发现其父母的ENPP1基因存在两个复合杂合突变,即c.130C>T(p.Q44X)和c.1112A>T(p.Y371F)。c.130C>T是一个无义突变,可导致蛋白质从第44个氨基酸处部分缺失,被定义为主要致病突变。c.1112A>T是一个错义突变,已被报道为与特发性婴儿动脉钙化(IIAC)相关的致病突变。因此,他被诊断为IIAC。给予他膦酸盐药物、抗高血压药物、抗惊厥治疗及呼吸支持。血压维持在正常上限。动脉钙化没有恶化。结论是,对于患有持续性高血压和广泛血管钙化的婴儿应考虑IIAC,应尽早进行影像学和基因检查以确诊。