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钼辅因子缺乏症患儿的围手术期护理

Perioperative Care of a Child With Molybdenum Cofactor Deficiency.

作者信息

Villalobos Edison E, Olakunle Ifeoluwa C, Tobias Joseph D, Smith Ashley

机构信息

Department of Anesthesiology & Pain Medicine, Nationwide Children's Hospital, Columbus, OH, USA.

Department of Anesthesiology & Pain Medicine, The Ohio State University College of Medicine, Columbus, OH, USA.

出版信息

J Med Cases. 2025 Apr;16(4):140-145. doi: 10.14740/jmc5111. Epub 2025 Apr 22.

Abstract

Molybdenum is a trace mineral that is a key component of several enzyme systems. In the human body, molybdenum is complexed with a pterin-based molybdenum cofactor (MOCO), to form the active center of molybdenum-based enzymes. MOCO is synthesized in a four-step process involving six proteins, iron, ATP, and copper. Defects in any of the individual genes involved in this biosynthesis can result in molybdenum cofactor deficiency (MoCD). MoCD is an autosomal recessive disorder with an estimated incidence of 1 in 100,000 - 200,000 live births. Although most patients appear normal at birth, intractable seizures typically develop within hours to days of life, along with feeding difficulties, and subsequent microcephaly, brain atrophy, and severe developmental delay. Mortality is high, with a reported median survival of 2.4 to 3 years. Given the associated end-organ involvement, anesthetic management may be required during radiologic imaging or surgery procedures. We present a 6-year-old child with MoCD type A, who required anesthetic care for a magnetic expansion control (MAGEC) rod insertion with posterior spinal instrumentation under general anesthesia. End-organ involvement of MoCD is presented, previous reports of anesthetic care reviewed, and options for perioperative management discussed.

摘要

钼是一种微量元素,是多种酶系统的关键组成部分。在人体内,钼与一种基于蝶呤的钼辅因子(MOCO)结合,形成钼基酶的活性中心。MOCO通过一个涉及六种蛋白质、铁、三磷酸腺苷(ATP)和铜的四步过程合成。参与这种生物合成的任何单个基因的缺陷都可能导致钼辅因子缺乏症(MoCD)。MoCD是一种常染色体隐性疾病,估计在每10万至20万例活产中发生率为1例。虽然大多数患者出生时看起来正常,但通常在出生后数小时至数天内会出现难治性癫痫发作,同时伴有喂养困难,随后出现小头畸形、脑萎缩和严重发育迟缓。死亡率很高,报告的中位生存期为2.4至3年。鉴于存在相关的终末器官受累情况,在进行放射影像学检查或手术操作期间可能需要进行麻醉管理。我们报告一名6岁的A型MoCD患儿,其在全身麻醉下进行带有后路脊柱内固定的磁性扩张控制(MAGEC)棒插入手术时需要麻醉护理。本文介绍了MoCD的终末器官受累情况,回顾了以往的麻醉护理报告,并讨论了围手术期管理的选择。

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Perioperative Care of a Child With Molybdenum Cofactor Deficiency.钼辅因子缺乏症患儿的围手术期护理
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Perioperative Management of Neurological Conditions.神经疾病的围手术期管理
Health Serv Insights. 2017 Jun 12;10:1178632917711942. doi: 10.1177/1178632917711942. eCollection 2017.
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Antiepileptic drugs and anesthesia.抗癫痫药物与麻醉
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Current evidence for the use of sugammadex in children.舒更葡糖钠用于儿童的现有证据。
Paediatr Anaesth. 2017 Feb;27(2):118-125. doi: 10.1111/pan.13050. Epub 2016 Nov 17.
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Molybdenum cofactor deficiency.钼辅因子缺乏症
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