缺钼辅助因子患儿围手术期护理。

IF 0.9
Journal of medical cases Pub Date : 2025-04-01 Epub Date: 2025-04-22 DOI:10.14740/jmc5111
Edison E Villalobos, Ifeoluwa C Olakunle, Joseph D Tobias, Ashley Smith
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引用次数: 0

摘要

钼是一种微量矿物质,是几种酶系统的关键成分。在人体内,钼与基于蝶呤的钼辅因子(MOCO)络合,形成钼基酶的活性中心。MOCO的合成需要四个步骤,包括六种蛋白质、铁、ATP和铜。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助因子缺乏症(MoCD)。MoCD是一种常染色体隐性遗传病,估计发病率为10万至20万活产婴儿中有1例。虽然大多数患者在出生时表现正常,但顽固性癫痫通常在出生后数小时至数天内发生,并伴有喂养困难,随后出现小头畸形、脑萎缩和严重的发育迟缓。死亡率很高,据报道中位生存期为2.4至3年。考虑到相关的终末器官受累,在放射成像或手术过程中可能需要麻醉管理。我们报告了一名6岁的a型MoCD患儿,他在全身麻醉下需要麻醉治疗磁扩张控制(MAGEC)棒插入并后路脊柱内固定。本文介绍了MoCD的终末器官受累情况,回顾了以前的麻醉护理报告,并讨论了围手术期管理的选择。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Perioperative Care of a Child With Molybdenum Cofactor Deficiency.

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.

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CiteScore
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