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一名因罕见变异导致的ⅢA型糖原贮积病女性患者的妊娠管理与临床结局

The Management and Clinical Outcomes of Pregnancy in a Female With Glycogen Storage Disease Type IIIA Caused by Rare Variant.

作者信息

Puente-Ruiz Nuria, Palaniappan Saru, Woodall Alison, Cooper Robert, Terry Allyson, Oldham Andrew, Rousseau Abigail, Campbell Christopher, Vasudevan Pradeep, Stepien Karolina M

机构信息

Adult Inherited Metabolic Diseases, Salford Royal NHS Foundation Trust Salford UK.

Department of Internal Medicine Marqués de Valdecilla University Hospital. Instituto de Investigación Valdecilla (IDIVAL). Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER) Santander Spain.

出版信息

JIMD Rep. 2025 Jul 2;66(4):e70030. doi: 10.1002/jmd2.70030. eCollection 2025 Jul.

Abstract

Glycogen storage disease type III (GSD III) is an inborn error of carbohydrate metabolism with an autosomal recessive inheritance pattern. Phenotypically, patients can manifest a broad clinical spectrum. Most patients affected with GSD IIIA (85%) have a non-functional GDE enzyme primarily affecting the liver and cardiac/skeletal muscle (Type IIIA). Initial clinical manifestations of GSD IIIA present in the first year of life. Presentation is very similar to GSD type I. Up to 98% of children affected have hepatomegaly, hypoglycaemia (53%) with marked ketosis (34%), short stature (49%), delayed puberty, and frequent infections (17%). In adulthood, they may have cirrhosis, adenomas, or hepatocarcinomas (11%), cardiomyopathy (58%) and myopathy (34%). Pregnancy has been documented in women with GSD III. Nutritional requirements are increased during pregnancy, especially in the third trimester. We report the management of a woman with GSD IIIA found to be compound heterozygous for two pathogenic AGL variants, c.798C>G p.(Tyr266Ter) and c.4258_4259ins? p.(Asp1420fs), who had a planned pregnancy. Cardiac outcomes are also described/discussed.

摘要

Ⅲ型糖原贮积病(GSD Ⅲ)是一种碳水化合物代谢的先天性缺陷,呈常染色体隐性遗传模式。从表型上看,患者可表现出广泛的临床症状。大多数患有ⅢA型GSD Ⅲ的患者(85%)有一种无功能的糖原脱支酶,主要影响肝脏和心脏/骨骼肌(ⅢA型)。ⅢA型GSD Ⅲ的初始临床表现出现在生命的第一年。表现与Ⅰ型糖原贮积病非常相似。高达98%的患病儿童有肝肿大、低血糖(53%)并伴有明显酮症(34%)、身材矮小(49%)、青春期延迟和频繁感染(17%)。在成年期,他们可能患有肝硬化、腺瘤或肝癌(11%)、心肌病(58%)和肌病(34%)。已有Ⅲ型糖原贮积病女性怀孕的记录。孕期营养需求增加,尤其是在孕晚期。我们报告了一名患有ⅢA型GSD Ⅲ的女性的治疗情况,该女性被发现为两种致病性AGL变异的复合杂合子,即c.798C>G p.(Tyr266Ter)和c.4258_4259ins? p.(Asp1420fs),她计划怀孕。还描述/讨论了心脏方面的结果。

本文引用的文献

2
Glycogen storage disease type IIIa in pregnant women: A guide to management.孕妇IIIa型糖原贮积病:管理指南
JIMD Rep. 2022 Mar 22;63(3):216-220. doi: 10.1002/jmd2.12282. eCollection 2022 May.
5
Cardiac Pathology in Glycogen Storage Disease Type III.Ⅲ型糖原贮积病的心脏病理学
JIMD Rep. 2012;6:65-72. doi: 10.1007/8904_2011_118. Epub 2012 Jan 31.
10

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