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因母亲3号染色体完全同源二体导致的常染色体隐性遗传性IV型糖原贮积病患儿。

Infant Born With Autosomal Recessive Glycogen Storage Disease Type IV due to Complete Maternal Isodisomy of Chromosome 3.

作者信息

Olsen Sigrid Skovby, Ernst Anja, Christensen Pia Sønderby, Björnsdóttir Ellen Dagmar, Mark Lasse Ringsted, Stefansen Albert Vejlin, Højland Allan Thomas

机构信息

Department of Clinical Genetics, Aalborg University Hospital, Aalborg, Denmark.

Department of Molecular Diagnostics, Aalborg University Hospital, Aalborg, Denmark.

出版信息

Case Rep Genet. 2025 Aug 27;2025:5577571. doi: 10.1155/crig/5577571. eCollection 2025.

Abstract

Uniparental disomy (UPD), the inheritance of two copies of a chromosome from one parent, can lead to recessive genetic disorders or imprinting effects. We report a case of autosomal recessive glycogen storage disease type 4 (GSD IV) due to maternal UPD of chromosome 3, representing the first reported instance of UPD leading to this rare disorder. To avoid an unjustified claim of misattributed paternity, the possibility of UPD should always be kept in mind in cases with the unique finding of the homozygous pathogenic variant only present in one parent. This case highlights the critical role of genetic counseling in uncovering rare genetic conditions and emphasizes the need for continued awareness of UPD in clinical genetics.

摘要

单亲二体性(UPD),即从一个亲本继承一条染色体的两个拷贝,可导致隐性遗传疾病或印记效应。我们报告了一例因3号染色体母源单亲二体性导致的常染色体隐性遗传性糖原贮积病4型(GSD IV),这是首次报道的由UPD导致的这种罕见疾病。为避免不合理的父系错认索赔,在仅在一个亲本中发现纯合致病变异这一独特发现的病例中,应始终牢记UPD的可能性。该病例突出了遗传咨询在发现罕见遗传病方面的关键作用,并强调了临床遗传学中持续关注UPD的必要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4a58/12408131/a7d38bc8115c/CRIG2025-5577571.001.jpg

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