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

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.

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Publication Details

Journal
Case Reports in Genetics
Published
2025-01-01
DOI
https://doi.org/10.1155/crig/5577571
Primary Topic
Genetic Syndromes and Imprinting
Type
article
Field-Weighted Citation Impact
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article

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

Anja Ernst, Lasse Ringsted Mark, Allan Thomas Højland, S Olsen et al.
Case Reports in Genetics
Genetic Syndromes and Imprinting
article

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

Anja Ernst, Lasse Ringsted Mark, Allan Thomas Højland, S Olsen, Poul Christensen, Ellen Dagmar Björnsdóttir, Albert Vejlin Stefansen
article en

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.

Case Reports in GeneticsVol. 2025(1)
Aalborg University Hospital (DK), Aalborg University (DK)
Openalex Percentile: Top 75%
Genetic Syndromes and Imprinting
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