| Literature DB >> 35716054 |
Daniah Albokhari1,2, Bobby G Ng3, Alis Guberinic4, Earnest James Paul Daniel5, Nicole M Engelhardt1, Rita Barone6, Agata Fiumara7, Livia Garavelli8, Gabriele Trimarchi8, Lynne Wolfe9, Kimiyo M Raymond10, Eva Morava4, Miao He5, Hudson H Freeze3, Christina Lam11,12, Andrew C Edmondson1.
Abstract
Congenital disorders of glycosylation are a continuously expanding group of monogenic disorders of glycoprotein and glycolipid glycan biosynthesis. These disorders mostly manifest with multisystem involvement. Individuals with ALG8-CDG commonly present with hypotonia, protein-losing enteropathy, and hepatic involvement. Here, we describe seven unreported individuals diagnosed with ALG8-CDG based on biochemical and molecular testing and we identify nine novel variants in ALG8, bringing the total to 26 individuals with ALG8-CDG in the medical literature. In addition to the typical multisystem involvement documented in ALG8-CDG, our cohort includes the two oldest patients reported and further expands the phenotype of ALG8-CDG to include stable intellectual disability, autism spectrum disorder and other neuropsychiatric symptoms. We further expand the clinical features in a variety of organ systems including ocular, musculoskeletal, dermatologic, endocrine, and cardiac abnormalities and suggest a comprehensive evaluation and monitoring strategy to improve clinical management.Entities:
Keywords: N-glycans; congenital disorders of glycosylation; lipid-linked oligosaccharides
Mesh:
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Year: 2022 PMID: 35716054 PMCID: PMC9474684 DOI: 10.1002/jimd.12527
Source DB: PubMed Journal: J Inherit Metab Dis ISSN: 0141-8955 Impact factor: 4.750