| Literature DB >> 33583022 |
Hind Alsharhan1,2,3, Bobby G Ng4, Earnest James Paul Daniel2, Jennifer Friedman5, Eniko K Pivnick6, Amal Al-Hashem7,8, Eissa Ali Faqeih9, Pengfei Liu10,11, Nicole M Engelhardt1, Kierstin N Keller1, Jie Chen2, Pamela A Mazzeo12, Jill A Rosenfeld10,11, Michael J Bamshad13,14,15, Deborah A Nickerson14,15, Kimiyo M Raymond16, Hudson H Freeze4, Miao He2, Andrew C Edmondson1, Christina Lam13,17.
Abstract
Congenital disorders of glycosylation (CDGs) are a continuously expanding group of monogenic disorders of glycoprotein and glycolipid biosynthesis that cause multisystem diseases. Individuals with ALG3-CDG frequently exhibit severe neurological involvement (epilepsy, microcephaly, and hypotonia), ocular anomalies, dysmorphic features, skeletal anomalies, and feeding difficulties. We present 10 unreported individuals diagnosed with ALG3-CDG based on molecular and biochemical testing with 11 novel variants in ALG3, bringing the total to 40 reported individuals. In addition to the typical multisystem disease seen in ALG3-CDG, we expand the symptomatology of ALG3-CDG to now include endocrine abnormalities, neural tube defects, mild aortic root dilatation, immunodeficiency, and renal anomalies. N-glycan analyses of these individuals showed combined deficiencies of hybrid glycans and glycan extension beyond Man5 GlcNAc2 consistent with their truncated lipid-linked precursor oligosaccharides. This spectrum of N-glycan changes is unique to ALG3-CDG. These expanded features of ALG3-CDG facilitate diagnosis and suggest that optimal management should include baseline endocrine, renal, cardiac, and immunological evaluation at the time of diagnosis and with ongoing monitoring.Entities:
Keywords: N-glycans; congenital disorders of glycosylation; endocrine; immunodeficiency; neural tube defect
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Year: 2021 PMID: 33583022 PMCID: PMC8282734 DOI: 10.1002/jimd.12367
Source DB: PubMed Journal: J Inherit Metab Dis ISSN: 0141-8955 Impact factor: 4.750