| Literature DB >> 28777499 |
Therese E Gadomski1,2, Melody Bolton1, Majid Alfadhel3, Chris Dvorak1, Olalekan A Ogunsakin1, Stephen L Nelson2, Eva Morava1,2.
Abstract
ALG13-CDG has been recently discovered as a disorder of severe developmental, intellectual and speech disability, microcephaly, visual abnormalities, seizures, hepatomegaly, coagulation abnormalities, and abnormal serumtransferrin isoelectric focusing in serum. A male with seizures, delayed motor, and speech development, but normal cognition carried a hemizygous, predicted pathogenic ALG13 variant (p.E463G). N-glycosylation studies in plasma were normal. ICAM-1 expression was decreased in patient fibroblasts, supporting the variant's pathogenicity. Adding D-galactose to the patient's fibroblast culture increased ICAM-1 expression in vitro, offering a potential treatment option in ALG13-CDG. The present report is a new example for an N-glycosylation disorder, that may present with normal transferrin isoform analysis, and also demonstrates, that CDG type I patients can have normal cognitive development.Entities:
Keywords: ICAM-1; galactose; glycosylation; normal TIEF; seizures
Mesh:
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Year: 2017 PMID: 28777499 DOI: 10.1002/ajmg.a.38377
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.802