Title | DPAGT1 Deficiency with Encephalopathy (DPAGT1-CDG): Clinical and Genetic Description of 11 New Patients. |
Publication Type | Journal Article |
Year of Publication | 2019 |
Authors | Ng, BG, Underhill, HR, Palm, L, Bengtson, P, Rozet, J-M, Gerber, S, Munnich, A, Zanlonghi, X, Stevens, CA, Kircher, M, Nickerson, DA, Buckingham, KJ, Josephson, KD, Shendure, J, Bamshad, MJ, Freeze, HH, Eklund, EA |
Corporate Authors | University of Washington Center for Mendelian Genomics |
Journal | JIMD Rep |
Volume | 44 |
Pagination | 85-92 |
Date Published | 2019 |
ISSN | 2192-8304 |
Abstract | Pathogenic mutations in DPAGT1 cause a rare type of a congenital disorder of glycosylation termed DPAGT1-CDG or, alternatively, a milder version with only myasthenia known as DPAGT1-CMS. Fourteen disease-causing mutations in 28 patients from 10 families have previously been reported to cause the systemic form, DPAGT1-CDG. We here report on another 11 patients from 8 families and add 10 new mutations. Most patients have a very severe disease course, where common findings are pronounced muscular hypotonia, intractable epilepsy, global developmental delay/intellectual disability, and early death. We also present data on three affected females that are young adults and have a somewhat milder, stable disease. Our findings expand both the molecular and clinical knowledge of previously published data but also widen the phenotypic spectrum of DPAGT1-CDG. |
DOI | 10.1007/8904_2018_128 |
Alternate Journal | JIMD Rep |
PubMed ID | 30117111 |
PubMed Central ID | PMC6323016 |
Grant List | R01 DK099551 / DK / NIDDK NIH HHS / United States U54 HG006493 / HG / NHGRI NIH HHS / United States UM1 HG006493 / HG / NHGRI NIH HHS / United States |