Nearly 50 congenital disorders of glycosylation (CDG) are known, but many patients biochemically diagnosed with CDG do not have mutations in known genes. Here, we describe a 16-year-old male who was born with microcephaly, developed intellectual disability, gastroesophageal reflux and a seizure disorder. We identified a de novo variant in the X-linked SSR4 gene which encodes a protein of the heterotetrameric translocon-associated protein (TRAP) complex. The c.316delT causes a p.F106Sfs*53 in SSR4 and also reduces expression of other TRAP complex proteins. The glycosylation marker Glyc-ER-GFP was used to confirm the underglycosylation in fibroblasts from the patient. Overexpression of the wild-type SSR4 allele partially restores glycosylation of the marker and of the other members of the TRAP complex. This is the first evidence that the TRAP complex, which binds to the oligosaccharyltransferase complex, is directly involved in N-glycosylation.
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http://dx.doi.org/10.1093/hmg/ddt550 | DOI Listing |
PeerJ
January 2025
CIRAD, UMR ASTRE, Montpellier, France.
Bats play key roles in ecosystem functions and provide services to human populations. There is a need to protect bat populations and to mitigate the risks associated with pathogen spillover. Caves are key habitats for many bat species, which use them as roosting and breeding sites.
View Article and Find Full Text PDFChembiochem
January 2025
University of Konstanz, Department of Chemistry, Universitaetsstrasse 10, 78457, Konstanz, GERMANY.
The ubiquitin (Ub) ligase E6AP, which is encoded by the UBE3A gene, has been associated with several human diseases including cervical cancer and Angelman syndrome, a neurodevelopmental disorder. Yet, our knowledge about disease-relevant substrates of E6AP is still limited. The formation of a thioester complex between Ub and the catalytic Cys residue of E6AP represents an essential intermediate step in E6AP-mediated ubiquitination.
View Article and Find Full Text PDFPolymers (Basel)
January 2025
Faculty of Materials Science and Engineering, Kyoto Institute of Technology, Sakyo, Kyoto 606-8585, Japan.
The transient dynamics of photocurrents for poly((4-diphenylamino)benzyl acrylate) (PDAA)-based photorefractive (PR) polymers sensitized with perylene bisimide derivative N,N'-diisopropylphenyl-1,6,7,12-tetrachloroperylene-3,4,9,10-tetracarboxyl bisimide (PBI) at various composition ratios were studied. The PR polymer included (4-(diphenylamino)phenyl)methanol (TPAOH) photoconductive plasticizer and (4-(azepan-1-yl)-benzylidene) malononitrile nonlinear optical dye as well, which are needed for inducing PR effects. All the photocurrents measured at 640 nm were well simulated by a two-trapping site model considering photocarrier generation and recombination processes of the charge transfer (CT) complex between PBI and PDAA.
View Article and Find Full Text PDFJ Cancer Prev
December 2024
Department of Nutrition and Food Science, College of Agriculture and Natural Resources, University of Maryland, College Park, MD, USA.
Identifying the roles of genes in cancer is critical in discovering potential genetic therapies for cancer care. Translocon-associated protein delta (TRAPδ), also known as signal sequence receptor 4 (SSR4), is a constituent unit in the TRAP/SSR complex that resides in the endoplasmic reticulum and plays a key role in transporting newly synthesized proteins into the endoplasmic reticulumn. However, its biological role in disease development remains unknown to date.
View Article and Find Full Text PDFJ Morphol
January 2025
Department of Biology, Gonzaga University, Spokane, Washington, USA.
For over a century researchers have marveled at the square-shaped toe tips of several species of climbing salamanders (genus Aneides), speculating about the function of large blood sinuses therein. Wandering salamanders (Aneides vagrans) have been reported to exhibit exquisite locomotor control while climbing, jumping, and gliding high (88 m) within the redwood canopy; however, a detailed investigation of their digital vascular system has yet to be conducted. Here, we describe the vascular and osteological structure of, and blood circulation through, the distal regions of the toes of A.
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