Furin mediates enhanced production of fibrillogenic ABri peptides in familial British dementia
Abstract
The genetic lesion underlying familial British dementia (FBD), an autosomal dominant neurodegenerative disorder, is a T-A transversion at the termination codon of the BRI gene. The mutant gene encodes BRI-L, the precursor of ABri peptides that accumulate in amyloid deposits in FBD brain. We now report that both BRI-L and its wild-type counterpart, BRI, were constitutively processed by the proprotein convertase, furin, resulting in the secretion of carboxyl-terminal peptides that encompass all or part of ABri. Elevated levels of peptides were generated from the mutant BRI precursor. Electron microscopic studies revealed that synthetic ABri peptides assembled into irregular, short fibrils. Collectively, our results support the view that enhanced furin-mediated processing of mutant BRI generates fibrillogenic peptides that initiate the pathogenesis of FBD.
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BRI2 is expressed at high levels in the brain.
“It is expressed at high levels in the brain and cleaved by furin or furin-like proteases at its C terminus to produce a 23 aa peptide (Bri2-23) (Kim et al., 1999;Choi et al., 2004) (Fig. 1 B).”
From BRI2 (ITM2b) Inhibits A Deposition In Vivo (2008) · cites this paper as Kim et al., 1999
The 23 aa peptide Bri23 is released from BRI2 by normal processing.
“It is expressed at high levels in the brain and cleaved by furin or furin-like proteases at its C terminus to produce a 23 aa peptide (Bri2-23) (Kim et al., 1999;Choi et al., 2004) (Fig. 1 B).”
From BRI2 (ITM2b) Inhibits A Deposition In Vivo (2008) · cites this paper as Kim et al., 1999
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References 25 · Cited by 154
- Authors
- Kim, S.-H., Wang, R., Gordon, D. J., Bass, J., Steiner, D. F., Lynn, D. G., Thinakaran, G., Meredith, S. C., & Sisodia, S. S.
- Affiliations
- University of Chicago (US)
- Rockefeller University (US)
- Howard Hughes Medical Institute (US)
- Corresponding author
- Sisodia, Sangram S.
- ORCID
- Venue
- Nature Neuroscience
- Published
- 1999-11
- Volume
- 2
- Issue
- 11
- Pages
- 984-988
- ISSN
- 1097-6256 (ISSN-L)
- 1546-1726
- OpenAlex
- W1536159446
- Cited by
- 154
- References
- 25
- Citations by year
- 2025: 5 · 2024: 5 · 2023: 4 · 2022: 5 · 2021: 3 · 2020: 3 · 2018: 4 · 2017: 2 · 2016: 4 · 2015: 9 · 2014: 5 · 2013: 4 · 2012: 3
- FWCI
- 6.42
- Type
- journal article
- Language
- en
- Open access
- closed
- Licence
- www.springer.com/tdm (tdm)
- Locations
- Nature Neuroscience (publishedVersion)
- PubMed (publishedVersion)
- Funders
- National Institute on Aging (AG 14248, P01AG014248)
- National Institute of General Medical Sciences (5T32 GM-07281, T32GM007281)
- Topics
- Health Sciences › Medicine › Physiology › Alzheimer's disease research and treatments
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Prion Diseases and Protein Misfolding
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Amyloidosis: Diagnosis, Treatment, Outcomes
- Keywords
- furin, familial British dementia, amyloid-beta, proprotein convertases, protein processing, amyloid fibrils, amyloid deposition, neurodegeneration
- MeSH terms
- Amyloid, Animals, Dementia, United Kingdom, Membrane Glycoproteins, Membrane Proteins, Microscopy, Electron, Neurofibrils, Peptide Fragments, Subtilisins, Tumor Cells, Cultured, Heredodegenerative Disorders, Nervous System, Furin, Adaptor Proteins, Signal Transducing, Mice
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