% IMPORTANT: The following is UTF-8 encoded. This means that in the presence
% of non-ASCII characters, it will not work with BibTeX 0.99 or older.
% Instead, you should use an up-to-date BibTeX implementation like “bibtex8” or
% “biber”.
@ARTICLE{Bronckers:136944,
author = {Bronckers, Antonius L J J and Gueneli, Nur and
Lüllmann-Rauch, Renate and Schneppenheim, Janna and Moraru,
Andreea P and Himmerkus, Nina and Bervoets, Theodore J and
Fluhrer, Regina and Everts, Vincent and Saftig, Paul and
Schröder, Bernd},
title = {{T}he intramembrane protease {SPPL}2{A} is critical for
tooth enamel formation.},
journal = {Journal of bone and mineral research},
volume = {28},
number = {7},
issn = {0884-0431},
address = {Hoboken, NJ [u.a.]},
publisher = {Wiley},
reportid = {DZNE-2020-03266},
pages = {1622-1630},
year = {2013},
abstract = {Intramembrane proteases are critically involved in signal
transduction and membrane protein turnover.
Signal-peptide-peptidase-like 2a (SPPL2A), a
presenilin-homologue residing in lysosomes/late endosomes,
cleaves type II-oriented transmembrane proteins. We recently
identified SPPL2A as the enzyme controlling turnover and
functions of the invariant chain (CD74) of the major
histocompatibility complex II (MHCII) and demonstrated
critical importance of this process for B cell development.
Surprisingly, we found that SPPL2A is critical for formation
of dental enamel. In Sppl2a knockout mice, enamel of the
erupted incisors was chalky white and rapidly eroded after
eruption. SPPL2A was found to be expressed in enamel
epithelium during secretory and maturation stage
amelogenesis. Mineral content of enamel in Sppl2a⁻/⁻
incisors was inhomogeneous and reduced by $∼20\%$ compared
to wild-type mice with the most pronounced reduction at the
mesial side. Frequently, disruption of the enamel layer and
localized detachment of the most superficial enamel layer
was observed in the knockout incisors leading to an uneven
enamel surface. In Sppl2a null mice, morphology and function
of secretory stage ameloblasts were not noticeably different
from that of wild-type mice. However, maturation stage
ameloblasts showed reduced height and a characteristic
undulation of the ameloblast layer with localized adherence
of the cells to the outer enamel. This was reflected in a
delayed and incomplete resorption of the proteinaceous
enamel matrix. Thus, we conclude that intramembrane
proteolysis by SPPL2A is essential for maintaining cellular
homeostasis of ameloblasts. Because modulation of SPPL2A
activity appears to be an attractive therapeutic target to
deplete B cells and treat autoimmunity, interference with
tooth enamel formation should be investigated as a possible
adverse effect of pharmacological SPPL2A inhibitors in
humans.},
keywords = {Ameloblasts: enzymology / Animals / Antigens,
Differentiation, B-Lymphocyte: genetics / Antigens,
Differentiation, B-Lymphocyte: metabolism / Aspartic Acid
Endopeptidases: genetics / Aspartic Acid Endopeptidases:
metabolism / Dental Enamel: enzymology / Dental Enamel:
growth $\&$ development / Histocompatibility Antigens Class
II: genetics / Histocompatibility Antigens Class II:
metabolism / Incisor: enzymology / Incisor: growth $\&$
development / Membrane Proteins: genetics / Membrane
Proteins: metabolism / Mice / Mice, Knockout / Proteolysis /
Antigens, Differentiation, B-Lymphocyte (NLM Chemicals) /
Histocompatibility Antigens Class II (NLM Chemicals) /
Membrane Proteins (NLM Chemicals) / invariant chain (NLM
Chemicals) / Aspartic Acid Endopeptidases (NLM Chemicals) /
SPPL2a protein, mouse (NLM Chemicals)},
cin = {AG Fluhrer},
ddc = {610},
cid = {I:(DE-2719)1110000-2},
pnm = {342 - Disease Mechanisms and Model Systems (POF3-342)},
pid = {G:(DE-HGF)POF3-342},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:23426979},
doi = {10.1002/jbmr.1895},
url = {https://pub.dzne.de/record/136944},
}