PittsburghV1, Main, Exploration, bibRecord, 003053

Enamelin Is Critical for Ameloblast Integrity and Enamel Ultrastructure Formation

Identifieur interne : 003053 ( Main/Exploration ); précédent : 003052; suivant : 003054

Enamelin Is Critical for Ameloblast Integrity and Enamel Ultrastructure Formation

Auteurs : Jan C.-C. Hu [États-Unis] ; Yuanyuan Hu [États-Unis] ; Yuhe Lu [États-Unis] ; Charles E. Smith [États-Unis, Canada] ; Rangsiyakorn Lertlam [États-Unis] ; John Timothy Wright [États-Unis] ; Cynthia Suggs [États-Unis] ; Marc D. Mckee [Canada] ; Elia Beniash [États-Unis] ; M. Enamul Kabir [États-Unis] ; James P. Simmer [États-Unis]

Source :

PLoS ONE [ 1932-6203 ] ; 2014.

RBID : PMC:3945975

Descripteurs français

English descriptors

KwdEn :
- Ameloblasts (metabolism), Amelogenin (metabolism), Amelogenin (ultrastructure), Animals, Animals, Newborn, Dental Enamel (embryology), Dental Enamel (metabolism), Dental Enamel (ultrastructure), Dental Enamel Proteins (genetics), Dental Enamel Proteins (metabolism), Dentin (embryology), Dentin (metabolism), Dentin (ultrastructure), Immunohistochemistry, Mice, Knockout, Mice, Transgenic, Microscopy, Electron, Scanning, Microscopy, Electron, Transmission, Molar (embryology), Molar (metabolism), Staining and Labeling (methods), beta-Galactosidase (genetics), beta-Galactosidase (metabolism).
MESH :
- chemical , genetics : Dental Enamel Proteins, beta-Galactosidase.
- chemical , metabolism : Amelogenin, Dental Enamel Proteins, beta-Galactosidase.
- embryology : Dental Enamel, Dentin, Molar.
- metabolism : Ameloblasts, Dental Enamel, Dentin, Molar.
- methods : Staining and Labeling.
- chemical , ultrastructure : Amelogenin, Dental Enamel, Dentin.
- Animals, Animals, Newborn, Immunohistochemistry, Mice, Knockout, Mice, Transgenic, Microscopy, Electron, Scanning, Microscopy, Electron, Transmission.

Abstract

Mutations in the human enamelin gene cause autosomal dominant hypoplastic amelogenesis imperfecta in which the affected enamel is thin or absent. Study of enamelin knockout NLS-lacZ knockin mice revealed that mineralization along the distal membrane of ameloblast is deficient, resulting in no true enamel formation. To determine the function of enamelin during enamel formation, we characterized the developing teeth of the Enam^−/− mice, generated amelogenin-driven enamelin transgenic mouse models, and then introduced enamelin transgenes into the Enam^−/− mice to rescue enamel defects. Mice at specific stages of development were subjected to morphologic and structural analysis using β-galactosidase staining, immunohistochemistry, and transmission and scanning electron microscopy. Enamelin expression was ameloblast-specific. In the absence of enamelin, ameloblasts pathology became evident at the onset of the secretory stage. Although the aggregated ameloblasts generated matrix-containing amelogenin, they were not able to create a well-defined enamel space or produce normal enamel crystals. When enamelin is present at half of the normal quantity, enamel was thinner with enamel rods not as tightly arranged as in wild type suggesting that a specific quantity of enamelin is critical for normal enamel formation. Enamelin dosage effect was further demonstrated in transgenic mouse lines over expressing enamelin. Introducing enamelin transgene at various expression levels into the Enam^−/− background did not fully recover enamel formation while a medium expresser in the Enam^+/− background did. Too much or too little enamelin abolishes the production of enamel crystals and prism structure. Enamelin is essential for ameloblast integrity and enamel formation.

Url:

http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3945975

DOI: 10.1371/journal.pone.0089303
PubMed: 24603688
PubMed Central: 3945975

Affiliations:

Le document en format XML

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<author><name sortKey="Suggs, Cynthia" sort="Suggs, Cynthia" uniqKey="Suggs C" first="Cynthia" last="Suggs">Cynthia Suggs</name>
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</nlm:aff>
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<author><name sortKey="Simmer, James P" sort="Simmer, James P" uniqKey="Simmer J" first="James P." last="Simmer">James P. Simmer</name>
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<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Ameloblasts (metabolism)</term>
<term>Amelogenin (metabolism)</term>
<term>Amelogenin (ultrastructure)</term>
<term>Animals</term>
<term>Animals, Newborn</term>
<term>Dental Enamel (embryology)</term>
<term>Dental Enamel (metabolism)</term>
<term>Dental Enamel (ultrastructure)</term>
<term>Dental Enamel Proteins (genetics)</term>
<term>Dental Enamel Proteins (metabolism)</term>
<term>Dentin (embryology)</term>
<term>Dentin (metabolism)</term>
<term>Dentin (ultrastructure)</term>
<term>Immunohistochemistry</term>
<term>Mice, Knockout</term>
<term>Mice, Transgenic</term>
<term>Microscopy, Electron, Scanning</term>
<term>Microscopy, Electron, Transmission</term>
<term>Molar (embryology)</term>
<term>Molar (metabolism)</term>
<term>Staining and Labeling (methods)</term>
<term>beta-Galactosidase (genetics)</term>
<term>beta-Galactosidase (metabolism)</term>
</keywords>
<keywords scheme="KwdFr" xml:lang="fr"><term>Améloblastes (métabolisme)</term>
<term>Amélogénine (métabolisme)</term>
<term>Amélogénine (ultrastructure)</term>
<term>Animaux</term>
<term>Animaux nouveau-nés</term>
<term>Coloration et marquage ()</term>
<term>Dentine (embryologie)</term>
<term>Dentine (métabolisme)</term>
<term>Dentine (ultrastructure)</term>
<term>Immunohistochimie</term>
<term>Microscopie électronique à balayage</term>
<term>Microscopie électronique à transmission</term>
<term>Molaire (embryologie)</term>
<term>Molaire (métabolisme)</term>
<term>Protéines de l'émail dentaire (génétique)</term>
<term>Protéines de l'émail dentaire (métabolisme)</term>
<term>Souris knockout</term>
<term>Souris transgéniques</term>
<term>beta-Galactosidase (génétique)</term>
<term>beta-Galactosidase (métabolisme)</term>
<term>Émail dentaire (embryologie)</term>
<term>Émail dentaire (métabolisme)</term>
<term>Émail dentaire (ultrastructure)</term>
</keywords>
<keywords scheme="MESH" type="chemical" qualifier="genetics" xml:lang="en"><term>Dental Enamel Proteins</term>
<term>beta-Galactosidase</term>
</keywords>
<keywords scheme="MESH" type="chemical" qualifier="metabolism" xml:lang="en"><term>Amelogenin</term>
<term>Dental Enamel Proteins</term>
<term>beta-Galactosidase</term>
</keywords>
<keywords scheme="MESH" qualifier="embryologie" xml:lang="fr"><term>Dentine</term>
<term>Molaire</term>
<term>Émail dentaire</term>
</keywords>
<keywords scheme="MESH" qualifier="embryology" xml:lang="en"><term>Dental Enamel</term>
<term>Dentin</term>
<term>Molar</term>
</keywords>
<keywords scheme="MESH" qualifier="génétique" xml:lang="fr"><term>Protéines de l'émail dentaire</term>
<term>beta-Galactosidase</term>
</keywords>
<keywords scheme="MESH" qualifier="metabolism" xml:lang="en"><term>Ameloblasts</term>
<term>Dental Enamel</term>
<term>Dentin</term>
<term>Molar</term>
</keywords>
<keywords scheme="MESH" qualifier="methods" xml:lang="en"><term>Staining and Labeling</term>
</keywords>
<keywords scheme="MESH" qualifier="métabolisme" xml:lang="fr"><term>Améloblastes</term>
<term>Amélogénine</term>
<term>Dentine</term>
<term>Molaire</term>
<term>Protéines de l'émail dentaire</term>
<term>beta-Galactosidase</term>
<term>Émail dentaire</term>
</keywords>
<keywords scheme="MESH" type="chemical" qualifier="ultrastructure" xml:lang="en"><term>Amelogenin</term>
<term>Dental Enamel</term>
<term>Dentin</term>
</keywords>
<keywords scheme="MESH" xml:lang="en"><term>Animals</term>
<term>Animals, Newborn</term>
<term>Immunohistochemistry</term>
<term>Mice, Knockout</term>
<term>Mice, Transgenic</term>
<term>Microscopy, Electron, Scanning</term>
<term>Microscopy, Electron, Transmission</term>
</keywords>
<keywords scheme="MESH" xml:lang="fr"><term>Amélogénine</term>
<term>Animaux</term>
<term>Animaux nouveau-nés</term>
<term>Coloration et marquage</term>
<term>Dentine</term>
<term>Immunohistochimie</term>
<term>Microscopie électronique à balayage</term>
<term>Microscopie électronique à transmission</term>
<term>Souris knockout</term>
<term>Souris transgéniques</term>
<term>Émail dentaire</term>
</keywords>
</textClass>
</profileDesc>
</teiHeader>
<front><div type="abstract" xml:lang="en"><p>Mutations in the human enamelin gene cause autosomal dominant hypoplastic <italic>amelogenesis imperfecta</italic>
 in which the affected enamel is thin or absent. Study of enamelin knockout NLS-<italic>lacZ</italic>
 knockin mice revealed that mineralization along the distal membrane of ameloblast is deficient, resulting in no true enamel formation. To determine the function of enamelin during enamel formation, we characterized the developing teeth of the <italic>Enam<sup>−/−</sup>
</italic>
 mice, generated amelogenin-driven enamelin transgenic mouse models, and then introduced enamelin transgenes into the <italic>Enam<sup>−/−</sup>
</italic>
 mice to rescue enamel defects. Mice at specific stages of development were subjected to morphologic and structural analysis using β-galactosidase staining, immunohistochemistry, and transmission and scanning electron microscopy. Enamelin expression was ameloblast-specific. In the absence of enamelin, ameloblasts pathology became evident at the onset of the secretory stage. Although the aggregated ameloblasts generated matrix-containing amelogenin, they were not able to create a well-defined enamel space or produce normal enamel crystals. When enamelin is present at half of the normal quantity, enamel was thinner with enamel rods not as tightly arranged as in wild type suggesting that a specific quantity of enamelin is critical for normal enamel formation. Enamelin dosage effect was further demonstrated in transgenic mouse lines over expressing enamelin. Introducing enamelin transgene at various expression levels into the <italic>Enam<sup>−/−</sup>
</italic>
 background did not fully recover enamel formation while a medium expresser in the <italic>Enam<sup>+/−</sup>
</italic>
 background did. Too much or too little enamelin abolishes the production of enamel crystals and prism structure. Enamelin is essential for ameloblast integrity and enamel formation.</p>
</div>
</front>
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Enamelin Is Critical for Ameloblast Integrity and Enamel Ultrastructure Formation

Enamelin Is Critical for Ameloblast Integrity and Enamel Ultrastructure Formation

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