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J Clin Med. 2018 Nov 8;7(11). pii: E426. doi: 10.3390/jcm7110426.

Maternal RANKL Reduces the Osteopetrotic Phenotype of Null Mutant Mouse Pups.

Author information

1
INSERM, UMR 1238, Faculté de Médecine, Université de Nantes, F-44035 Nantes, France. Benjamin.navet@univ-nantes.fr.
2
INSERM, UMR 1238, Faculté de Médecine, Université de Nantes, F-44035 Nantes, France. jorge.vargas@udea.edu.co.
3
Department of Basic Studies, Faculty of Odontology, University of Antioquia, Medellin AA 1226, Colombia. jorge.vargas@udea.edu.co.
4
INSERM, UMR-1138, Equipe 5, Centre de Recherche des Cordeliers, F-75006 Paris, France. dea.gama10@gmail.com.
5
INSERM, UMR 1238, Faculté de Médecine, Université de Nantes, F-44035 Nantes, France. jerome.amiaud@univ-nantes.fr.
6
Department of Pathology and Laboratory Medicine, School of Medicine, University of Pennsylvania, Philadelphia, PA 19104, USA. ychoi3@pennmedicine.upenn.edu.
7
Department of Immunology, School of Medicine, Juntendo University, Tokyo 113-8421, Japan. hyagita@med.juntendo.ac.jp.
8
CNRS, UPR-9021, Laboratoire Immunologie et Chimie Thérapeutiques, Institut de Biologie Moléculaire et Cellulaire (IBMC), Université de Strasbourg, F-67084 Strasbourg, France. c.mueller@ibmc-cnrs.unistra.fr.
9
INSERM, UMR 1238, Faculté de Médecine, Université de Nantes, F-44035 Nantes, France. Francoise.redini@univ-nantes.fr.
10
INSERM, LEA Sarcoma Research Unit, Department of Oncology and Human Metabolism, Medical School, University of Sheffield, Sheffield S10 2RX, UK. Dominique.Heymann@univ-nantes.fr.
11
INSERM, UMR 1232, LabCT, Université de Nantes, Université d'Angers, Institut de Cancérologie de l'Ouest, site René Gauducheau, F-44805 Saint-Herblain, France. Dominique.Heymann@univ-nantes.fr.
12
INSERM, UMR-1138, Equipe 5, Centre de Recherche des Cordeliers, F-75006 Paris, France. Bea.castaneda.1@gmail.com.
13
INSERM, UMR 1238, Faculté de Médecine, Université de Nantes, F-44035 Nantes, France. frederic.lezot@univ-nantes.fr.

Abstract

RANKL signalization is implicated in the morphogenesis of various organs, including the skeleton. Mice invalidated for Rankl present an osteopetrotic phenotype that was less severe than anticipated, depending on RANKL's implication in morphogenesis. The hypothesis of an attenuated phenotype, as a result of compensation during gestation by RANKL of maternal origin, was thus brought into question. In order to answer this question, Rankl null mutant pups from null mutant parents were generated, and the phenotype analyzed. The results validated the presence of a more severe osteopetrotic phenotype in the second-generation null mutant with perinatal lethality. The experiments also confirmed that RANKL signalization plays a part in the morphogenesis of skeletal elements through its involvement in cell-to-cell communication, such as in control of osteoclast differentiation. To conclude, we have demonstrated that the phenotype associated with Rankl invalidation is attenuated through compensation by RANKL of maternal origin.

KEYWORDS:

RANKL; bone; mandible; morphogenesis; osteoclast; skeletal growth; tooth

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