Lineage-specific mutation of Lmx1b provides new insights into distinct regulation of suture development in different areas of the calvaria
The calvaria (top part of the skull) is made of pieces of bone as well as multiple soft tissue joints called sutures. The latter is crucial to the growth and morphogenesis of the skull, and thus a loss of calvarial sutures can lead to severe congenital defects in humans. During embryogenesis, the ca...
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Published in | Frontiers in physiology Vol. 14; p. 1225118 |
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Main Authors | , , , , , , , , , , , |
Format | Journal Article |
Language | English |
Published |
Frontiers Media S.A
01.08.2023
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Subjects | |
Online Access | Get full text |
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Summary: | The calvaria (top part of the skull) is made of pieces of bone as well as multiple soft tissue joints called sutures. The latter is crucial to the growth and morphogenesis of the skull, and thus a loss of calvarial sutures can lead to severe congenital defects in humans. During embryogenesis, the calvaria develops from the cranial mesenchyme covering the brain, which contains cells originating from the neural crest and the mesoderm. While the mechanism that patterns the cranial mesenchyme into bone and sutures is not well understood, function of
Lmx1b
, a gene encoding a LIM-domain homeodomain transcription factor, plays a key role in this process. In the current study, we investigated a difference in the function of
Lmx1b
in different parts of the calvaria using neural crest-specific and mesoderm-specific
Lmx1b
mutants. We found that
Lmx1b
was obligatory for development of the interfrontal suture and the anterior fontanel along the dorsal midline of the skull, but not for the posterior fontanel over the midbrain. Also,
Lmx1b
mutation in the neural crest-derived mesenchyme, but not the mesoderm-derived mesenchyme, had a non-cell autonomous effect on coronal suture development. Furthermore, overexpression of
Lmx1b
in the neural crest lineage had different effects on the position of the coronal suture on the apical part and the basal part. Other unexpected phenotypes of
Lmx1b
mutants led to an additional finding that the coronal suture and the sagittal suture are of dual embryonic origin. Together, our data reveal a remarkable level of regional specificity in regulation of calvarial development. |
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Bibliography: | ObjectType-Article-1 SourceType-Scholarly Journals-1 ObjectType-Feature-2 content type line 23 Reviewed by: Nan E. Hatch, University of Michigan, United States D’Juan Farmer, University of California, Los Angeles, United States These authors have contributed equally to this work Emily L. Durham, Children’s Hospital of Philadelphia, United States Edited by: Greg Holmes, Icahn School of Medicine at Mount Sinai, United States |
ISSN: | 1664-042X 1664-042X |
DOI: | 10.3389/fphys.2023.1225118 |