Coordinated multiple cellular processes in tongue development Article Swipe
Maiko Kawasaki
,
Katsushige Kawasaki
,
Finsa Tisna Sari
,
Takehisa Kudo
,
Jun Nihara
,
Madoka Kitamura
,
Takahiro Nagai
,
Vanessa Utama
,
Yoko Ishida
,
Fumiya Meguro
,
Takayuki Nishimura
,
Yuan Kogure
,
Satoshi Maruyama
,
Jun‐ichi Tanuma
,
Yoshito Kakihara
,
Takeyasu Maeda
,
Sarah Ghafoor
,
Roman Hossein Khonsari
,
Pierre Corre
,
Paul T. Sharpe
,
Martyn T. Cobourne
,
Brunella Franco
,
Atsushi Ohazama
·
YOU?
·
· 2023
· Open Access
·
· DOI: https://doi.org/10.1101/2023.01.16.524234
YOU?
·
· 2023
· Open Access
·
· DOI: https://doi.org/10.1101/2023.01.16.524234
Dysfunction of primary cilia leads to genetic disorder, ciliopathies, which shows various malformations in many vital organs such as brain. Multiple tongue deformities including cleft, hamartoma and ankyloglossia are also seen in ciliopathies, which yield difficulties in fundamental functions such as mastication and vocalization. Here, we found these tongue anomalies in mice with mutation of ciliary protein. Abnormal cranial neural crest-derived cells (CNCC) failed to evoke Hh signal for differentiation of mesoderm-derived cells into myoblasts, which resulted in abnormal differentiation of mesoderm-derived cells into adipocytes. The ectopic adipose subsequently arrested migration of other mesoderm-derived cells and CNCC. Some aberrant CNCC abnormally differentiated into osteoblasts due to the lack of Hh signal, which migrated into tongue to form ectopic bone. Ankyloglossia was caused by aberrant cell migration due to lack of non-canonical Wnt signaling. In addition to ciliopathies, these tongue anomalies are often observed as non-familial condition in human. We found that these tongue deformities could be reproduced in wild-type mice by simple mechanical manipulations in CNCC to disturb cellular processes which were disrupted in mutant mice. Thus, tongue development requires coordinated multiple cellular processes (cell-cell contact, migration and differentiation). Our results provide hints for possible future treatment in ciliopathies.
Related Topics
Concepts
Ciliopathies
Cilium
Biology
Cell biology
Mesoderm
Cranial neural crest
Ectoderm
Ciliopathy
Anatomy
Neuroscience
Genetics
Neural crest
Phenotype
Embryonic stem cell
Embryogenesis
Gene
Embryo
Metadata
- Type
- preprint
- Language
- en
- Landing Page
- https://doi.org/10.1101/2023.01.16.524234
- https://www.biorxiv.org/content/biorxiv/early/2023/01/18/2023.01.16.524234.full.pdf
- OA Status
- green
- References
- 66
- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W4317209881
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
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https://openalex.org/W4317209881Canonical identifier for this work in OpenAlex
- DOI
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https://doi.org/10.1101/2023.01.16.524234Digital Object Identifier
- Title
-
Coordinated multiple cellular processes in tongue developmentWork title
- Type
-
preprintOpenAlex work type
- Language
-
enPrimary language
- Publication year
-
2023Year of publication
- Publication date
-
2023-01-18Full publication date if available
- Authors
-
Maiko Kawasaki, Katsushige Kawasaki, Finsa Tisna Sari, Takehisa Kudo, Jun Nihara, Madoka Kitamura, Takahiro Nagai, Vanessa Utama, Yoko Ishida, Fumiya Meguro, Takayuki Nishimura, Yuan Kogure, Satoshi Maruyama, Jun‐ichi Tanuma, Yoshito Kakihara, Takeyasu Maeda, Sarah Ghafoor, Roman Hossein Khonsari, Pierre Corre, Paul T. Sharpe, Martyn T. Cobourne, Brunella Franco, Atsushi OhazamaList of authors in order
- Landing page
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https://doi.org/10.1101/2023.01.16.524234Publisher landing page
- PDF URL
-
https://www.biorxiv.org/content/biorxiv/early/2023/01/18/2023.01.16.524234.full.pdfDirect link to full text PDF
- Open access
-
YesWhether a free full text is available
- OA status
-
greenOpen access status per OpenAlex
- OA URL
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https://www.biorxiv.org/content/biorxiv/early/2023/01/18/2023.01.16.524234.full.pdfDirect OA link when available
- Concepts
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Ciliopathies, Cilium, Biology, Cell biology, Mesoderm, Cranial neural crest, Ectoderm, Ciliopathy, Anatomy, Neuroscience, Genetics, Neural crest, Phenotype, Embryonic stem cell, Embryogenesis, Gene, EmbryoTop concepts (fields/topics) attached by OpenAlex
- Cited by
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0Total citation count in OpenAlex
- References (count)
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66Number of works referenced by this work
- Related works (count)
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10Other works algorithmically related by OpenAlex
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