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PMID:19097194

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Citation

Imuta, Y, Nishioka, N, Kiyonari, H and Sasaki, H (2009) Short limbs, cleft palate, and delayed formation of flat proliferative chondrocytes in mice with targeted disruption of a putative protein kinase gene, Pkdcc (AW548124). Dev. Dyn. 238:210-22

Abstract

During long bone development, round proliferative chondrocytes (RPCs) differentiate into flat proliferative chondrocytes (FPCs), and then into hypertrophic chondrocytes (HCs). FPCs and HCs support longitudinal bone growth. Here we show that a putative protein kinase gene, Pkdcc (AW548124), is required for longitudinal bone growth. We originally found Pkdcc expressed in the head organizer, but it is also expressed throughout embryogenesis and in various adult tissues. Pkdcc-/- embryos had no head organizer-related defects, but showed various morphological abnormalities at birth, including short limbs, cleft palate, sternal dysraphia, and shortened intestine. In the long bones of the limbs, only the mineralized regions were shortened, and the cartilage length was normal. In the humerus, Pkdcc was strongly expressed in the early FPCs, and FPC and HC formation was delayed in Pkdcc-/- mutants. Together, these data indicate that Pkdcc encodes a protein kinase that is required for the appropriate timing of FPC differentiation.

Links

PubMed Online version:10.1002/dvdy.21822

Keywords

Amino Acid Sequence; Animals; Base Sequence; Bone and Bones/anatomy & histology; Bone and Bones/physiology; Chondrocytes/cytology; Chondrocytes/pathology; Chondrocytes/physiology; Cleft Palate/genetics; Embryo, Mammalian/anatomy & histology; Embryo, Mammalian/physiology; Gene Expression Regulation, Developmental; In Situ Hybridization; Intestines/abnormalities; Limb Deformities, Congenital/genetics; Mice; Mice, Knockout; Molecular Sequence Data; Protein Kinases/genetics; Protein Kinases/metabolism; Sequence Alignment

Significance

Annotations

Gene product Qualifier GO Term Evidence Code with/from Aspect Extension Notes Status

MOUSE:PKDCC

involved_in

GO:0030501: positive regulation of bone mineralization

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

MOUSE:PKDCC

involved_in

GO:0032332: positive regulation of chondrocyte differentiation

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

MOUSE:PKDCC

involved_in

GO:0048566: embryonic digestive tract development

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

MOUSE:PKDCC

involved_in

GO:0060021: roof of mouth development

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete


See also

References

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