DOI: 10.1242/dev.124.1.113. Specific and redundant functions of <i>Gli2</i> and <i>Gli3</i> zinc finger genes in skeletal patterning and development

Specific and redundant functions of Gli2 and Gli3 zinc finger genes in skeletal patterning and development

10.1242/dev.124.1.113

Crossref journal-article

The Company of Biologists

Development (237)

Abstract

ABSTRACT The correct patterning of vertebrate skeletal elements is controlled by inductive interactions. Two vertebrate hedgehog proteins, Sonic hedgehog and Indian hedgehog, have been implicated in skeletal development. During somite differentiation and limb development, Sonic hedgehog functions as an inductive signal from the notochord, floor plate and zone of polarizing activity. Later in skeletogenesis, Indian hedgehog functions as a regulator of chondrogenesis during endochondral ossification. The vertebrate Gli zinc finger proteins are putative transcription factors that respond to Hedgehog signaling. In Drosophila, the Gli homolog cubitus interruptus is required for the activation of hedgehog targets and also functions as a repressor of hedgehog expression. We show here that Gli2 mutant mice exhibit severe skeletal abnormalities including cleft palate, tooth defects, absence of vertebral body and intervertebral discs, and shortened limbs and sternum. Interestingly, Gli2 and Gli3 (C.-c. Hui and A. L. Joyner (1993). Nature Genet. 3, 241-246) mutant mice exhibit different subsets of skeletal defects indicating that they implement specific functions in the development of the neural crest, somite and lateral plate mesoderm derivatives. Although Gli2 and Gli3 are not functionally equivalent, double mutant analysis indicates that, in addition to their specific roles, they also serve redundant functions during skeletal development. The role of Gli2 and Gli3 in Hedgehog signaling during skeletal development is discussed.

Bibliography

Mo, R., Freer, A. M., Zinyk, D. L., Crackower, M. A., Michaud, J., Heng, H. H.-Q., Chik, K. W., Shi, X.-M., Tsui, L.-C., Cheng, S. H., Joyner, A. L., & Hui, C. (1997). Specific and redundant functions of Gli2 and Gli3 zinc finger genes in skeletal patterning and development. Development, 124(1), 113â123.

Authors 12

Rong Mo (first)
Anne Marie Freer (additional)
Dawn L. Zinyk (additional)
Michael A. Crackower (additional)
Jacques Michaud (additional)
Henry H.-Q. Heng (additional)
Ki Wai Chik (additional)
Xiao-Mei Shi (additional)
Lap-Chee Tsui (additional)
Shuk Han Cheng (additional)
Alexandra L. Joyner (additional)
Chi-chung Hui (additional)

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Dates

Type	When
Created	4 years, 4 months ago (April 25, 2021, 10:43 p.m.)
Deposited	6 months, 2 weeks ago (Feb. 13, 2025, 7:09 p.m.)
Indexed	16 minutes ago (Aug. 29, 2025, 7:42 p.m.)
Issued	28 years, 7 months ago (Jan. 1, 1997)
Published	28 years, 7 months ago (Jan. 1, 1997)
Published Online	28 years, 7 months ago (Jan. 1, 1997)
Published Print	28 years, 7 months ago (Jan. 1, 1997)

Funders 0

None

BibTeX

@article{Mo_1997, title={Specific and redundant functions of Gli2 and Gli3 zinc finger genes in skeletal patterning and development}, volume={124}, ISSN={1477-9129}, url={http://dx.doi.org/10.1242/dev.124.1.113}, DOI={10.1242/dev.124.1.113}, number={1}, journal={Development}, publisher={The Company of Biologists}, author={Mo, Rong and Freer, Anne Marie and Zinyk, Dawn L. and Crackower, Michael A. and Michaud, Jacques and Heng, Henry H.-Q. and Chik, Ki Wai and Shi, Xiao-Mei and Tsui, Lap-Chee and Cheng, Shuk Han and Joyner, Alexandra L. and Hui, Chi-chung}, year={1997}, month=jan, pages={113–123} }

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  "abstract": "<jats:title>ABSTRACT</jats:title>\n               <jats:p>The correct patterning of vertebrate skeletal elements is controlled by inductive interactions. Two vertebrate hedgehog proteins, Sonic hedgehog and Indian hedgehog, have been implicated in skeletal development. During somite differentiation and limb development, Sonic hedgehog functions as an inductive signal from the notochord, floor plate and zone of polarizing activity. Later in skeletogenesis, Indian hedgehog functions as a regulator of chondrogenesis during endochondral ossification. The vertebrate Gli zinc finger proteins are putative transcription factors that respond to Hedgehog signaling. In Drosophila, the Gli homolog cubitus interruptus is required for the activation of hedgehog targets and also functions as a repressor of hedgehog expression. We show here that Gli2 mutant mice exhibit severe skeletal abnormalities including cleft palate, tooth defects, absence of vertebral body and intervertebral discs, and shortened limbs and sternum. Interestingly, Gli2 and Gli3 (C.-c. Hui and A. L. Joyner (1993). Nature Genet. 3, 241-246) mutant mice exhibit different subsets of skeletal defects indicating that they implement specific functions in the development of the neural crest, somite and lateral plate mesoderm derivatives. Although Gli2 and Gli3 are not functionally equivalent, double mutant analysis indicates that, in addition to their specific roles, they also serve redundant functions during skeletal development. The role of Gli2 and Gli3 in Hedgehog signaling during skeletal development is discussed.</jats:p>",
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