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Molecular and Cellular Biology, April 2000, p. 2498-2504, Vol. 20, No. 7
0270-7306/00/$04.00+0
Copyright © 2000, American Society for Microbiology. All rights reserved.

Expression and Functional Analysis of Uch-L3 during Mouse Development

Laurie Jo Kurihara, Ekaterina Semenova, John M. Levorse, and Shirley M. Tilghman*

Howard Hughes Medical Institute and Department of Molecular Biology, Princeton University, Princeton, New Jersey 08544

Received 16 December 1999/Accepted 21 December 1999

Mice homozygous for the s1Acrg deletion at the Ednrb locus arrest at embryonic day 8.5. To determine the molecular basis of this defect, we initiated positional cloning of the s1Acrg minimal region. The mouse Uch-L3 (ubiquitin C-terminal hydrolase L3) gene was mapped within the s1Acrg minimal region. Because Uch-L3 transcripts were present in embryonic structures relevant to the s1Acrg phenotype, we created a targeted mutation in Uch-L3 to address its role during development and its possible contribution to the s1Acrg phenotype. Mice homozygous for the mutation Uch-L3Delta 3-7 were viable, with no obvious developmental or histological abnormalities. Although high levels of Uch-L3 RNA were detected in testes and thymus, Uch-L3Delta 3-7 homozygotes were fertile, and no defect in intrathymic T-cell differentiation was detected. We conclude that the s1Acrg phenotype is either complex and multigenic or due to the loss of another gene within the region. We propose that Uch-L3 may be functionally redundant with its homologue Uch-L1.

* Corresponding author. Mailing address: Department of Molecular Biology, Princeton University, Princeton, NJ 08544. Phone: (609) 258-2900. Fax: (609) 258-3345. E-mail: stilghman{at}molbio.princeton.edu.

Molecular and Cellular Biology, April 2000, p. 2498-2504, Vol. 20, No. 7
0270-7306/00/$04.00+0
Copyright © 2000, American Society for Microbiology. All rights reserved.


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