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Human Molecular Genetics, 2002, Vol. 11, No. 26 3345-3350
© 2002 Oxford University Press

Expression analyses and interaction with the anaphase promoting complex protein Apc2 suggest a role for inversin in primary cilia and involvement in the cell cycle

David Morgan1,{dagger}, Lorraine Eley1,{dagger}, John Sayer2, Tom Strachan1,*, Laura M. Yates1, A. Scott Craighead1 and Judith A. Goodship1

1Institute of Human Genetics, University of Newcastle, International Centre for Life, Central Parkway, Newcastle upon Tyne. NE1 3BZ and 2Department of Physiological Sciences, University of Newcastle, Medical School, Framlington Place, University of Newcastle upon Tyne, Newcastle upon Tyne. NE2 4HH

Received September 5, 2002; Accepted October 15, 2002

Homozygous inv mice lack a functional inversin protein and exhibit situs inversus plus severe cystic changes in the kidney and pancreas. Although the inversin sequence has provided few clues to its function, we and others have previously identified calmodulin as a binding partner. We now provide evidence that inversin interacts with the anaphase promoting complex protein Apc2. As expected of an Apc2 target, inversin possesses D-boxes and site-directed mutagenesis of the well-conserved D-box residues abrogates inversin–Apc2 interaction. An inversin-specific antibody reveals a dynamic expression pattern throughout the cell cycle and strong expression in the primary cilia of renal epithelium. Our data support a role for inversin in primary cilia and involvement in the cell cycle. Mutations of the proteins polaris, cystin and polycystin-2 which are expressed in renal epithelium primary cilia, lead to renal cystic changes. Aberrant cell proliferation is also involved in cyst development. The data reported here suggest that inversin may provide a link between these two mechanisms.

* To whom correspondence should be addressed at: Institute of Human Genetics, University of Newcastle upon Tyne, International Centre for Life, Central Parkway, Newcastle upon Tyne. NE1 3BZ, UK. Tel.: +44 1912418616; Fax: +44 1912418699; Email: tom.strachan{at}ncl.ac.uk

{dagger} The authors wish it to be known that, in their opinion, these two authors should be considered as joint First Authors.


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