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Human Molecular Genetics Advance Access published online on March 3, 2004
Human Molecular Genetics, doi:10.1093/hmg/ddh102
© 2004 by Oxford University Press
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©2004 Oxford University Press

Article

Huntingtin Interacting Protein 1 mutations lead to abnormal hematopoiesis, spinal defects and cataracts

Katherine I. Oravecz-Wilson 1, Mark J. Kiel 2, Lina Li 1, Dinesh S. Rao 1, Djenann Saint-Dic 1, Priti D. Kumar 1, Melissa M. Provot 1, Kurt D. Hankenson 3, Venkat N. Reddy 4, Andrew P. Lieberman 5, Sean J. Morrison 2, and Theodora S. Ross 1*

1 Department of Internal Medicine, University of Michigan Medical School, Ann Arbor, MI 48109
2 Department of Internal Medicine, University of Michigan Medical School, Ann Arbor, MI 48109; Howard Hughes Medical Institute, University of Michigan Medical School, Ann Arbor, MI 48109
3 Orthopaedic Research Laboratory, University of Michigan Medical School, Ann Arbor, MI 48109
4 Kellogg Eye Center, University of Michigan Medical School, Ann Arbor, MI 48109
5 Department of Pathology, University of Michigan Medical School, Ann Arbor, MI 48109

* To whom correspondence should be addressed. E-mail: tsross{at}umich.edu.


  Abstract

Huntingtin Interacting Protein 1 (HIP1) binds clathrin and AP2, is overexpressed in multiple human tumors, and transforms fibroblasts. The function of HIP1 is unknown although it is thought to play a fundamental role in clathrin trafficking. Gene targeted Hip1-/- mice develop premature testicular degeneration and severe spinal deformities. But although HIP1 is expressed in many tissues including the spleen and bone marrow and was part of a leukemogenic translocation, its role in hematopoiesis has not been examined. In this study we report that three different mutations of murine Hip1 lead to hematopoietic abnormalities reflected by diminished early progenitor frequencies and resistance to 5-FU induced bone marrow toxicity. Two of the Hip1 mutant lines also display the previously described spinal defects. These observations indicate that, in addition to being required for the survival/proliferation of cancer cells and germline progenitors, HIP1 is also required for the survival/proliferation of diverse types of somatic cells, including hematopoietic progenitors.


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