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Human Molecular Genetics Advance Access originally published online on March 3, 2004
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Human Molecular Genetics, 2004, Vol. 13, No. 8 851-867
DOI: 10.1093/hmg/ddh102
Human Molecular Genetics, Vol. 13, No. 8 © Oxford University Press 2004; all rights reserved

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

Katherine I. Oravecz-Wilson1, Mark J. Kiel1,2, Lina Li1, Dinesh S. Rao1, Djenann Saint-Dic1, Priti D. Kumar1, Melissa M. Provot1, Kurt D. Hankenson3, Venkat N. Reddy4, Andrew P. Lieberman5, Sean J. Morrison1,2 and Theodora S. Ross1,*

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

Received December 31, 2003; Accepted February 16, 2004

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. Yet, 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.

* To whom correspondence should be addressed. Tel: +1 7346155509; Email: tsross{at}umich.edu


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