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Human Molecular Genetics Advance Access published online on September 30, 2004
Human Molecular Genetics, doi:10.1093/hmg/ddh311
© 2004 by Oxford University Press
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Pituitary hypoplasia and respiratory distress syndrome in Prop1 knockout mice

Igor O. Nasonkin 1, Robert D. Ward 2, Lori T. Raetzman 3, Audrey F. Seasholtz 4, Thomas L. Saunders 5, Patrick J. Gillespie 6, and Sally A. Camper 7*

1 Department of Human Genetics, University of Michigan Medical School, Ann Arbor, MI 48109; Department of Genetics,University of Georgia, Life Sciences Building, Athens, GA 30602
2 Program in Cellular and Molecular Biology, University of Michigan Medical School, Ann Arbor, MI 48109
3 Department of Human Genetics, University of Michigan Medical School, Ann Arbor, MI 48109
4 Program in Cellular and Molecular Biology, University of Michigan Medical School, Ann Arbor, MI 48109; Department Biological Chemistry, University of Michigan Medical School, Ann Arbor, MI 48109; Mental Health Research Institute, University of Michigan Medical School, Ann Arbor, MI 48109
5 Department Internal Medicine, University of Michigan Medical School, Ann Arbor, MI 48109; Transgenic Animal Model Core, University of Michigan Medical School, Ann Arbor, MI 48109
6 Transgenic Animal Model Core, University of Michigan Medical School, Ann Arbor, MI 48109; Transgenic Technologies, Laboratory Animal Resources, Pfizer, 2800 Plymouth Road, Ann Arbor, MI 48105
7 Department of Human Genetics, University of Michigan Medical School, 4301 MSRB III, 1500 W. Med. Ctr. Dr., Ann Arbor, MI 48109-0638; Program in Cellular and Molecular Biology, University of Michigan Medical School, 4301 MSRB III, 1500 W. Med. Ctr. Dr., Ann Arbor, MI 48109-0638; Department Internal Medicine, University of Michigan Medical School, 4301 MSRB III, 1500 W. Med. Ctr. Dr., Ann Arbor, MI 48109-0638; Transgenic Animal Model Core, University of Michigan Medical School, 4301 MSRB III, 1500 W. Med. Ctr. Dr., Ann Arbor, MI 48109-0638

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


  Abstract

Mutations in Prophet of PIT1 (Prop1), one of several homeodomain transcription factors that are required for the development of the anterior pituitary gland, are the predominant cause of MPHD (Multiple Pituitary Hormone Deficiency) in humans. We show that deletion of Prop1 in mice causes severe pituitary hypoplasia with failure of the entire Pit1 lineage and delayed gonadotrope development. The pituitary hormone deficiencies cause secondary endocrine problems and a high rate of perinatal mortality due to respiratory distress. Lung atelectasis in mutants correlates with reduced levels of NKX2.1 and surfactant. Lethality of mice homozygous for either the null allele or a spontaneous hypomorphic allele is strongly influenced by genetic background. Prop1-null mice are an excellent model for MPHD and may be useful for testing the efficacy of pharmaceutical intervention for neonatal respiratory distress.


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