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Human Molecular Genetics Advance Access originally published online on September 30, 2004
Human Molecular Genetics 2004 13(22):2727-2735; doi:10.1093/hmg/ddh311
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Human Molecular Genetics, Vol. 13, No. 22 © Oxford University Press 2004; all rights reserved

Pituitary hypoplasia and respiratory distress syndrome in Prop1 knockout mice

Igor O. Nasonkin1,{dagger}, Robert D. Ward2, Lori T. Raetzman1, Audrey F. Seasholtz2,3,4, Thomas L. Saunders5,6, Patrick J. Gillespie6,{ddagger} and Sally A. Camper1,2,5,6,*

1Department of Human Genetics, 2Program in Cellular and Molecular Biology, 3Department of Biological Chemistry, 4Mental Health Research Institute, 5Department of Internal Medicine and 6Transgenic Animal Model Core, University of Michigan Medical School, Ann Arbor, MI 48109, USA

Received May 28, 2004; Revised September 13, 2004; Accepted September 20, 2004

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.

* To whom correspondence should be addressed at: 4301 MSRB III, 1500 W. Med. Center Drive Ann Arbor, MI 48109-0638, USA. Tel: +1 7347630682; Fax: +1 7347637672; Email: scamper{at}umich.edu


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