一次線毛とBBS4は出生後の下垂体発生に必要

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Updated: Jan 13, 2026

Dissection and Coronal Slice Preparation of Developing Mouse Pituitary Gland

Dissection and Coronal Slice Preparation of Developing Mouse Pituitary Gland

Published on: November 16, 2017

Kathryn M Brewer¹, Katlyn K Brewer¹, Nicholas C Richardson¹

¹Department of Biology, Indiana University - Indianapolis, Indianapolis, IN, USA.

Developmental biology

|January 9, 2026

Abstract:

Primary cilia orchestrate several signaling pathways, and their disruption results in pleiotropic disorders called ciliopathies. Bardet-Beidl syndrome (BBS), one ciliopathy, provides insights into cilia function in many tissues. Using a mouse model of BBS, Bbs4 knockout (Bbs4^-/-), we found that adult Bbs4^-/- pituitaries are hypoplastic and have increased gonadotroph populations. Similarly, pituitary deletion of IFT88, required for ciliogenesis, attenuated growth and increased gonadotrophs. The developing Bbs4^-/- pituitary experienced mildly reduced Hedgehog (HH) signaling. Isolated Bbs4^-/- pituitary stem cells exhibited reduced HH signal responsiveness and expression of stem cell markers. These data demonstrate that cilia and BBS function are necessary for pituitary growth. We propose that altered cilia-mediated patterning of the pituitary contribute to physiological features of ciliopathies such as BBS.

キーワード:

Bardet-Biedl症候群線毛線毛病ゴナドトロピン下垂体

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Evaluation of Planar-Cell-Polarity Phenotypes in Ciliopathy Mouse Mutant Cochlea

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