Literature DB >> 27621108

Mild pituitary phenotype in 3- and 12-month-old Aip-deficient male mice.

Anne-Lise Lecoq1, Philippe Zizzari2, Mirella Hage1, Lyvianne Decourtye3, Clovis Adam4, Say Viengchareun1, Johannes D Veldhuis5, Valérie Geoffroy6, Marc Lombès1, Virginie Tolle2, Anne Guillou7, Auli Karhu8, Laurent Kappeler3, Philippe Chanson9, Peter Kamenický10.   

Abstract

Germline mutations in the aryl hydrocarbon receptor-interacting protein (AIP) gene predispose humans to pituitary adenomas, particularly of the somatotroph lineage. Mice with global heterozygous inactivation of Aip (Aip(+/-)) also develop pituitary adenomas but differ from AIP-mutated patients by the high penetrance of pituitary disease. The endocrine phenotype of these mice is unknown. The aim of this study was to determine the endocrine phenotype of Aip(+/-) mice by assessing the somatic growth, ultradian pattern of GH secretion and IGF1 concentrations of longitudinally followed male mice at 3 and 12 months of age. As the early stages of pituitary tumorigenesis are controversial, we also studied the pituitary histology and somatotroph cell proliferation in these mice. Aip(+/-) mice did not develop gigantism but exhibited a leaner phenotype than wild-type mice. Analysis of GH pulsatility by deconvolution in 12-month-old Aip(+/-) mice showed a mild increase in total GH secretion, a conserved GH pulsatility pattern, but a normal IGF1 concentration. No pituitary adenomas were detected up to 12 months of age. An increased ex vivo response to GHRH of pituitary explants from 3-month-old Aip(+/-) mice, together with areas of enlarged acini identified on reticulin staining in the pituitary of some Aip(+/-) mice, was suggestive of somatotroph hyperplasia. Global heterozygous Aip deficiency in mice is accompanied by subtle increase in GH secretion, which does not result in gigantism. The absence of pituitary adenomas in 12-month-old Aip(+/-) mice in our experimental conditions demonstrates the important phenotypic variability of this congenic mouse model.
© 2016 Society for Endocrinology.

Entities:  

Keywords:  AIP; aryl hydrocarbon receptor-interacting protein; germline mutations; growth hormone; pituitary adenoma

Mesh:

Substances:

Year:  2016        PMID: 27621108     DOI: 10.1530/JOE-16-0190

Source DB:  PubMed          Journal:  J Endocrinol        ISSN: 0022-0795            Impact factor:   4.286


  5 in total

1.  Prkar1a haploinsufficiency ameliorates the growth hormone excess phenotype in Aip-deficient mice.

Authors:  Marie Helene Schernthaner-Reiter; Giampaolo Trivellin; Thomas Roetzer; Johannes A Hainfellner; Matthew F Starost; Constantine A Stratakis
Journal:  Hum Mol Genet       Date:  2020-10-10       Impact factor: 6.150

Review 2.  The 3PAs: An Update on the Association of Pheochromocytomas, Paragangliomas, and Pituitary Tumors.

Authors:  Paraskevi Xekouki; Ana Brennand; Ben Whitelaw; Karel Pacak; Constantine A Stratakis
Journal:  Horm Metab Res       Date:  2018-10-01       Impact factor: 2.936

3.  Somatotroph-Specific Aip-Deficient Mice Display Pretumorigenic Alterations in Cell-Cycle Signaling.

Authors:  Mary P Gillam; Cheol Ryong Ku; Yang Jong Lee; Jean Kim; Se Hoon Kim; Sue Ji Lee; Byungjin Hwang; JaeHyung Koo; Rhonda D Kineman; Hiroaki Kiyokawa; Eun Jig Lee
Journal:  J Endocr Soc       Date:  2017-01-13

4.  RET signalling provides tumorigenic mechanism and tissue specificity for AIP-related somatotrophinomas.

Authors:  Miguel Chenlo; Fernando Oroz-Gonjar; Márta Korbonits; Clara V Alvarez; Angela R Garcia-Rendueles; Antonia Solomou; Anisha Mistry; Sayka Barry; Carles Gaston-Massuet; Montserrat Garcia-Lavandeira; Sihara Perez-Romero; Maria Suarez-Fariña; Alberto Pradilla-Dieste; Carlos Dieguez; Patrick Mehlen
Journal:  Oncogene       Date:  2021-09-29       Impact factor: 9.867

Review 5.  Novel Insights into Pituitary Tumorigenesis: Genetic and Epigenetic Mechanisms.

Authors:  Vinaya Srirangam Nadhamuni; Márta Korbonits
Journal:  Endocr Rev       Date:  2020-12-01       Impact factor: 19.871

  5 in total

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