Literature DB >> 24736975

Generation of mice encoding a conditional null allele of Gcm2.

Ziqiang Yuan1, Evan E Opas, Chakravarthy Vrikshajanani, Steven K Libutti, Michael A Levine.   

Abstract

Glial cells missing homolog 2 (GCM2) is a transcription factor that is expressed predominately in the pharyngeal pouches and, at later stages, in the developing and mature parathyroid glands. In humans, loss of GCM2 function, either through recessive apomorphic mutations or dominant inhibitor mutations in the human GCM2 gene, leads to isolated hypoparathyroidism. In mice, homozygous disruption of Gcm2 by conventional gene targeting results in parathyroid aplasia and hypoparathyroidism. In this study, we report the generation and functional characterization of mice encoding a conditional null allele of Gcm2. We demonstrate the functional integrity of the conditional Gcm2 allele and report successful in vivo deletion of exon 2 using Cre recombinase. The mice with conditional deletion of Gcm2 displayed phenotypes similar to those previously described for a conventional Gcm2 knockout, including perinatal lethality, hypocalemia, low or undetectable serum levels of parathyroid hormone, and absent parathyroid glands. The production of a conditional mutant allele for Gcm2 represents a valuable resource for the study of the temporal- and spatial-specific roles for Gcm2, and for understanding the postnatal activities of GCM2 protein.

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Year:  2014        PMID: 24736975      PMCID: PMC4086301          DOI: 10.1007/s11248-014-9799-7

Source DB:  PubMed          Journal:  Transgenic Res        ISSN: 0962-8819            Impact factor:   2.788


  43 in total

Review 1.  Chronicles of a switch hunt: gcm genes in development.

Authors:  M Wegner; D Riethmacher
Journal:  Trends Genet       Date:  2001-05       Impact factor: 11.639

2.  Gcm2 and Foxn1 mark early parathyroid- and thymus-specific domains in the developing third pharyngeal pouch.

Authors:  J Gordon; A R Bennett; C C Blackburn; N R Manley
Journal:  Mech Dev       Date:  2001-05       Impact factor: 1.882

Review 3.  FGF23 and the parathyroid.

Authors:  Justin Silver; Tally Naveh-Many
Journal:  Adv Exp Med Biol       Date:  2012       Impact factor: 2.622

4.  Interactions between calcium and phosphorus in the regulation of the production of fibroblast growth factor 23 in vivo.

Authors:  Stephen J Quinn; Alex R B Thomsen; Jian L Pang; Lakshmi Kantham; Hans Bräuner-Osborne; Martin Pollak; David Goltzman; Edward M Brown
Journal:  Am J Physiol Endocrinol Metab       Date:  2012-12-11       Impact factor: 4.310

5.  MafB interacts with Gcm2 and regulates parathyroid hormone expression and parathyroid development.

Authors:  Akiyo Kamitani-Kawamoto; Michito Hamada; Takashi Moriguchi; Masashi Miyai; Fumie Saji; Ikuji Hatamura; Keizo Nishikawa; Hiroshi Takayanagi; Seiji Hitoshi; Kazuhiro Ikenaka; Toshihiko Hosoya; Yoshiki Hotta; Satoru Takahashi; Kohsuke Kataoka
Journal:  J Bone Miner Res       Date:  2011-10       Impact factor: 6.741

6.  A missense glial cells missing homolog B (GCMB) mutation, Asn502His, causes autosomal dominant hypoparathyroidism.

Authors:  Samantha M Mirczuk; Michael R Bowl; M Andrew Nesbit; Treena Cranston; Carl Fratter; Jeremy Allgrove; Caroline Brain; Rajesh V Thakker
Journal:  J Clin Endocrinol Metab       Date:  2010-05-12       Impact factor: 5.958

7.  Genetic ablation of parathyroid glands reveals another source of parathyroid hormone.

Authors:  T Günther; Z F Chen; J Kim; M Priemel; J M Rueger; M Amling; J M Moseley; T J Martin; D J Anderson; G Karsenty
Journal:  Nature       Date:  2000-07-13       Impact factor: 49.962

8.  A novel mutation in the GCM2 gene causing severe congenital isolated hypoparathyroidism.

Authors:  Daniel Doyle; Susan M Kirwin; Katia Sol-Church; Michael A Levine
Journal:  J Pediatr Endocrinol Metab       Date:  2012       Impact factor: 1.634

9.  Increased osteopontin contributes to inhibition of bone mineralization in FGF23-deficient mice.

Authors:  Quan Yuan; Yan Jiang; Xuefeng Zhao; Tadatoshi Sato; Michael Densmore; Christiane Schüler; Reinhold G Erben; Marc D McKee; Beate Lanske
Journal:  J Bone Miner Res       Date:  2014-03       Impact factor: 6.741

10.  Thymus-associated parathyroid hormone has two cellular origins with distinct endocrine and immunological functions.

Authors:  Zhijie Liu; Alison Farley; Lizhen Chen; Beth J Kirby; Christopher S Kovacs; C Clare Blackburn; Nancy R Manley
Journal:  PLoS Genet       Date:  2010-12-23       Impact factor: 5.917

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  3 in total

1.  GCM2-Activating Mutations in Familial Isolated Hyperparathyroidism.

Authors:  Bin Guan; James M Welch; Julie C Sapp; Hua Ling; Yulong Li; Jennifer J Johnston; Electron Kebebew; Leslie G Biesecker; William F Simonds; Stephen J Marx; Sunita K Agarwal
Journal:  Am J Hum Genet       Date:  2016-10-13       Impact factor: 11.025

2.  Gcm2 regulates the maintenance of parathyroid cells in adult mice.

Authors:  Taku Yamada; Norifumi Tatsumi; Akane Anraku; Hideaki Suzuki; Sahoko Kamejima; Taketo Uchiyama; Ichiro Ohkido; Takashi Yokoo; Masataka Okabe
Journal:  PLoS One       Date:  2019-01-24       Impact factor: 3.240

3.  Limits to in vivo fate changes of epithelia in thymus and parathyroid by ectopic expression of transcription factors Gcm2 and Foxn1.

Authors:  Daisuke Nagakubo; Mayumi Hirakawa; Norimasa Iwanami; Thomas Boehm
Journal:  Sci Rep       Date:  2022-08-08       Impact factor: 4.996

  3 in total

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