Literature DB >> 29926465

IL11RA-related Crouzon-like autosomal recessive craniosynostosis in 10 new patients: Resemblances and differences.

E Brischoux-Boucher1, A Trimouille2, G Baujat3, A Goldenberg4, E Schaefer5, B Guichard6, P Hannequin7, G Paternoster8, S Baer5, C Cabrol1, E Weber9, G Godfrin10, M Lenoir11, D Lacombe2, C Collet12, L Van Maldergem1,13,14.   

Abstract

By describing 10 new patients recruited in centres for Human Genetics, we further delineate the clinical spectrum of a Crouzon-like craniosynostosis disorder, officially termed craniosynostosis and dental anomalies (MIM614188). Singularly, it is inherited according to an autosomal recessive mode of inheritance. We identified six missense mutations in IL11RA, a gene encoding the alpha subunit of interleukin 11 receptor, 4 of them being novel, including 2 in the Ig-like C2-type domain. A subset of patients had an associated connective tissue disorder with joint hypermobility and intervertebral discs fragility. A smaller number of teeth anomalies than that previously reported in the two large series of patients evaluated in dental institutes points toward an ascertainment bias.
© 2018 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  Crouzon syndrome; IL11RA; craniosynostosis; hyperlaxity

Mesh:

Substances:

Year:  2018        PMID: 29926465     DOI: 10.1111/cge.13409

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  12 in total

1.  A variant in IL6ST with a selective IL-11 signaling defect in human and mouse.

Authors:  Tobias Schwerd; Freia Krause; Stephen R F Twigg; Andrew O M Wilkie; Dirk Schmidt-Arras; Holm H Uhlig; Dominik Aschenbrenner; Yin-Huai Chen; Uwe Borgmeyer; Miryam Müller; Santiago Manrique; Neele Schumacher; Steven A Wall; Jonathan Jung; Timo Damm; Claus-Christian Glüer; Jürgen Scheller; Stefan Rose-John; E Yvonne Jones; Arian Laurence
Journal:  Bone Res       Date:  2020-06-11       Impact factor: 13.567

2.  The structure of the extracellular domains of human interleukin 11α receptor reveals mechanisms of cytokine engagement.

Authors:  Riley D Metcalfe; Kaheina Aizel; Courtney O Zlatic; Paul M Nguyen; Craig J Morton; Daisy Sio-Seng Lio; Heung-Chin Cheng; Renwick C J Dobson; Michael W Parker; Paul R Gooley; Tracy L Putoczki; Michael D W Griffin
Journal:  J Biol Chem       Date:  2020-04-24       Impact factor: 5.157

Review 3.  IL-11 in cardiac and renal fibrosis: Late to the party but a central player.

Authors:  Benjamin Corden; Eleonora Adami; Mark Sweeney; Sebastian Schafer; Stuart A Cook
Journal:  Br J Pharmacol       Date:  2020-02-22       Impact factor: 8.739

Review 4.  Interleukin-11 signaling underlies fibrosis, parenchymal dysfunction, and chronic inflammation of the airway.

Authors:  Benjamin Ng; Stuart A Cook; Sebastian Schafer
Journal:  Exp Mol Med       Date:  2020-12-01       Impact factor: 8.718

5.  Ameliorative Effects of Osthole on Experimental Renal Fibrosis in vivo and in vitro by Inhibiting IL-11/ERK1/2 Signaling.

Authors:  Fan Wu; Yan Zhao; Qingqing Shao; Ke Fang; Ruolan Dong; Shujun Jiang; Fuer Lu; Jinlong Luo; Guang Chen
Journal:  Front Pharmacol       Date:  2021-05-13       Impact factor: 5.810

Review 6.  New insights into IL-6 family cytokines in metabolism, hepatology and gastroenterology.

Authors:  Maria D Giraldez; David Carneros; Christoph Garbers; Stefan Rose-John; Matilde Bustos
Journal:  Nat Rev Gastroenterol Hepatol       Date:  2021-07-01       Impact factor: 46.802

Review 7.  Structural Understanding of Interleukin 6 Family Cytokine Signaling and Targeted Therapies: Focus on Interleukin 11.

Authors:  Riley D Metcalfe; Tracy L Putoczki; Michael D W Griffin
Journal:  Front Immunol       Date:  2020-07-16       Impact factor: 7.561

8.  A variant in IL6ST with a selective IL-11 signaling defect in human and mouse.

Authors:  Tobias Schwerd; Freia Krause; Stephen R F Twigg; Andrew O M Wilkie; Dirk Schmidt-Arras; Holm H Uhlig; Dominik Aschenbrenner; Yin-Huai Chen; Uwe Borgmeyer; Miryam Müller; Santiago Manrique; Neele Schumacher; Steven A Wall; Jonathan Jung; Timo Damm; Claus-Christian Glüer; Jürgen Scheller; Stefan Rose-John; E Yvonne Jones; Arian Laurence
Journal:  Bone Res       Date:  2020-06-11       Impact factor: 13.567

9.  Similarities and differences between IL11 and IL11RA1 knockout mice for lung fibro-inflammation, fertility and craniosynostosis.

Authors:  Benjamin Ng; Anissa A Widjaja; Sivakumar Viswanathan; Jinrui Dong; Sonia P Chothani; Stella Lim; Shamini G Shekeran; Jessie Tan; Narelle E McGregor; Emma C Walker; Natalie A Sims; Sebastian Schafer; Stuart A Cook
Journal:  Sci Rep       Date:  2021-07-08       Impact factor: 4.379

Review 10.  Different roles of interleukin 6 and interleukin 11 in the liver: implications for therapy.

Authors:  Anissa A Widjaja; Sonia P Chothani; Stuart A Cook
Journal:  Hum Vaccin Immunother       Date:  2020-06-12       Impact factor: 3.452

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