Literature DB >> 30680470

Mutated SON putatively causes a cancer syndrome comprising high-risk medulloblastoma combined with café-au-lait spots.

Celine Chiu1, Stefanie Loth1, Michaela Kuhlen1, Sebastian Ginzel1, Jörg Schaper2, Thorsten Rosenbaum3, Torsten Pietsch4, Arndt Borkhardt1, Jessica I Hoell5.   

Abstract

Medulloblastoma is the most frequent malignant brain tumor in childhood. This highly malignant neoplasm occurs usually before 10 years of age and more frequently in boys. The 5-year event-free survival rate for high-risk medulloblastoma is low at 62% despite a multimodal therapy including surgical resection, radiation therapy and chemotherapy. We report the case of a boy, who was born to consanguineous parents. Prominently, he had multiple café-au-lait spots. At the age of 3 years he was diagnosed with a high-risk metastatic medulloblastoma. The patient died only 11 months after diagnosis of a fulminant relapse presenting as meningeal and spinal dissemination. Whole-exome sequencing of germline DNA was employed to detect the underlying mutation for this putative cancer syndrome presenting with the combination of medulloblastoma and skin alterations. After screening all possible homozygous gene SNVs, we identified a mutation of SON, an essential protein in cell cycle regulation and cell proliferation, as the most likely genetic cause.

Entities:  

Keywords:  Cancer syndrome; Medulloblastoma; SON mutation; Skin alteration

Mesh:

Substances:

Year:  2019        PMID: 30680470     DOI: 10.1007/s10689-019-00121-z

Source DB:  PubMed          Journal:  Fam Cancer        ISSN: 1389-9600            Impact factor:   2.375


  18 in total

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2.  Thirteen new patients with guanidinoacetate methyltransferase deficiency and functional characterization of nineteen novel missense variants in the GAMT gene.

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Journal:  Hum Mutat       Date:  2014-03-06       Impact factor: 4.878

3.  Constitutional mismatch repair-deficiency and whole-exome sequencing as the means of the rapid detection of the causative MSH6 defect.

Authors:  J I Hoell; M Gombert; S Ginzel; S Loth; P Landgraf; V Käfer; M Streiter; A Prokop; M Weiss; R Thiele; A Borkhardt
Journal:  Klin Padiatr       Date:  2014-11-28       Impact factor: 1.349

Review 4.  Childhood cancer predisposition syndromes-A concise review and recommendations by the Cancer Predisposition Working Group of the Society for Pediatric Oncology and Hematology.

Authors:  Tim Ripperger; Stefan S Bielack; Arndt Borkhardt; Ines B Brecht; Birgit Burkhardt; Gabriele Calaminus; Klaus-Michael Debatin; Hedwig Deubzer; Uta Dirksen; Cornelia Eckert; Angelika Eggert; Miriam Erlacher; Gudrun Fleischhack; Michael C Frühwald; Astrid Gnekow; Gudrun Goehring; Norbert Graf; Helmut Hanenberg; Julia Hauer; Barbara Hero; Simone Hettmer; Katja von Hoff; Martin Horstmann; Juliane Hoyer; Thomas Illig; Peter Kaatsch; Roland Kappler; Kornelius Kerl; Thomas Klingebiel; Udo Kontny; Uwe Kordes; Dieter Körholz; Ewa Koscielniak; Christof M Kramm; Michaela Kuhlen; Andreas E Kulozik; Britta Lamottke; Ivo Leuschner; Dietmar R Lohmann; Andrea Meinhardt; Markus Metzler; Lüder H Meyer; Olga Moser; Michaela Nathrath; Charlotte M Niemeyer; Rainer Nustede; Kristian W Pajtler; Claudia Paret; Mareike Rasche; Dirk Reinhardt; Olaf Rieß; Alexandra Russo; Stefan Rutkowski; Brigitte Schlegelberger; Dominik Schneider; Reinhard Schneppenheim; Martin Schrappe; Christopher Schroeder; Dietrich von Schweinitz; Thorsten Simon; Monika Sparber-Sauer; Claudia Spix; Martin Stanulla; Doris Steinemann; Brigitte Strahm; Petra Temming; Kathrin Thomay; Andre O von Bueren; Peter Vorwerk; Olaf Witt; Marcin Wlodarski; Willy Wössmann; Martin Zenker; Stefanie Zimmermann; Stefan M Pfister; Christian P Kratz
Journal:  Am J Med Genet A       Date:  2017-02-07       Impact factor: 2.802

5.  Treatment of Children and Adolescents With Metastatic Medulloblastoma and Prognostic Relevance of Clinical and Biologic Parameters.

Authors:  André O von Bueren; Rolf-Dieter Kortmann; Katja von Hoff; Carsten Friedrich; Martin Mynarek; Klaus Müller; Tobias Goschzik; Anja Zur Mühlen; Nicolas Gerber; Monika Warmuth-Metz; Niels Soerensen; Frank Deinlein; Martin Benesch; Isabella Zwiener; Robert Kwiecien; Andreas Faldum; Udo Bode; Gudrun Fleischhack; Volker Hovestadt; Marcel Kool; David Jones; Paul Northcott; Joachim Kuehl; Stefan Pfister; Torsten Pietsch; Stefan Rutkowski
Journal:  J Clin Oncol       Date:  2016-10-31       Impact factor: 44.544

6.  A method and server for predicting damaging missense mutations.

Authors:  Ivan A Adzhubei; Steffen Schmidt; Leonid Peshkin; Vasily E Ramensky; Anna Gerasimova; Peer Bork; Alexey S Kondrashov; Shamil R Sunyaev
Journal:  Nat Methods       Date:  2010-04       Impact factor: 28.547

7.  Re-innervation patterns by peptidergic Substance-P, non-peptidergic P2X3, and myelinated NF-200 nerve fibers in epidermis and dermis of rats with neuropathic pain.

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Journal:  Exp Neurol       Date:  2012-12-07       Impact factor: 5.330

Review 8.  Smelling the roses and seeing the light: gene therapy for ciliopathies.

Authors:  Jeremy C McIntyre; Corey L Williams; Jeffrey R Martens
Journal:  Trends Biotechnol       Date:  2013-04-17       Impact factor: 19.536

9.  A framework for variation discovery and genotyping using next-generation DNA sequencing data.

Authors:  Mark A DePristo; Eric Banks; Ryan Poplin; Kiran V Garimella; Jared R Maguire; Christopher Hartl; Anthony A Philippakis; Guillermo del Angel; Manuel A Rivas; Matt Hanna; Aaron McKenna; Tim J Fennell; Andrew M Kernytsky; Andrey Y Sivachenko; Kristian Cibulskis; Stacey B Gabriel; David Altshuler; Mark J Daly
Journal:  Nat Genet       Date:  2011-04-10       Impact factor: 38.330

10.  Deriving the consequences of genomic variants with the Ensembl API and SNP Effect Predictor.

Authors:  William McLaren; Bethan Pritchard; Daniel Rios; Yuan Chen; Paul Flicek; Fiona Cunningham
Journal:  Bioinformatics       Date:  2010-06-18       Impact factor: 6.937

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  2 in total

1.  SON DNA-binding protein mediates macrophage autophagy and responses to intracellular infection.

Authors:  David J Gregory; Glen M DeLoid; Sharon L Salmon; Dennis W Metzger; Igor Kramnik; Lester Kobzik
Journal:  FEBS Lett       Date:  2020-06-19       Impact factor: 4.124

2.  Establishing the phenotypic spectrum of ZTTK syndrome by analysis of 52 individuals with variants in SON.

Authors:  Alexander J M Dingemans; Kim M G Truijen; Jung-Hyun Kim; Zahide Alaçam; Laurence Faivre; Kathleen M Collins; Erica H Gerkes; Mieke van Haelst; Ingrid M B H van de Laar; Kristin Lindstrom; Mathilde Nizon; James Pauling; Edyta Heropolitańska-Pliszka; Astrid S Plomp; Caroline Racine; Rani Sachdev; Margje Sinnema; Jon Skranes; Hermine E Veenstra-Knol; Eline A Verberne; Anneke T Vulto-van Silfhout; Marlon E F Wilsterman; Eun-Young Erin Ahn; Bert B A de Vries; Lisenka E L M Vissers
Journal:  Eur J Hum Genet       Date:  2021-09-15       Impact factor: 5.351

  2 in total

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