Literature DB >> 25810209

Delineation of clinical features in Wiedemann-Steiner syndrome caused by KMT2A mutations.

N Miyake1, Y Tsurusaki1, E Koshimizu1, N Okamoto2, T Kosho3, N J Brown4,5, T Y Tan5,6, P J J Yap5, H Suzumura7, T Tanaka8, T Nagai9, M Nakashima1, H Saitsu1, N Niikawa10, N Matsumoto1.   

Abstract

Wiedemann-Steiner syndrome (WSS) is an autosomal dominant congenital anomaly syndrome characterized by hairy elbows, dysmorphic facial appearances (hypertelorism, thick eyebrows, downslanted and vertically narrow palpebral fissures), pre- and post-natal growth deficiency, and psychomotor delay. WSS is caused by heterozygous mutations in KMT2A (also known as MLL), a gene encoding a histone methyltransferase. Here, we identify six novel KMT2A mutations in six WSS patients, with four mutations occurring de novo. Interestingly, some of the patients were initially diagnosed with atypical Kabuki syndrome, which is caused by mutations in KMT2D or KDM6A, genes also involved in histone methylation. KMT2A mutations and clinical features are summarized in our six patients together with eight previously reported patients. Furthermore, clinical comparison of the two syndromes is discussed in detail.
© 2015 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  KDM6A; KMT2A; KMT2D; Kabuki syndrome; Wiedemann-Steiner syndrome; clinical comparison

Mesh:

Substances:

Year:  2015        PMID: 25810209     DOI: 10.1111/cge.12586

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


  22 in total

1.  Kabuki syndrome genes KMT2D and KDM6A: functional analyses demonstrate critical roles in craniofacial, heart and brain development.

Authors:  Peter M Van Laarhoven; Leif R Neitzel; Anita M Quintana; Elizabeth A Geiger; Elaine H Zackai; David E Clouthier; Kristin B Artinger; Jeffrey E Ming; Tamim H Shaikh
Journal:  Hum Mol Genet       Date:  2015-05-13       Impact factor: 6.150

Review 2.  Intellectual Disability: When the Hypertrichosis Is a Clue.

Authors:  Lidia Pezzani; Donatella Milani; Gianluca Tadini
Journal:  J Pediatr Genet       Date:  2015-09-28

3.  Molecular and cellular issues of KMT2A variants involved in Wiedemann-Steiner syndrome.

Authors:  Nicolas Lebrun; Irina Giurgea; Alice Goldenberg; Anne Dieux; Alexandra Afenjar; Jamal Ghoumid; Bertrand Diebold; Léo Mietton; Audrey Briand-Suleau; Pierre Billuart; Thierry Bienvenu
Journal:  Eur J Hum Genet       Date:  2017-12-04       Impact factor: 4.246

4.  Patients with a Kabuki syndrome phenotype demonstrate DNA methylation abnormalities.

Authors:  Nara Sobreira; Martha Brucato; Li Zhang; Christine Ladd-Acosta; Chrissie Ongaco; Jane Romm; Kimberly F Doheny; Regina C Mingroni-Netto; Debora Bertola; Chong A Kim; Ana Ba Perez; Maria I Melaragno; David Valle; Vera A Meloni; Hans T Bjornsson
Journal:  Eur J Hum Genet       Date:  2017-11-07       Impact factor: 4.246

Review 5.  Epigenetic Mistakes in Neurodevelopmental Disorders.

Authors:  Giuseppina Mastrototaro; Mattia Zaghi; Alessandro Sessa
Journal:  J Mol Neurosci       Date:  2017-03-02       Impact factor: 3.444

6.  Wiedemann-Steiner Syndrome: A Rare Differential Diagnosis of Neurodevelopmental Delay and Dysmorphic Features.

Authors:  Kursat Bora Carman; Emre Kaplan; Cefa Nil Aslan; Sinem Kocagil; Oguz Cilinigr; Coskun Yarar
Journal:  J Pediatr Genet       Date:  2020-09-23

Review 7.  Regulation, functions and transmission of bivalent chromatin during mammalian development.

Authors:  Trisha A Macrae; Julie Fothergill-Robinson; Miguel Ramalho-Santos
Journal:  Nat Rev Mol Cell Biol       Date:  2022-08-26       Impact factor: 113.915

8.  Missense variants causing Wiedemann-Steiner syndrome preferentially occur in the KMT2A-CXXC domain and are accurately classified using AlphaFold2.

Authors:  Tinna Reynisdottir; Kimberley Jade Anderson; Leandros Boukas; Hans Tomas Bjornsson
Journal:  PLoS Genet       Date:  2022-06-21       Impact factor: 6.020

9.  RNA Sequencing and Pathway Analysis Identify Important Pathways Involved in Hypertrichosis and Intellectual Disability in Patients with Wiedemann-Steiner Syndrome.

Authors:  Léo Mietton; Nicolas Lebrun; Irina Giurgea; Alice Goldenberg; Benjamin Saintpierre; Juliette Hamroune; Alexandra Afenjar; Pierre Billuart; Thierry Bienvenu
Journal:  Neuromolecular Med       Date:  2018-07-16       Impact factor: 3.843

Review 10.  Reprogramming of the epigenome in neurodevelopmental disorders.

Authors:  Khadija D Wilson; Elizabeth G Porter; Benjamin A Garcia
Journal:  Crit Rev Biochem Mol Biol       Date:  2021-10-02       Impact factor: 8.697

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