Literature DB >> 33300088

Identification of a novel de novo KMT2B variant in a Greek dystonia patient via exome sequencing genotype-phenotype correlations of all published cases.

Chrysoula Marogianni1, Despoina Georgouli1, Katerina Dadouli2, Panagiotis Ntellas3, Dimitrios Rikos1, Georgios M Hadjigeorgiou1,4, Cleanthi Spanaki5, Georgia Xiromerisiou6.   

Abstract

Mutations in Lysine-Specific Histone Methyltransferase 2B gene (KMT2B) have been reported to be associated with isolated and complex early-onset generalized dystonia. We describe clinico-genetic features on a Greek patient with a novel de novo variant and demonstrate the phenotypic spectrum of KMT2B variants. We performed whole exome sequencing (WES), in a Greek patient with sporadic generalized dystonia. Additionally, we performed a systematic review of all published cases with KMT2B variants. The patient presented with isolated and mild generalized dystonia. We identified a novel splice site variant that was confirmed by Sanger sequencing and was not found in parents. This is the first reported KMT2B variant, in the Greek population. This case report further highlights the growing trend of identifying genetic diseases previously restricted to few cases in many different ethnic groups worldwide via exome sequencing. In the systematic review, we evaluated the mutation pathogenicity in all previously reported cases to investigate possible phenotype-genotype correlations. Greater mutation numbers in different populations will be important and mutation-specific functional studies will be essential to identify the pathogenicity of the various KMT2B variants.

Entities:  

Keywords:  Early age onset; Good response to DBS; KMT2B dystonia; Mild generalized dystonia; Novel mutation

Mesh:

Substances:

Year:  2020        PMID: 33300088     DOI: 10.1007/s11033-020-06057-3

Source DB:  PubMed          Journal:  Mol Biol Rep        ISSN: 0301-4851            Impact factor:   2.316


  24 in total

1.  KMT2B: A new twist in dystonia genetics.

Authors:  Bettina Balint; Enza Maria Valente
Journal:  Mov Disord       Date:  2017-02-20       Impact factor: 10.338

2.  KMT2B rare missense variants in generalized dystonia.

Authors:  Michael Zech; Robert Jech; Petra Havránková; Anna Fečíková; Riccardo Berutti; Dušan Urgošík; David Kemlink; Tim M Strom; Jan Roth; Evžen Růžička; Juliane Winkelmann
Journal:  Mov Disord       Date:  2017-05-18       Impact factor: 10.338

3.  Haploinsufficiency of KMT2B, Encoding the Lysine-Specific Histone Methyltransferase 2B, Results in Early-Onset Generalized Dystonia.

Authors:  Michael Zech; Sylvia Boesch; Esther M Maier; Ingo Borggraefe; Katharina Vill; Franco Laccone; Veronika Pilshofer; Andres Ceballos-Baumann; Bader Alhaddad; Riccardo Berutti; Werner Poewe; Tobias B Haack; Bernhard Haslinger; Tim M Strom; Juliane Winkelmann
Journal:  Am J Hum Genet       Date:  2016-11-10       Impact factor: 11.025

4.  The early-onset torsion dystonia gene (DYT1) encodes an ATP-binding protein.

Authors:  L J Ozelius; J W Hewett; C E Page; S B Bressman; P L Kramer; C Shalish; D de Leon; M F Brin; D Raymond; D P Corey; S Fahn; N J Risch; A J Buckler; J F Gusella; X O Breakefield
Journal:  Nat Genet       Date:  1997-09       Impact factor: 38.330

5.  Mutations in the histone methyltransferase gene KMT2B cause complex early-onset dystonia.

Authors:  Esther Meyer; Keren J Carss; Julia Rankin; John M E Nichols; Detelina Grozeva; Agnel P Joseph; Niccolo E Mencacci; Apostolos Papandreou; Joanne Ng; Serena Barral; Adeline Ngoh; Hilla Ben-Pazi; Michel A Willemsen; David Arkadir; Angela Barnicoat; Hagai Bergman; Sanjay Bhate; Amber Boys; Niklas Darin; Nicola Foulds; Nicholas Gutowski; Alison Hills; Henry Houlden; Jane A Hurst; Zvi Israel; Margaret Kaminska; Patricia Limousin; Daniel Lumsden; Shane McKee; Shibalik Misra; Shekeeb S Mohammed; Vasiliki Nakou; Joost Nicolai; Magnus Nilsson; Hardev Pall; Kathryn J Peall; Gregory B Peters; Prab Prabhakar; Miriam S Reuter; Patrick Rump; Reeval Segel; Margje Sinnema; Martin Smith; Peter Turnpenny; Susan M White; Dagmar Wieczorek; Sarah Wiethoff; Brian T Wilson; Gidon Winter; Christopher Wragg; Simon Pope; Simon J H Heales; Deborah Morrogh; Alan Pittman; Lucinda J Carr; Belen Perez-Dueñas; Jean-Pierre Lin; Andre Reis; William A Gahl; Camilo Toro; Kailash P Bhatia; Nicholas W Wood; Erik-Jan Kamsteeg; Wui K Chong; Paul Gissen; Maya Topf; Russell C Dale; Jonathan R Chubb; F Lucy Raymond; Manju A Kurian
Journal:  Nat Genet       Date:  2016-12-19       Impact factor: 38.330

6.  Broad domains of histone 3 lysine 4 trimethylation are associated with transcriptional activation in CA1 neurons of the hippocampus during memory formation.

Authors:  Bridget E Collins; J David Sweatt; Celeste B Greer
Journal:  Neurobiol Learn Mem       Date:  2019-04-16       Impact factor: 2.877

7.  The relevance of gene panels in movement disorders diagnosis: A lab perspective.

Authors:  Chiara Reale; Celeste Panteghini; Miryam Carecchio; Barbara Garavaglia
Journal:  Eur J Paediatr Neurol       Date:  2018-01-31       Impact factor: 3.140

8.  Targeted next-generation sequencing for the molecular diagnosis of hereditary angioedema due to C1-inhibitor deficiency.

Authors:  Gedeon Loules; Maria Zamanakou; Faidra Parsopoulou; Sofia Vatsiou; Fotis Psarros; Dorottya Csuka; Grzegorz Porebski; Krystyna Obtulowicz; Anna Valerieva; Maria Staevska; Alberto López-Lera; Margarita López-Trascasa; Dumitru Moldovan; Markus Magerl; Marcus Maurer; Matthaios Speletas; Henriette Farkas; Anastasios E Germenis
Journal:  Gene       Date:  2018-05-16       Impact factor: 3.688

Review 9.  Dystonia: an update on phenomenology, classification, pathogenesis and treatment.

Authors:  Bettina Balint; Kailash P Bhatia
Journal:  Curr Opin Neurol       Date:  2014-08       Impact factor: 5.710

10.  A general framework for estimating the relative pathogenicity of human genetic variants.

Authors:  Martin Kircher; Daniela M Witten; Preti Jain; Brian J O'Roak; Gregory M Cooper; Jay Shendure
Journal:  Nat Genet       Date:  2014-02-02       Impact factor: 38.330

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  1 in total

1.  Early-onset generalized dystonia caused by a new mutation in the KMT2B gene: Case report

Authors:  Yully Andrea Rangel; Eugenia Espinosa
Journal:  Biomedica       Date:  2022-09-02       Impact factor: 1.173

  1 in total

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