Literature DB >> 35040536

Near complete deletion of KMT2D in a college student.

Catherine Gooch1, Jaclyn Paige Souder2, Matthew L Tedder3, Jennifer Kerkhof4, Jennifer A Lee3, Raymond J Louie3, Bekim Sadikovic4,5, Robin S Fletcher3, Nathaniel H Robin1.   

Abstract

Pathogenic variants in KMT2D are typically associated with Kabuki syndrome (KS), a rare multisystem disorder. KS is characterized by facial dysmorphisms, intellectual disability, skeletal and dermatoglyphic differences, and poor growth. Seventy percent of individuals with clinically diagnosed KS have a confirmed pathogenic variant in KMT2D or less commonly KDM6A. The majority of mutations found in KMT2D are de novo nonsense or frameshift, with deletions and duplications rarely reported in the literature. Here, we present the case of near complete deletion of KMT2D in a college student with normal intelligence discovered via exome sequencing and EpiSign methylation testing. This case provides evidence that large deletions in KMT2D are compatible with normal intelligence and presents EpiSign as a method for discovering molecular causes of KS not identified by traditional molecular testing.
© 2022 Wiley Periodicals LLC.

Entities:  

Keywords:  KMT2D; Kabuki syndrome; deletion; molecular testing

Mesh:

Year:  2022        PMID: 35040536      PMCID: PMC8995339          DOI: 10.1002/ajmg.a.62652

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  34 in total

1.  MLL2 mutation spectrum in 45 patients with Kabuki syndrome.

Authors:  Aimée D C Paulussen; Alexander P A Stegmann; Marinus J Blok; Demis Tserpelis; Crool Posma-Velter; Yvonne Detisch; Eric E J G L Smeets; Annemieke Wagemans; Jaap J P Schrander; Marie-José H van den Boogaard; Jasper van der Smagt; Arie van Haeringen; Irene Stolte-Dijkstra; Wilhelmina S Kerstjens-Frederikse; Grazia M Mancini; Marja W Wessels; Raoul C M Hennekam; Maaike Vreeburg; Joep Geraedts; Thomy de Ravel; Jean-Pierre Fryns; Hubert J Smeets; Koenraad Devriendt; Constance T R M Schrander-Stumpel
Journal:  Hum Mutat       Date:  2010-12-07       Impact factor: 4.878

2.  Spectrum of MLL2 (ALR) mutations in 110 cases of Kabuki syndrome.

Authors:  Mark C Hannibal; Kati J Buckingham; Sarah B Ng; Jeffrey E Ming; Anita E Beck; Margaret J McMillin; Heidi I Gildersleeve; Abigail W Bigham; Holly K Tabor; Heather C Mefford; Joseph Cook; Koh-ichiro Yoshiura; Tadashi Matsumoto; Naomichi Matsumoto; Noriko Miyake; Hidefumi Tonoki; Kenji Naritomi; Tadashi Kaname; Toshiro Nagai; Hirofumi Ohashi; Kenji Kurosawa; Jia-Woei Hou; Tohru Ohta; Deshung Liang; Akira Sudo; Colleen A Morris; Siddharth Banka; Graeme C Black; Jill Clayton-Smith; Deborah A Nickerson; Elaine H Zackai; Tamim H Shaikh; Dian Donnai; Norio Niikawa; Jay Shendure; Michael J Bamshad
Journal:  Am J Med Genet A       Date:  2011-06-10       Impact factor: 2.802

3.  Speech and language in a genotyped cohort of individuals with Kabuki syndrome.

Authors:  Angela T Morgan; Cristina Mei; Annette Da Costa; Joanne Fifer; Damien Lederer; Valérie Benoit; Margaret J McMillin; Kati J Buckingham; Michael J Bamshad; Kate Pope; Susan M White
Journal:  Am J Med Genet A       Date:  2015-03-08       Impact factor: 2.802

4.  Genomic DNA Methylation Signatures Enable Concurrent Diagnosis and Clinical Genetic Variant Classification in Neurodevelopmental Syndromes.

Authors:  Erfan Aref-Eshghi; David I Rodenhiser; Laila C Schenkel; Hanxin Lin; Cindy Skinner; Peter Ainsworth; Guillaume Paré; Rebecca L Hood; Dennis E Bulman; Kristin D Kernohan; Kym M Boycott; Philippe M Campeau; Charles Schwartz; Bekim Sadikovic
Journal:  Am J Hum Genet       Date:  2018-01-04       Impact factor: 11.025

5.  AMPK is a direct adenylate charge-regulated protein kinase.

Authors:  Jonathan S Oakhill; Rohan Steel; Zhi-Ping Chen; John W Scott; Naomi Ling; Shanna Tam; Bruce E Kemp
Journal:  Science       Date:  2011-06-17       Impact factor: 47.728

6.  Mutation Update for Kabuki Syndrome Genes KMT2D and KDM6A and Further Delineation of X-Linked Kabuki Syndrome Subtype 2.

Authors:  Nina Bögershausen; Vincent Gatinois; Vera Riehmer; Hülya Kayserili; Jutta Becker; Michaela Thoenes; Pelin Özlem Simsek-Kiper; Mouna Barat-Houari; Nursel H Elcioglu; Dagmar Wieczorek; Sigrid Tinschert; Guillaume Sarrabay; Tim M Strom; Aurélie Fabre; Gareth Baynam; Elodie Sanchez; Gudrun Nürnberg; Umut Altunoglu; Yline Capri; Bertrand Isidor; Didier Lacombe; Carole Corsini; Valérie Cormier-Daire; Damien Sanlaville; Fabienne Giuliano; Kim-Hanh Le Quan Sang; Honorine Kayirangwa; Peter Nürnberg; Thomas Meitinger; Koray Boduroglu; Barbara Zoll; Stanislas Lyonnet; Andreas Tzschach; Alain Verloes; Nataliya Di Donato; Isabelle Touitou; Christian Netzer; Yun Li; David Geneviève; Gökhan Yigit; Bernd Wollnik
Journal:  Hum Mutat       Date:  2016-07-07       Impact factor: 4.878

7.  Molecular, clinical and neuropsychological study in 31 patients with Kabuki syndrome and KMT2D mutations.

Authors:  N Lehman; A C Mazery; A Visier; C Baumann; D Lachesnais; Y Capri; A Toutain; S Odent; M Mikaty; C Goizet; E Taupiac; M L Jacquemont; E Sanchez; E Schaefer; V Gatinois; L Faivre; D Minot; H Kayirangwa; K-H L Q Sang; N Boddaert; S Bayard; D Lacombe; S Moutton; I Touitou; M Rio; J Amiel; S Lyonnet; D Sanlaville; M C Picot; D Geneviève
Journal:  Clin Genet       Date:  2017-05-18       Impact factor: 4.438

8.  Yes-associated protein (YAP) promotes cell survival by inhibiting proapoptotic dendrin signaling.

Authors:  Kirk N Campbell; Jenny S Wong; Ritu Gupta; Katsuhiko Asanuma; Marius Sudol; John Cijiang He; Peter Mundel
Journal:  J Biol Chem       Date:  2013-05-10       Impact factor: 5.157

9.  Correction: "Congenital hyperinsulinism as the presenting feature of Kabuki syndrome: clinical and molecular characterization of 10 affected individuals".

Authors:  Kai Lee Yap; Amy E Knight Johnson; David Fischer; Priscilla Kandikatla; Jacea Deml; Viswateja Nelakuditi; Sara Halbach; George S Jeha; Lindsay C Burrage; Olaf Bodamer; Valeria C Benavides; Andrea M Lewis; Sian Ellard; Pratik Shah; Declan Cody; Alejandro Diaz; Aishwarya Devarajan; Lisa Truong; Siri Atma W Greeley; Diva D De Leon; Andrew C Edmondson; Soma Das; Paul Thornton; Darrel Waggoner; Daniela Del Gaudio
Journal:  Genet Med       Date:  2019-01       Impact factor: 8.822

10.  Congenital hyperinsulinism as the presenting feature of Kabuki syndrome: clinical and molecular characterization of 9 affected individuals.

Authors:  Kai Lee Yap; Amy E Knight Johnson; David Fischer; Priscilla Kandikatla; Jacea Deml; Viswateja Nelakuditi; Sara Halbach; George S Jeha; Lindsay C Burrage; Olaf Bodamer; Valeria C Benavides; Andrea M Lewis; Sian Ellard; Pratik Shah; Declan Cody; Alejandro Diaz; Aishwarya Devarajan; Lisa Truong; Siri Atma W Greeley; Diva D De Leó-Crutchlow; Andrew C Edmondson; Soma Das; Paul Thornton; Darrel Waggoner; Daniela Del Gaudio
Journal:  Genet Med       Date:  2018-06-15       Impact factor: 8.822

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