Literature DB >> 26762561

EZH2 mutation in an adolescent with Weaver syndrome developing acute myeloid leukemia and secondary hemophagocytic lymphohistiocytosis.

Jakob Usemann1,2, Thomas Ernst3, Vivien Schäfer3, Kai Lehmberg4, Karl Seeger1.   

Abstract

Weaver syndrome is an overgrowth syndrome characterized by pre- and postnatal overgrowth with distinctive craniofacial appearance. Mutations in the enhancer of zeste homolog 2 (EZH2) gene were found to cause Weaver syndrome, and have been associated with hematologic malignancies, including acute myeloid leukemia (AML). We present the first report of a patient with Weaver syndrome, who developed AML and harbored an EZH2 mutation. The clinical course of the 16-year-old female adolescent patient was complicated by a secondary hemophagocytic lymphohistiocytosis. Genomic DNA was isolated from bone marrow cells at AML diagnosis. Polymerase chain reactions were performed with primers covering all exons of the EZH2 gene. We found a novel heterozygous EZH2 mutation within exon 5 that caused an amino acid change from proline to leucine at position 132 (p.Pro132Leu) within the catalytic D1 domain. Analysis of a remission sample also showed this mutation, indicating a germline mutation. It remains to be elucidated whether EZH2 mutations contribute to disease severity in specific AML cases.
© 2016 Wiley Periodicals, Inc.

Entities:  

Keywords:  EZH2; Weaver syndrome; acute myeloid leukemia; hemophagocytic lymphohistiocytosis; malignancy; overgrowth

Mesh:

Substances:

Year:  2016        PMID: 26762561     DOI: 10.1002/ajmg.a.37562

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


  7 in total

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Authors:  Illja J Diets; Roos van der Donk; Kristina Baltrunaite; Esmé Waanders; Margot R F Reijnders; Alexander J M Dingemans; Rolph Pfundt; Anneke T Vulto-van Silfhout; Laurens Wiel; Christian Gilissen; Julien Thevenon; Laurence Perrin; Alexandra Afenjar; Caroline Nava; Boris Keren; Sarah Bartz; Bethany Peri; Gea Beunders; Nienke Verbeek; Koen van Gassen; Isabelle Thiffault; Maxime Cadieux-Dion; Lina Huerta-Saenz; Matias Wagner; Vassiliki Konstantopoulou; Julia Vodopiutz; Matthias Griese; Annekatrien Boel; Bert Callewaert; Han G Brunner; Tjitske Kleefstra; Nicoline Hoogerbrugge; Bert B A de Vries; Vivian Hwa; Andrew Dauber; Jayne Y Hehir-Kwa; Roland P Kuiper; Marjolijn C J Jongmans
Journal:  Am J Hum Genet       Date:  2019-03-28       Impact factor: 11.025

2.  Ezh2 Mutations Found in the Weaver Overgrowth Syndrome Cause a Partial Loss of H3K27 Histone Methyltransferase Activity.

Authors:  Julian C Lui; Kevin M Barnes; Lijin Dong; Shanna Yue; Evan Graber; Robert Rapaport; Andrew Dauber; Ola Nilsson; Jeffrey Baron
Journal:  J Clin Endocrinol Metab       Date:  2018-04-01       Impact factor: 5.958

Review 3.  Germline mutations predisposing to diffuse large B-cell lymphoma.

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Review 5.  Novel de novo mutation affecting two adjacent aminoacids in the EED gene in a patient with Weaver syndrome.

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7.  Constitutional 2p16.3 deletion including MSH6 and FBXO11 in a boy with developmental delay and diffuse large B-cell lymphoma.

Authors:  N van Engelen; F van Dijk; E Waanders; A Buijs; M A Vermeulen; J L C Loeffen; R P Kuiper; M C J Jongmans
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  7 in total

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