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Monozygotic Twins With Neurofibromatosis Type 1 (NF1) Display Differences in Methylation of NF1 Gene Promoter Elements, 5' Untranslated region, Exon and Intron 1

Published online by Cambridge University Press:  21 February 2012

Anja Harder*
Affiliation:
Department of Neuropathology, Charité — Universitätsmedizin Berlin, Germany; Institute of Neuropathy, University Hospital Münster, Münster, Germany. anja.harder@ukmuenster.de
Sabrina Titze
Affiliation:
Department of Neuropathology, Charité — Universitätsmedizin Berlin, Germany.
Lena Herbst
Affiliation:
Institute of Medical Genetics, Charité — Universitätsmedizin Berlin, Germany.
Thomas Harder
Affiliation:
Clinic of Obstetrics, Division of Experimental Obstetrics, Charité — Universitätsmedizin Berlin, Germany.
Katrin Guse
Affiliation:
Department of Neuropathology, Charité — Universitätsmedizin Berlin, Germany.
Sigrid Tinschert
Affiliation:
Institute of Clinical Genetics, Medical Faculty Carl Gustav Carus, Technical University Dresden, Germany.
Dieter Kaufmann
Affiliation:
Institute of Human Genetics, University of Ulm, Germany.
Thorsten Rosenbaum
Affiliation:
Children's Hospital, Klinikum Lüdenscheid, Lüdenscheid, Germany.
Victor Felix Mautner
Affiliation:
Laboratory for Tumor Biology and Developmental Disorders, Department of Maxillofacial Surgery, University Hospital Eppendorf, Hamburg, Germany.
Elke Windt
Affiliation:
Otto Heubner Centre for Child Care, Department of Social Pediatrics, Charité — Universitätsmedizin Berlin, Germany.
Ute Wahlländer-Danek
Affiliation:
Private practice, Großhesselohe, Munich, Germany.
Katharina Wimmer
Affiliation:
Department of Medical Genetics, Molecular and Clinical Pharmacology, Clinical Genetics Section, Medical University Innsbruck, Austria.
Stefan Mundlos
Affiliation:
Institute of Medical Genetics, Charité — Universitätsmedizin Berlin, Germany.
Hartmut Peters
Affiliation:
Institute of Medical Genetics, Charité — Universitätsmedizin Berlin, Germany.
*
*Address for correspondence: Anja Harder, Institute of Neuropathology, University Hospital Münster, Domagkstrasse 19, 48129 Münster, Germany.

Abstract

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Neurofibromatosis type 1 (NF1) is a common autosomal dominant disorder caused by heterozygotic inactivation of the NF1 tumor suppressor gene at 17q11.2. The associated phenotypes are highly variable, and modifying genes have been proposed to explain at least in part the intriguing expressivity. Given that haploinsufficiency of the NF1 gene product neurofibromin is responsible for some of the clinical manifestations, variations in expression of the wildtype NF1 allele might modify the phenotype. We therefore investigated epigenetic molecular modifications that could result in variable expression of the normal NF1 allele. To exclude confounding by DNA sequence variations, we analyzed monozygotic twin pairs with NF1 who presented with several discordant features. We fine-mapped the methylation pattern of a nearly 1 kb NF1 promoter region in lymphocytes of 8 twin pairs. All twin pairs showed significant intra-pair differences in methylation, especially of specific promoter subregions such as 5'UTR, exon 1 and intron 1 (+7 to +622), transcription factor binding sites and promoter elements like NF1HCS. Furthermore, we detected significant intra-pair differences in cytosine methylation for the region from -249 to -234 with regard to discordance for optic glioma with a higher grade of methylation in glioma cases. In conclusion, our findings of epigenetic differences of the NF1 promoter in leukocytes within mono zygotic twin pairs may serve as a proof of principle for other tissues. The results point towards a role of methylation patterns of the normal NF1 allele for expression differences and for modification of the NF1 phenotype.

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Articles
Copyright
Copyright © Cambridge University Press 2010