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@ARTICLE{Chiu:140749,
author = {Chiu, Celine and Loth, Stefanie and Kuhlen, Michaela and
Ginzel, Sebastian and Schaper, Jörg and Rosenbaum, Thorsten
and Pietsch, Torsten and Borkhardt, Arndt and Hoell, Jessica
I},
title = {{M}utated {SON} putatively causes a cancer syndrome
comprising high-risk medulloblastoma combined with
café-au-lait spots.},
journal = {Familial cancer},
volume = {18},
number = {3},
issn = {1389-9600},
address = {Dordrecht [u.a.]},
publisher = {Springer Science + Business Media B.V},
reportid = {DZNE-2020-07071},
pages = {353-358},
year = {2019},
abstract = {Medulloblastoma is the most frequent malignant brain tumor
in childhood. This highly malignant neoplasm occurs usually
before 10 years of age and more frequently in boys. The
5-year event-free survival rate for high-risk
medulloblastoma is low at $62\%$ despite a multimodal
therapy including surgical resection, radiation therapy and
chemotherapy. We report the case of a boy, who was born to
consanguineous parents. Prominently, he had multiple
café-au-lait spots. At the age of 3 years he was diagnosed
with a high-risk metastatic medulloblastoma. The patient
died only 11 months after diagnosis of a fulminant relapse
presenting as meningeal and spinal dissemination.
Whole-exome sequencing of germline DNA was employed to
detect the underlying mutation for this putative cancer
syndrome presenting with the combination of medulloblastoma
and skin alterations. After screening all possible
homozygous gene SNVs, we identified a mutation of SON, an
essential protein in cell cycle regulation and cell
proliferation, as the most likely genetic cause.},
keywords = {Cafe-au-Lait Spots: genetics / Cerebellar Neoplasms:
genetics / Child, Preschool / Consanguinity / DNA-Binding
Proteins: genetics / Fatal Outcome / Humans / Male /
Medulloblastoma: genetics / Minor Histocompatibility
Antigens: genetics / Pedigree / Point Mutation / Syndrome /
DNA-Binding Proteins (NLM Chemicals) / Minor
Histocompatibility Antigens (NLM Chemicals) / SON protein,
human (NLM Chemicals)},
cin = {Brainbank (Bonn)},
ddc = {610},
cid = {I:(DE-2719)1011009},
pnm = {345 - Population Studies and Genetics (POF3-345)},
pid = {G:(DE-HGF)POF3-345},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:30680470},
doi = {10.1007/s10689-019-00121-z},
url = {https://pub.dzne.de/record/140749},
}