Multiple tumors due to mosaic genome-wide paternal uniparental disomy.


Journal

Pediatric blood & cancer
ISSN: 1545-5017
Titre abrégé: Pediatr Blood Cancer
Pays: United States
ID NLM: 101186624

Informations de publication

Date de publication:
06 2019
Historique:
received: 08 11 2018
revised: 01 03 2019
accepted: 05 03 2019
pubmed: 19 3 2019
medline: 21 12 2019
entrez: 19 3 2019
Statut: ppublish

Résumé

Mosaic genome-wide paternal uniparental disomy is an infrequently described disorder in which affected individuals have signs and symptoms that may resemble Beckwith-Wiedemann syndrome. In addition, they can develop multiple benign and malignant tumors throughout life. Routine molecular diagnostics may not detect the (characteristic) low level of mosaicism, and the diagnosis is likely to be missed. Genetic counseling and a life-long alertness for the development of tumors is indicated. We describe the long diagnostic process of a patient who already had a tumor at birth and developed multiple tumors in childhood and adulthood. Furthermore, we offer clues to recognize the entity.

Identifiants

pubmed: 30882989
doi: 10.1002/pbc.27715
doi:

Types de publication

Case Reports Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

e27715

Informations de copyright

© 2019 The Authors. Pediatric Blood & Cancer Published by Wiley Periodicals, Inc.

Auteurs

Floor A M Postema (FAM)

Department of Pediatrics, Emma Children's Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, the Netherlands.
Princess Máxima Center for Pediatric oncology, Utrecht, the Netherlands.

Jet Bliek (J)

Department of Clinical Genetics, Amsterdam UMC, University of Amsterdam, Amsterdam, the Netherlands.

Carel J M van Noesel (CJM)

Department of Pathology, Amsterdam UMC, University of Amsterdam, Amsterdam, the Netherlands.

Laura J C M van Zutven (LJCM)

Department of Clinical Genetics, Erasmus Medical Center, Rotterdam, the Netherlands.

Jan C Oosterwijk (JC)

Department of Genetics, University Medical Center Groningen, Groningen, the Netherlands.

Saskia M J Hopman (SMJ)

Department of Genetics, University Medical Center Utrecht, Utrecht, the Netherlands.

Johannes H M Merks (JHM)

Princess Máxima Center for Pediatric oncology, Utrecht, the Netherlands.

Raoul C Hennekam (RC)

Department of Pediatrics, Emma Children's Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, the Netherlands.

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Classifications MeSH