Gene dosage effects of the imprinted delta-like homologue 1 (dlk1/pref1) in development: implications for the evolution of imprinting.

Genomic imprinting is a normal process that causes genes to be expressed according to parental origin. The selective advantage conferred by imprinting is not understood but is hypothesised to act on dosage-critical genes. Here, we report a unique model in which the consequences of a single, double,...

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Main Authors: Simao Teixeira da Rocha, Marika Charalambous, Shau-Ping Lin, Isabel Gutteridge, Yoko Ito, Dionne Gray, Wendy Dean, Anne C Ferguson-Smith
Format: Article
Language:English
Published: Public Library of Science (PLoS) 2009-02-01
Series:PLoS Genetics
Online Access:https://journals.plos.org/plosgenetics/article/file?id=10.1371/journal.pgen.1000392&type=printable
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author Simao Teixeira da Rocha
Marika Charalambous
Shau-Ping Lin
Isabel Gutteridge
Yoko Ito
Dionne Gray
Wendy Dean
Anne C Ferguson-Smith
author_facet Simao Teixeira da Rocha
Marika Charalambous
Shau-Ping Lin
Isabel Gutteridge
Yoko Ito
Dionne Gray
Wendy Dean
Anne C Ferguson-Smith
author_sort Simao Teixeira da Rocha
collection DOAJ
description Genomic imprinting is a normal process that causes genes to be expressed according to parental origin. The selective advantage conferred by imprinting is not understood but is hypothesised to act on dosage-critical genes. Here, we report a unique model in which the consequences of a single, double, and triple dosage of the imprinted Dlk1/Pref1, normally repressed on the maternally inherited chromosome, can be assessed in the growing embryo. BAC-transgenic mice were generated that over-express Dlk1 from endogenous regulators at all sites of embryonic activity. Triple dosage causes lethality associated with major organ abnormalities. Embryos expressing a double dose of Dlk1, recapitulating loss of imprinting, are growth enhanced but fail to thrive in early life, despite the early growth advantage. Thus, any benefit conferred by increased embryonic size is offset by postnatal lethality. We propose a negative correlation between gene dosage and survival that fixes an upper limit on growth promotion by Dlk1, and we hypothesize that trade-off between growth and lethality might have driven imprinting at this locus.
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id doaj-art-1ec664d8d0ac4ffd9cf9bb3a14b211f9
institution OA Journals
issn 1553-7390
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language English
publishDate 2009-02-01
publisher Public Library of Science (PLoS)
record_format Article
series PLoS Genetics
spelling doaj-art-1ec664d8d0ac4ffd9cf9bb3a14b211f92025-08-20T02:17:24ZengPublic Library of Science (PLoS)PLoS Genetics1553-73901553-74042009-02-0152e100039210.1371/journal.pgen.1000392Gene dosage effects of the imprinted delta-like homologue 1 (dlk1/pref1) in development: implications for the evolution of imprinting.Simao Teixeira da RochaMarika CharalambousShau-Ping LinIsabel GutteridgeYoko ItoDionne GrayWendy DeanAnne C Ferguson-SmithGenomic imprinting is a normal process that causes genes to be expressed according to parental origin. The selective advantage conferred by imprinting is not understood but is hypothesised to act on dosage-critical genes. Here, we report a unique model in which the consequences of a single, double, and triple dosage of the imprinted Dlk1/Pref1, normally repressed on the maternally inherited chromosome, can be assessed in the growing embryo. BAC-transgenic mice were generated that over-express Dlk1 from endogenous regulators at all sites of embryonic activity. Triple dosage causes lethality associated with major organ abnormalities. Embryos expressing a double dose of Dlk1, recapitulating loss of imprinting, are growth enhanced but fail to thrive in early life, despite the early growth advantage. Thus, any benefit conferred by increased embryonic size is offset by postnatal lethality. We propose a negative correlation between gene dosage and survival that fixes an upper limit on growth promotion by Dlk1, and we hypothesize that trade-off between growth and lethality might have driven imprinting at this locus.https://journals.plos.org/plosgenetics/article/file?id=10.1371/journal.pgen.1000392&type=printable
spellingShingle Simao Teixeira da Rocha
Marika Charalambous
Shau-Ping Lin
Isabel Gutteridge
Yoko Ito
Dionne Gray
Wendy Dean
Anne C Ferguson-Smith
Gene dosage effects of the imprinted delta-like homologue 1 (dlk1/pref1) in development: implications for the evolution of imprinting.
PLoS Genetics
title Gene dosage effects of the imprinted delta-like homologue 1 (dlk1/pref1) in development: implications for the evolution of imprinting.
title_full Gene dosage effects of the imprinted delta-like homologue 1 (dlk1/pref1) in development: implications for the evolution of imprinting.
title_fullStr Gene dosage effects of the imprinted delta-like homologue 1 (dlk1/pref1) in development: implications for the evolution of imprinting.
title_full_unstemmed Gene dosage effects of the imprinted delta-like homologue 1 (dlk1/pref1) in development: implications for the evolution of imprinting.
title_short Gene dosage effects of the imprinted delta-like homologue 1 (dlk1/pref1) in development: implications for the evolution of imprinting.
title_sort gene dosage effects of the imprinted delta like homologue 1 dlk1 pref1 in development implications for the evolution of imprinting
url https://journals.plos.org/plosgenetics/article/file?id=10.1371/journal.pgen.1000392&type=printable
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