Cited by 131Circadian rhythm and melatoninGenetics and Neurodevelopmental DisordersSleep and Wakefulness Research

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Fragile X-Related Proteins Regulate Mammalian Circadian Behavioral Rhythms

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Zhang, Jing, Fang, Zhe, Jud, Corinne, Vansteensel, Mariska J., Kaasik, Krista, Lee, Cheng Chi, Albrecht, Urs, Tamanini, Filippo, Meijer, Johanna H., Oostra, Ben A., Nelson, David L.·2008·The American Journal of Human Genetics

DOI 10.1016/j.ajhg.2008.06.003PMC PMC2443847

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Abstract

Fragile X syndrome results from the absence of the fragile X mental retardation 1 (FMR1) gene product (FMRP). FMR1 has two paralogs in vertebrates: fragile X related gene 1 and 2 (FXR1 and FXR2). Here we show that Fmr1/Fxr2 double knockout (KO) and Fmr1 KO/Fxr2 heterozygous animals exhibit a loss of rhythmic activity in a light:dark (LD) cycle, and that Fmr1 or Fxr2 KO mice display a shorter free-running period of locomotor activity in total darkness (DD). Molecular analysis and in vitro electrophysiological studies suggest essentially normal function of cells in the suprachiasmatic nucleus (SCN) in Fmr1/Fxr2 double KO mice. However, the cyclical patterns of abundance of several core clock component messenger (m) RNAs are altered in the livers of double KO mice. Furthermore, FXR2P alone or FMRP and FXR2P together can increase PER1- or PER2-mediated BMAL1-Neuronal PAS2 (NPAS2) transcriptional activity in a dose-dependent manner. These data collectively demonstrate that FMR1 and FXR2 are required for the presence of rhythmic circadian behavior in mammals and suggest that this role may be relevant to sleep and other behavioral alterations observed in fragile X patients.

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Authors: Zhang, J., Fang, Z., Jud, C., Vansteensel, M. J., Kaasik, K., Lee, C. C., Albrecht, U., Tamanini, F., Meijer, J. H., Oostra, B. A., & Nelson, D. L.
Affiliations: Baylor College of Medicine (US)
University of Fribourg (CH)
Leiden University Medical Center (NL)
The University of Texas Health Science Center at Houston (US)
Erasmus MC (NL)
Corresponding author: Nelson, David L.
ORCID: Zhang, Jing
Vansteensel, Mariska J.
Kaasik, Krista
Albrecht, Urs
Meijer, Johanna H.
Nelson, David L.
Venue: The American Journal of Human Genetics
Publisher: Elsevier BV
Published: 2008-07
Volume: 83
Issue: 1
Pages: 43-52
ISSN: 0002-9297 (ISSN-L)
DOI: 10.1016/j.ajhg.2008.06.003
PMC: PMC2443847
OpenAlex: W2107880079
Cited by: 131
References: 39
Citations by year: 2026: 3 · 2025: 9 · 2024: 7 · 2023: 8 · 2022: 5 · 2021: 5 · 2020: 6 · 2019: 8 · 2018: 8 · 2017: 8 · 2016: 6 · 2015: 6 · 2014: 9 · 2013: 8 · 2012: 4
FWCI: 4.59
Type: journal article
Language: en
Open access: bronze
doi.org
Licence: www.elsevier.com/tdm/userlicense/1.0/ (tdm)
www.elsevier.com/open-access/userlicense/1.0/ (vor, 1844 d)
Locations: The American Journal of Human Genetics (publishedVersion)
PubMed Central (publishedVersion)
PubMed (publishedVersion)
EUR Research Repository (Erasmus University Rotterdam) (publishedVersion)
reroDoc Digital Library (submittedVersion)
EUR Research Repository (Erasmus University Rotterdam) (submittedVersion)
RePub (Erasmus University Rotterdam) (submittedVersion, other-oa)
RePub (Erasmus University Rotterdam) (submittedVersion)
Funders: Schweizerischer Nationalfonds zur Förderung der Wissenschaftlichen Forschung
National Institutes of Health
Intellectual and Developmental Disabilities Research Center
National Institute of Child Health and Human Development
Eunice Kennedy Shriver National Institute of Child Health and Human Development (HD 24064, HD-38038, P30HD024064, R01HD038038)
Topics: Life Sciences › Neuroscience › Endocrine and Autonomic Systems › Circadian rhythm and melatonin
Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
Life Sciences › Neuroscience › Cognitive Neuroscience › Sleep and Wakefulness Research
Keywords: fragile X syndrome, FMRP, FXR1, FXR2, circadian rhythm, locomotor activity, suprachiasmatic nucleus, clock genes, Per1, Per2, Bmal1, NPAS2, sleep, liver transcriptome
MeSH terms: Animals, Behavior, Animal, Circadian Rhythm, Electrophysiology, Flavoproteins, Fragile X Syndrome, Gene Expression Regulation, Heterozygote, Liver, Male, Mammals, Mice, Inbred C57BL, Motor Activity, Nerve Tissue Proteins, Neurons, Nuclear Proteins, RNA, Messenger, Suprachiasmatic Nucleus, Time Factors, Transcription Factors, RNA-Binding Proteins, In Situ Hybridization, Mice, Knockout, Cell Cycle Proteins, Mice, Basic Helix-Loop-Helix Transcription Factors, Basic Helix-Loop-Helix Proteins, ARNTL Transcription Factors, Cryptochromes, Period Circadian Proteins

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Fragile X-Related Proteins Regulate Mammalian Circadian Behavioral Rhythms

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