The Drosophila melanogaster homolog of UBE3A is not imprinted in neurons

被引:5
|
作者
Hope, Kevin A. [1 ,2 ]
LeDoux, Mark S. [1 ,2 ]
Reiter, Lawrence T. [1 ,2 ,3 ]
机构
[1] Univ Tennessee, Ctr Hlth Sci, Dept Neurol, 855 Monroe Ave,Link 415, Memphis, TN 38163 USA
[2] Univ Tennessee, Ctr Hlth Sci, Dept Anat & Neurobiol, Memphis, TN 38163 USA
[3] Univ Tennessee, Ctr Hlth Sci, Pediat, Memphis, TN 38163 USA
基金
美国国家卫生研究院;
关键词
Climbing assay; DGRP; Drosophila melanogaster; ERG; imprinting; synaptic transmission; SNPs; UBE3A; PRADER-WILLI SYNDROMES; ANGELMAN-SYNDROME; UBIQUITIN LIGASE; EXPRESSION; UBE3A/E6-AP; DISORDERS; BRAIN; GENE;
D O I
10.1080/15592294.2016.1214783
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
of the In mammals, expression of UBE3A is epigenetically regulated in neurons and expression is restricted to the maternal copy of UBE3A. A recent report claimed that Drosophila melanogaster UBE3A homolog (Dube3a) is preferentially expressed from the maternal allele in fly brain, inferring an imprinting mechanism. However, complex epigenetic regulatory features of the mammalian imprinting center are not present in Drosophila, and allele specific expression of Dube3a has not been documented. We used behavioral and electrophysiological analysis Dube3a loss-of-function allele (Dube3a(15b)) to investigate Dube3a imprinting in fly neurons. We found that motor impairment (climbing ability) and a newly-characterized defect in synaptic transmission are independent of parental inheritance of the Dube3a(15b) allele. Furthermore, expression analysis of coding single nucleotide polymorphisms (SNPs) in Dube3a did not reveal allele specific expression differences among reciprocal crosses. These data indicate that Dube3a is neither imprinted nor preferentially expressed from the maternal allele in fly neurons.
引用
收藏
页码:637 / 642
页数:6
相关论文
共 50 条
  • [31] Unraveling the Roles of UBE3A in Neurodevelopment and Neurodegeneration
    Yang, Xin
    Huang, Yu-Wen Alvin
    INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES, 2025, 26 (05)
  • [32] Spatial and temporal silencing of the human maternal UBE3A gene
    Daily, Jennifer
    Smith, Amanda G.
    Weeber, Edwin J.
    EUROPEAN JOURNAL OF PAEDIATRIC NEUROLOGY, 2012, 16 (06) : 587 - 591
  • [33] Screening of UBE3A gene in patients referred for Angelman Syndrome
    Tzagkaraki, Evmorfia
    Sofocleous, Christalena
    Helen, Fryssira-Kanioura
    Dinopoulos, Argyris
    Goulielmos, Georgios
    Mavrou, Ariadni
    Sofia, Kitsiou-Tzeli
    Kanavakis, Emmanuel
    EUROPEAN JOURNAL OF PAEDIATRIC NEUROLOGY, 2013, 17 (04) : 366 - 373
  • [34] Persistent neuronal Ube3a expression in the suprachiasmatic nucleus of Angelman syndrome model mice
    Jones, Kelly A.
    Han, Ji Eun
    DeBruyne, Jason P.
    Philpot, Benjamin D.
    SCIENTIFIC REPORTS, 2016, 6
  • [35] Ube3a-ATS is an atypical RNA polymerase II transcript that represses the paternal expression of Ube3a
    Meng, Linyan
    Person, Richard E.
    Beaudet, Arthur L.
    HUMAN MOLECULAR GENETICS, 2012, 21 (13) : 3001 - 3012
  • [36] Angelman syndrome-derived neurons display late onset of paternal UBE3A silencing
    Stanurova, Jana
    Neureiter, Anika
    Hiber, Michaela
    Kessler, Hannah de Oliveira
    Stolp, Kristin
    Goetzke, Roman
    Klein, Diana
    Bankfalvi, Agnes
    Klump, Hannes
    Steenpass, Laura
    SCIENTIFIC REPORTS, 2016, 6
  • [37] An Autism-Linked Mutation Disables Phosphorylation Control of UBE3A
    Yi, Jason J.
    Berrios, Janet
    Newbern, Jason M.
    Snider, William D.
    Philpot, Benjamin D.
    Hahn, Klaus M.
    Zylka, Mark J.
    CELL, 2015, 162 (04) : 795 - 807
  • [38] The Neuronal Circuit of the Dorsal Circadian Clock Neurons in Drosophila melanogaster
    Reinhard, Nils
    Schubert, Frank K.
    Bertolini, Enrico
    Hagedorn, Nicolas
    Manoli, Giulia
    Sekiguchi, Manabu
    Yoshii, Taishi
    Rieger, Dirk
    Helfrich-Foerster, Charlotte
    FRONTIERS IN PHYSIOLOGY, 2022, 13
  • [39] Maternal Ube3a Loss Disrupts Sleep Homeostasis But Leaves Circadian Rhythmicity Largely Intact
    Ehlen, J. Christopher
    Jones, Kelly A.
    Pinckney, Lennisha
    Gray, Cloe L.
    Burette, Susan
    Weinberg, Richard J.
    Evans, Jennifer A.
    Brager, Allison J.
    Zylka, Mark J.
    Paul, Ketema N.
    Philpot, Benjamin D.
    DeBruyne, Jason P.
    JOURNAL OF NEUROSCIENCE, 2015, 35 (40) : 13587 - 13598
  • [40] Generation and Characterization of a Novel Angelman Syndrome Mouse Model with a Full Deletion of the Ube3a Gene
    Syding, Linn Amanda
    Kubik-Zahorodna, Agnieszka
    Nickl, Petr
    Novosadova, Vendula
    Kopkanova, Jana
    Kasparek, Petr
    Prochazka, Jan
    Sedlacek, Radislav
    CELLS, 2022, 11 (18)