PMID- 22087250 OWN - NLM STAT- MEDLINE DCOM- 20120518 LR - 20211021 IS - 1932-6203 (Electronic) IS - 1932-6203 (Linking) VI - 6 IP - 11 DP - 2011 TI - Elevated levels of the vesicular monoamine transporter and a novel repetitive behavior in the Drosophila model of fragile X syndrome. PG - e27100 LID - 10.1371/journal.pone.0027100 [doi] LID - e27100 AB - Fragile X Syndrome (FXS) is characterized by mental impairment and autism in humans, and it often features hyperactivity and repetitive behaviors. The mechanisms for the disease, however, remain poorly understood. Here we report that the dfmr1 mutant in the Drosophila model of FXS grooms excessively, which may be regulated differentially by two signaling pathways. Blocking metabotropic glutamate receptor signaling enhances grooming in dfmr1 mutant flies, whereas blocking the vesicular monoamine transporter (VMAT) suppresses excessive grooming. dfmr1 mutant flies also exhibit elevated levels of VMAT mRNA and protein. These results suggest that enhanced monoamine signaling correlates with repetitive behaviors and hyperactivity associated with FXS. FAU - Tauber, John M AU - Tauber JM AD - Department of Zoology, University of Oklahoma, Norman, Oklahoma, United States of America. FAU - Vanlandingham, Phillip A AU - Vanlandingham PA FAU - Zhang, Bing AU - Zhang B LA - eng GR - R01 NS060878/NS/NINDS NIH HHS/United States PT - Journal Article PT - Research Support, N.I.H., Extramural PT - Research Support, Non-U.S. Gov't DEP - 20111102 PL - United States TA - PLoS One JT - PloS one JID - 101285081 RN - 0 (Drosophila Proteins) RN - 0 (FMR1 protein, Drosophila) RN - 0 (RNA, Messenger) RN - 0 (Vesicular Monoamine Transport Proteins) RN - 139135-51-6 (Fragile X Mental Retardation Protein) SB - IM MH - Animals MH - Behavior, Animal MH - Disease Models, Animal MH - Drosophila Proteins MH - Drosophila melanogaster/*metabolism MH - Fragile X Mental Retardation Protein MH - Fragile X Syndrome/etiology/*physiopathology MH - Grooming MH - Obsessive Behavior/*etiology MH - RNA, Messenger/analysis MH - Signal Transduction MH - Vesicular Monoamine Transport Proteins/analysis/*metabolism PMC - PMC3206932 COIS- Competing Interests: The authors have declared that no competing interests exist. EDAT- 2011/11/17 06:00 MHDA- 2012/05/19 06:00 PMCR- 2011/11/02 CRDT- 2011/11/17 06:00 PHST- 2011/06/30 00:00 [received] PHST- 2011/10/10 00:00 [accepted] PHST- 2011/11/17 06:00 [entrez] PHST- 2011/11/17 06:00 [pubmed] PHST- 2012/05/19 06:00 [medline] PHST- 2011/11/02 00:00 [pmc-release] AID - PONE-D-11-12273 [pii] AID - 10.1371/journal.pone.0027100 [doi] PST - ppublish SO - PLoS One. 2011;6(11):e27100. doi: 10.1371/journal.pone.0027100. Epub 2011 Nov 2.