{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,21]],"date-time":"2026-05-21T04:03:22Z","timestamp":1779336202674,"version":"3.51.4"},"reference-count":54,"publisher":"The Company of Biologists","issue":"5","license":[{"start":{"date-parts":[[2020,8,3]],"date-time":"2020-08-03T00:00:00Z","timestamp":1596412800000},"content-version":"vor","delay-in-days":2313,"URL":"http:\/\/creativecommons.org\/licenses\/by\/3.0"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2014,5,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Wolfram syndrome (WFS) is a progressive neurodegenerative disease characterized by diabetes insipidus, diabetes mellitus, optic atrophy, and deafness. WFS1 and WFS2 are caused by recessive mutations in the genes Wolfram Syndrome 1 (WFS1) and CDGSH iron sulfur domain 2 (CISD2), respectively. To explore the function of CISD2, we performed genetic studies in flies with altered expression of its Drosophila orthologue, cisd2. Surprisingly, flies with strong ubiquitous RNAi-mediated knockdown of cisd2 had no obvious signs of altered life span, stress resistance, locomotor behavior or several other phenotypes. We subsequently found in a targeted genetic screen, however, that altered function of cisd2 modified the effects of overexpressing the fly orthologues of two lysosomal storage disease genes, palmitoyl-protein thioesterase 1 (PPT1 in humans, Ppt1 in flies) and ceroid-lipofuscinosis, neuronal 3 (CLN3 in humans, cln3 in flies), on eye morphology in flies. We also found that cln3 modified the effects of overexpressing Ppt1 in the eye and that overexpression of cln3 interacted with a loss of function mutation in cisd2 to disrupt locomotor ability in flies. Follow-up multi-species bioinformatic analyses suggested that a gene network centered on CISD2, PPT1 and CLN3 might impact disease through altered carbohydrate metabolism, protein folding and endopeptidase activity. Human genetic studies indicated that copy number variants (duplications and deletions) including CLN3, and possibly another gene in the CISD2\/PPT1\/CLN3 network, are over-represented in individuals with developmental delay. Our studies indicate that cisd2, Ppt1 and cln3 function in concert in flies, suggesting that CISD2, PPT1 and CLN3 might also function coordinately in humans. Further, our studies raise the possibility that WFS2 and some lysosomal storage disorders might be influenced by common mechanisms and that the underlying genes might have previously unappreciated effects on developmental delay.<\/jats:p>","DOI":"10.1242\/bio.20147559","type":"journal-article","created":{"date-parts":[[2014,4,5]],"date-time":"2014-04-05T04:28:13Z","timestamp":1396672093000},"page":"342-352","source":"Crossref","is-referenced-by-count":10,"title":["Genetic studies in <i>Drosophila<\/i> and humans support a model for the concerted function of <i>CISD2<\/i>, <i>PPT1<\/i> and <i>CLN3<\/i> in disease"],"prefix":"10.1242","volume":"3","author":[{"given":"Melanie A.","family":"Jones","sequence":"first","affiliation":[{"name":"Department of Human and Molecular Genetics, Virginia Commonwealth University, Richmond, VA 23298, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sami","family":"Amr","sequence":"additional","affiliation":[{"name":"Department of Human and Molecular Genetics, Virginia Commonwealth University, Richmond, VA 23298, USA"},{"name":"Molecular Biology and Genetics Program, Virginia Commonwealth University, Richmond, VA 23298, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Aerial","family":"Ferebee","sequence":"additional","affiliation":[{"name":"Department of Human and Molecular Genetics, Virginia Commonwealth University, Richmond, VA 23298, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Phung","family":"Huynh","sequence":"additional","affiliation":[{"name":"Department of Human and Molecular Genetics, Virginia Commonwealth University, Richmond, VA 23298, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jill A.","family":"Rosenfeld","sequence":"additional","affiliation":[{"name":"Signature Genomic Laboratories, Spokane, WA 99207, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael F.","family":"Miles","sequence":"additional","affiliation":[{"name":"Department of Pharmacology and Toxicology, Virginia Commonwealth University, Richmond, VA 23298, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andrew G.","family":"Davies","sequence":"additional","affiliation":[{"name":"Department of Pharmacology and Toxicology, Virginia Commonwealth University, Richmond, VA 23298, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christopher A.","family":"Korey","sequence":"additional","affiliation":[{"name":"Department of Biology, College of Charleston, Charleston, SC 29401, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"John M.","family":"Warrick","sequence":"additional","affiliation":[{"name":"Department of Biology, University of Richmond, Richmond, VA 23173, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rita","family":"Shiang","sequence":"additional","affiliation":[{"name":"Department of Human and Molecular Genetics, Virginia Commonwealth University, Richmond, VA 23298, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sarah H.","family":"Elsea","sequence":"additional","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX 77030, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Santhosh","family":"Girirajan","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, Pennsylvania State University, University Park, PA 16802, USA"},{"name":"Department of Anthropology, Pennsylvania State University, University Park, PA 16802, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mike","family":"Grotewiel","sequence":"additional","affiliation":[{"name":"Department of Human and Molecular Genetics, Virginia Commonwealth University, Richmond, VA 23298, USA"},{"name":"Molecular Biology and Genetics Program, Virginia Commonwealth University, Richmond, VA 23298, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2014,4,4]]},"reference":[{"key":"2021111216401097600_b1","doi-asserted-by":"publisher","first-page":"3389","DOI":"10.1093\/nar\/25.17.3389","article-title":"Gapped BLAST and PSI-BLAST: a new generation of protein database search programs.","volume":"25","author":"Altschul","year":"1997","journal-title":"Nucleic Acids Res."},{"key":"2021111216401097600_b2","doi-asserted-by":"publisher","first-page":"673","DOI":"10.1086\/520961","article-title":"A homozygous mutation in a novel zinc-finger protein, ERIS, is responsible for Wolfram syndrome 2.","volume":"81","author":"Amr","year":"2007","journal-title":"Am. J. Hum. Genet."},{"key":"2021111216401097600_b3","doi-asserted-by":"publisher","first-page":"641","DOI":"10.1097\/GIM.0b013e3181ef4286","article-title":"Recurrent 200-kb deletions of 16p11.2 that include the SH2B1 gene are associated with developmental delay and obesity.","volume":"12","author":"Bachmann-Gagescu","year":"2010","journal-title":"Genet. Med."},{"key":"2021111216401097600_b4","doi-asserted-by":"publisher","first-page":"8","DOI":"10.1186\/1755-8166-1-8","article-title":"Expanding the clinical phenotype of the 3q29 microdeletion syndrome and characterization of the reciprocal microduplication.","volume":"1","author":"Ballif","year":"2008","journal-title":"Mol. Cytogenet."},{"key":"2021111216401097600_b5","doi-asserted-by":"publisher","first-page":"469","DOI":"10.1111\/j.1399-0004.2008.01094.x","article-title":"Identification of a previously unrecognized microdeletion syndrome of 16q11.2q12.2.","volume":"74","author":"Ballif","year":"2008","journal-title":"Clin. Genet."},{"key":"2021111216401097600_b6","doi-asserted-by":"publisher","first-page":"1458","DOI":"10.1016\/S0140-6736(95)92473-6","article-title":"Neurodegeneration and diabetes: UK nationwide study of Wolfram (DIDMOAD) syndrome.","volume":"346","author":"Barrett","year":"1995","journal-title":"Lancet"},{"key":"2021111216401097600_b7","doi-asserted-by":"publisher","first-page":"259","DOI":"10.1002\/ajmg.a.30621","article-title":"Use of targeted array-based CGH for the clinical diagnosis of chromosomal imbalance: is less more?","volume":"134A","author":"Bejjani","year":"2005","journal-title":"Am. J. Med. Genet."},{"key":"2021111216401097600_b8","doi-asserted-by":"publisher","first-page":"666","DOI":"10.1038\/nature08689","article-title":"Large, rare chromosomal deletions associated with severe early-onset obesity.","volume":"463","author":"Bochukova","year":"2010","journal-title":"Nature"},{"key":"2021111216401097600_b9","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1242\/dev.118.2.401","article-title":"Targeted gene expression as a means of altering cell fates and generating dominant phenotypes.","volume":"118","author":"Brand","year":"1993","journal-title":"Development"},{"key":"2021111216401097600_b10","doi-asserted-by":"publisher","first-page":"209","DOI":"10.1534\/genetics.106.067983","article-title":"Genetic modifiers of Drosophila palmitoyl-protein thioesterase 1-induced degeneration.","volume":"176","author":"Buff","year":"2007","journal-title":"Genetics"},{"key":"2021111216401097600_b11","doi-asserted-by":"publisher","first-page":"2895","DOI":"10.1093\/hmg\/11.23.2895","article-title":"Genetic modulation of polyglutamine toxicity by protein conjugation pathways in Drosophila.","volume":"11","author":"Chan","year":"2002","journal-title":"Hum. Mol. Genet."},{"key":"2021111216401097600_b12","doi-asserted-by":"publisher","first-page":"1183","DOI":"10.1101\/gad.1779509","article-title":"Cisd2 deficiency drives premature aging and causes mitochondria-mediated defects in mice.","volume":"23","author":"Chen","year":"2009","journal-title":"Genes Dev."},{"key":"2021111216401097600_b13","doi-asserted-by":"publisher","first-page":"838","DOI":"10.1038\/ng.909","article-title":"A copy number variation morbidity map of developmental delay.","volume":"43","author":"Cooper","year":"2011","journal-title":"Nat. Genet."},{"key":"2021111216401097600_b14","doi-asserted-by":"publisher","first-page":"351","DOI":"10.1038\/ng0495-351","article-title":"Lung disease in the cystic fibrosis mouse exposed to bacterial pathogens.","volume":"9","author":"Davidson","year":"1995","journal-title":"Nat. Genet."},{"key":"2021111216401097600_b15","doi-asserted-by":"publisher","first-page":"1921","DOI":"10.1056\/NEJMoa1206524","article-title":"Diagnostic exome sequencing in persons with severe intellectual disability.","volume":"367","author":"de Ligt","year":"2012","journal-title":"N. Engl. J. Med."},{"key":"2021111216401097600_b16","doi-asserted-by":"publisher","first-page":"151","DOI":"10.1038\/nature05954","article-title":"A genome-wide transgenic RNAi library for conditional gene inactivation in Drosophila.","volume":"448","author":"Dietzl","year":"2007","journal-title":"Nature"},{"key":"2021111216401097600_b17","doi-asserted-by":"publisher","first-page":"1196","DOI":"10.1038\/ejhg.2010.102","article-title":"Paternally inherited microdeletion at 15q11.2 confirms a significant role for the SNORD116 C\/D box snoRNA cluster in Prader-Willi syndrome.","volume":"18","author":"Duker","year":"2010","journal-title":"Eur. J. Hum. Genet."},{"key":"2021111216401097600_b18","doi-asserted-by":"publisher","first-page":"1357","DOI":"10.1534\/genetics.106.057463","article-title":"Metabolic disruption in Drosophila bang-sensitive seizure mutants.","volume":"173","author":"Fergestad","year":"2006","journal-title":"Genetics"},{"key":"2021111216401097600_b19","doi-asserted-by":"publisher","first-page":"39609","DOI":"10.1074\/jbc.M507426200","article-title":"WFS1 is a novel component of the unfolded protein response and maintains homeostasis of the endoplasmic reticulum in pancreatic beta-cells.","volume":"280","author":"Fonseca","year":"2005","journal-title":"J. Biol. Chem."},{"key":"2021111216401097600_b20","doi-asserted-by":"publisher","first-page":"651","DOI":"10.1016\/S0092-8674(00)81385-9","article-title":"Reiterative use of the EGF receptor triggers differentiation of all cell types in the Drosophila eye.","volume":"87","author":"Freeman","year":"1996","journal-title":"Cell"},{"key":"2021111216401097600_b21","doi-asserted-by":"publisher","first-page":"945","DOI":"10.1038\/nature05412","article-title":"The cytohesin Steppke is essential for insulin signalling in Drosophila.","volume":"444","author":"Fuss","year":"2006","journal-title":"Nature"},{"key":"2021111216401097600_b22","doi-asserted-by":"publisher","first-page":"386","DOI":"10.1016\/j.exger.2005.02.005","article-title":"Rapid iterative negative geotaxis (RING): a new method for assessing age-related locomotor decline in Drosophila.","volume":"40","author":"Gargano","year":"2005","journal-title":"Exp. Gerontol."},{"key":"2021111216401097600_b23","doi-asserted-by":"publisher","first-page":"R176","DOI":"10.1093\/hmg\/ddq366","article-title":"Phenotypic variability and genetic susceptibility to genomic disorders.","volume":"19","author":"Girirajan","year":"2010","journal-title":"Hum. Mol. Genet."},{"key":"2021111216401097600_b24","doi-asserted-by":"publisher","first-page":"203","DOI":"10.1146\/annurev-genet-102209-163544","article-title":"Human copy number variation and complex genetic disease.","volume":"45","author":"Girirajan","year":"2011","journal-title":"Annu. Rev. Genet."},{"key":"2021111216401097600_b25","doi-asserted-by":"publisher","first-page":"2379","DOI":"10.1534\/genetics.105.053306","article-title":"Palmitoyl-protein thioesterase 1 deficiency in Drosophila melanogaster causes accumulation of abnormal storage material and reduced life span.","volume":"172","author":"Hickey","year":"2006","journal-title":"Genetics"},{"key":"2021111216401097600_b26","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1093\/nar\/gkn923","article-title":"Bioinformatics enrichment tools: paths toward the comprehensive functional analysis of large gene lists.","volume":"37","author":"Huang","year":"2009","journal-title":"Nucleic Acids Res."},{"key":"2021111216401097600_b27","doi-asserted-by":"publisher","first-page":"44","DOI":"10.1038\/nprot.2008.211","article-title":"Systematic and integrative analysis of large gene lists using DAVID bioinformatics resources.","volume":"4","author":"Huang","year":"2009","journal-title":"Nat. Protoc."},{"key":"2021111216401097600_b28","doi-asserted-by":"publisher","first-page":"285","DOI":"10.1016\/j.neuron.2012.04.009","article-title":"De novo gene disruptions in children on the autistic spectrum.","volume":"74","author":"Iossifov","year":"2012","journal-title":"Neuron"},{"key":"2021111216401097600_b29","doi-asserted-by":"publisher","first-page":"532","DOI":"10.1016\/j.exger.2009.05.007","article-title":"A forward genetic screen in Drosophila implicates insulin signaling in age-related locomotor impairment.","volume":"44","author":"Jones","year":"2009","journal-title":"Exp. Gerontol."},{"key":"2021111216401097600_b30","doi-asserted-by":"publisher","first-page":"30","DOI":"10.1186\/1471-2202-4-30","article-title":"An over-expression system for characterizing Ppt1 function in Drosophila.","volume":"4","author":"Korey","year":"2003","journal-title":"BMC Neurosci."},{"key":"2021111216401097600_b31","doi-asserted-by":"publisher","first-page":"803","DOI":"10.1016\/j.freeradbiomed.2009.06.021","article-title":"Sod2 knockdown in the musculature has whole-organism consequences in Drosophila.","volume":"47","author":"Martin","year":"2009","journal-title":"Free Radic. Biol. Med."},{"key":"2021111216401097600_b32","doi-asserted-by":"publisher","first-page":"D64","DOI":"10.1093\/nar\/gks1048","article-title":"The UCSC Genome Browser database: extensions and updates 2013.","volume":"41","author":"Meyer","year":"2013","journal-title":"Nucleic Acids Res."},{"key":"2021111216401097600_b33","doi-asserted-by":"publisher","first-page":"S4","DOI":"10.1186\/gb-2008-9-s1-s4","article-title":"GeneMANIA: a real-time multiple association network integration algorithm for predicting gene function.","volume":"9 Suppl. 1","author":"Mostafavi","year":"2008","journal-title":"Genome Biol."},{"key":"2021111216401097600_b34","doi-asserted-by":"publisher","first-page":"242","DOI":"10.1038\/nature11011","article-title":"Patterns and rates of exonic de novo mutations in autism spectrum disorders.","volume":"485","author":"Neale","year":"2012","journal-title":"Nature"},{"key":"2021111216401097600_b35","doi-asserted-by":"publisher","first-page":"585","DOI":"10.1038\/ng.835","article-title":"Exome sequencing in sporadic autism spectrum disorders identifies severe de novo mutations.","volume":"43","author":"O'Roak","year":"2011","journal-title":"Nat. Genet."},{"key":"2021111216401097600_b36","doi-asserted-by":"publisher","first-page":"246","DOI":"10.1038\/nature10989","article-title":"Sporadic autism exomes reveal a highly interconnected protein network of de novo mutations.","volume":"485","author":"O'Roak","year":"2012","journal-title":"Nature"},{"key":"2021111216401097600_b37","doi-asserted-by":"publisher","first-page":"52755","DOI":"10.1074\/jbc.M310331200","article-title":"Wolframin expression induces novel ion channel activity in endoplasmic reticulum membranes and increases intracellular calcium.","volume":"278","author":"Osman","year":"2003","journal-title":"J. Biol. Chem."},{"key":"2021111216401097600_b38","doi-asserted-by":"publisher","first-page":"369","DOI":"10.1038\/ejhg.2013.141","article-title":"An unusual clinical severity of 16p11.2 deletion syndrome caused by unmasked recessive mutation of CLN3.","volume":"22","author":"Pebrel-Richard","year":"2014","journal-title":"Eur. J. Hum. Genet."},{"key":"2021111216401097600_b39","doi-asserted-by":"publisher","first-page":"146","DOI":"10.1203\/pdr.0b013e31802d8a4a","article-title":"Neuronal ceroid lipofuscinosis: a common pathway?","volume":"61","author":"Persaud-Sawin","year":"2007","journal-title":"Pediatr. Res."},{"key":"2021111216401097600_b40","doi-asserted-by":"publisher","first-page":"1674","DOI":"10.1016\/S0140-6736(12)61480-9","article-title":"Range of genetic mutations associated with severe non-syndromic sporadic intellectual disability: an exome sequencing study.","volume":"380","author":"Rauch","year":"2012","journal-title":"Lancet"},{"key":"2021111216401097600_b41","doi-asserted-by":"publisher","first-page":"W307","DOI":"10.1093\/nar\/gkr378","article-title":"g:Profiler \u2013 a web server for functional interpretation of gene lists (2011 update).","volume":"39","author":"Reimand","year":"2011","journal-title":"Nucleic Acids Res."},{"key":"2021111216401097600_b42","doi-asserted-by":"publisher","first-page":"26","DOI":"10.1007\/s11689-009-9037-4","article-title":"Speech delays and behavioral problems are the predominant features in individuals with developmental delays and 16p11.2 microdeletions and microduplications.","volume":"2","author":"Rosenfeld","year":"2010","journal-title":"J Neurodev Disord"},{"key":"2021111216401097600_b43","doi-asserted-by":"publisher","first-page":"237","DOI":"10.1038\/nature10945","article-title":"De novo mutations revealed by whole-exome sequencing are strongly associated with autism.","volume":"485","author":"Sanders","year":"2012","journal-title":"Nature"},{"key":"2021111216401097600_b44","doi-asserted-by":"publisher","first-page":"352","DOI":"10.1002\/ana.410390312","article-title":"Wolfram syndrome: hereditary diabetes mellitus with brainstem and optic atrophy.","volume":"39","author":"Scolding","year":"1996","journal-title":"Ann. Neurol."},{"key":"2021111216401097600_b45","doi-asserted-by":"publisher","first-page":"559","DOI":"10.1146\/annurev.biochem.73.011303.073954","article-title":"Palmitoylation of intracellular signaling proteins: regulation and function.","volume":"73","author":"Smotrys","year":"2004","journal-title":"Annu. Rev. Biochem."},{"key":"2021111216401097600_b46","doi-asserted-by":"publisher","first-page":"2021","DOI":"10.1093\/hmg\/7.13.2021","article-title":"Diabetes insipidus, diabetes mellitus, optic atrophy and deafness (DIDMOAD) caused by mutations in a novel gene (wolframin) coding for a predicted transmembrane protein.","volume":"7","author":"Strom","year":"1998","journal-title":"Hum. Mol. Genet."},{"key":"2021111216401097600_b47","doi-asserted-by":"publisher","first-page":"667","DOI":"10.1093\/hmg\/ddn396","article-title":"Interactions between the juvenile Batten disease gene, CLN3, and the Notch and JNK signalling pathways.","volume":"18","author":"Tuxworth","year":"2009","journal-title":"Hum. Mol. Genet."},{"key":"2021111216401097600_b48","doi-asserted-by":"publisher","first-page":"584","DOI":"10.1038\/376584a0","article-title":"Mutations in the palmitoyl protein thioesterase gene causing infantile neuronal ceroid lipofuscinosis.","volume":"376","author":"Vesa","year":"1995","journal-title":"Nature"},{"key":"2021111216401097600_b49","doi-asserted-by":"publisher","first-page":"671","DOI":"10.1038\/nature08727","article-title":"A new highly penetrant form of obesity due to deletions on chromosome 16p11.2.","volume":"463","author":"Walters","year":"2010","journal-title":"Nature"},{"key":"2021111216401097600_b50","doi-asserted-by":"publisher","first-page":"W214","DOI":"10.1093\/nar\/gkq537","article-title":"The GeneMANIA prediction server: biological network integration for gene prioritization and predicting gene function.","volume":"38","author":"Warde-Farley","year":"2010","journal-title":"Nucleic Acids Res."},{"key":"2021111216401097600_b51","doi-asserted-by":"publisher","first-page":"425","DOI":"10.1038\/70532","article-title":"Suppression of polyglutamine-mediated neurodegeneration in Drosophila by the molecular chaperone HSP70.","volume":"23","author":"Warrick","year":"1999","journal-title":"Nat. Genet."},{"key":"2021111216401097600_b52","doi-asserted-by":"publisher","first-page":"667","DOI":"10.1056\/NEJMoa075974","article-title":"Association between microdeletion and microduplication at 16p11.2 and autism.","volume":"358","author":"Weiss","year":"2008","journal-title":"N. Engl. J. Med."},{"key":"2021111216401097600_b53","doi-asserted-by":"publisher","first-page":"5318","DOI":"10.1073\/pnas.0701078104","article-title":"MitoNEET is an iron-containing outer mitochondrial membrane protein that regulates oxidative capacity.","volume":"104","author":"Wiley","year":"2007","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2021111216401097600_b54","first-page":"715","article-title":"Diabetes mellitus and simple optic atrophy among siblings: report of four cases.","volume":"13","author":"Wolfram","year":"1938","journal-title":"Mayo Clin. Proc."}],"container-title":["Biology Open"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.logists.com\/bio\/bio\/article-pdf\/3\/5\/342\/2114141\/bio-03-05-342.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.logists.com\/bio\/bio\/article-pdf\/3\/5\/342\/2114141\/bio-03-05-342.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,11,12]],"date-time":"2021-11-12T16:40:19Z","timestamp":1636735219000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/bio\/article\/3\/5\/342\/194\/Genetic-studies-in-Drosophila-and-humans-support-a"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2014,4,4]]},"references-count":54,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2014,5,15]]}},"URL":"https:\/\/doi.org\/10.1242\/bio.20147559","relation":{},"ISSN":["2046-6390"],"issn-type":[{"value":"2046-6390","type":"electronic"}],"subject":[],"published":{"date-parts":[[2014,4,4]]}}}