000169351 001__ 169351 000169351 005__ 20240610143745.0 000169351 0247_ $$2doi$$a10.1093/hmg/ddac307 000169351 0247_ $$2pmid$$apmid:36611008 000169351 0247_ $$2ISSN$$a0964-6906 000169351 0247_ $$2ISSN$$a1460-2083 000169351 0247_ $$2altmetric$$aaltmetric:140977788 000169351 037__ $$aDZNE-2023-00126 000169351 041__ $$aEnglish 000169351 082__ $$a570 000169351 1001_ $$aNitta, Yohei$$b0 000169351 245__ $$aDirect evaluation of neuroaxonal degeneration with the causative genes of neurodegenerative diseases in drosophila using the automated axon quantification system, MeDUsA. 000169351 260__ $$aOxford$$bOxford Univ. Press$$c2023 000169351 3367_ $$2DRIVER$$aarticle 000169351 3367_ $$2DataCite$$aOutput Types/Journal article 000169351 3367_ $$0PUB:(DE-HGF)16$$2PUB:(DE-HGF)$$aJournal Article$$bjournal$$mjournal$$s1718023048_5323 000169351 3367_ $$2BibTeX$$aARTICLE 000169351 3367_ $$2ORCID$$aJOURNAL_ARTICLE 000169351 3367_ $$00$$2EndNote$$aJournal Article 000169351 520__ $$aDrosophila is an excellent model organism for studying human neurodegenerative diseases (NDs). However, there is still almost no experimental system that could directly observe the degeneration of neurons and automatically quantify axonal degeneration. In this study, we created MeDUsA (a 'method for the quantification of degeneration using fly axons'), a standalone executable computer program based on Python that combines a pre-trained deep-learning masking tool with an axon terminal counting tool. This software automatically quantifies the number of retinal R7 axons in Drosophila from a confocal z-stack image series. Using this software, we were able to directly demonstrate that axons were degenerated by the representative causative genes of NDs for the first time in Drosophila. The fly retinal axon is an excellent experimental system that is capable of mimicking the pathology of axonal degeneration in human NDs. MeDUsA rapidly and accurately quantifies axons in Drosophila photoreceptor neurons. It enables large-scale research into axonal degeneration, including screening to identify genes or drugs that mediate axonal toxicity caused by ND proteins and diagnose the pathological significance of novel variants of human genes in axons. 000169351 536__ $$0G:(DE-HGF)POF4-351$$a351 - Brain Function (POF4-351)$$cPOF4-351$$fPOF IV$$x0 000169351 588__ $$aDataset connected to CrossRef, PubMed, , Journals: pub.dzne.de 000169351 650_7 $$2NLM Chemicals$$aDrosophila Proteins 000169351 650_2 $$2MeSH$$aAnimals 000169351 650_2 $$2MeSH$$aHumans 000169351 650_2 $$2MeSH$$aDrosophila: genetics 000169351 650_2 $$2MeSH$$aDrosophila: metabolism 000169351 650_2 $$2MeSH$$aNeurodegenerative Diseases: metabolism 000169351 650_2 $$2MeSH$$aAxons: metabolism 000169351 650_2 $$2MeSH$$aNeurons: metabolism 000169351 650_2 $$2MeSH$$aDrosophila Proteins: genetics 000169351 650_2 $$2MeSH$$aDrosophila Proteins: metabolism 000169351 7001_ $$aKawai, Hiroki$$b1 000169351 7001_ $$aMaki, Ryuto$$b2 000169351 7001_ $$aOsaka, Jiro$$b3 000169351 7001_ $$aHakeda-Suzuki, Satoko$$b4 000169351 7001_ $$aNagai, Yoshitaka$$b5 000169351 7001_ $$0P:(DE-2719)9000534$$aDoubková, Karolína$$b6$$udzne 000169351 7001_ $$aUehara, Tomoko$$b7 000169351 7001_ $$aWatanabe, Kenji$$b8 000169351 7001_ $$aKosaki, Kenjiro$$b9 000169351 7001_ $$aSuzuki, Takashi$$b10 000169351 7001_ $$0P:(DE-2719)2810271$$aTavosanis, Gaia$$b11$$udzne 000169351 7001_ $$0P:(DE-2719)2810439$$aSugie, Atsushi$$b12$$udzne 000169351 773__ $$0PERI:(DE-600)1474816-2$$a10.1093/hmg/ddac307$$gp. ddac307$$n9$$p1524-1538$$tHuman molecular genetics$$v32$$x0964-6906$$y2023 000169351 8564_ $$uhttps://pub.dzne.de/record/169351/files/DZNE-2023-00126_Restricted.pdf 000169351 8564_ $$uhttps://pub.dzne.de/record/169351/files/DZNE-2023-00126_Restricted.pdf?subformat=pdfa$$xpdfa 000169351 909CO $$ooai:pub.dzne.de:169351$$pVDB 000169351 9101_ $$0I:(DE-588)1065079516$$6P:(DE-2719)9000534$$aDeutsches Zentrum für Neurodegenerative Erkrankungen$$b6$$kDZNE 000169351 9101_ $$0I:(DE-588)1065079516$$6P:(DE-2719)2810271$$aDeutsches Zentrum für Neurodegenerative Erkrankungen$$b11$$kDZNE 000169351 9101_ $$0I:(DE-HGF)0$$6P:(DE-2719)2810439$$aExternal Institute$$b12$$kExtern 000169351 9131_ $$0G:(DE-HGF)POF4-351$$1G:(DE-HGF)POF4-350$$2G:(DE-HGF)POF4-300$$3G:(DE-HGF)POF4$$4G:(DE-HGF)POF$$aDE-HGF$$bGesundheit$$lNeurodegenerative Diseases$$vBrain Function$$x0 000169351 9141_ $$y2023 000169351 915__ $$0StatID:(DE-HGF)1190$$2StatID$$aDBCoverage$$bBiological Abstracts$$d2022-11-18 000169351 915__ $$0StatID:(DE-HGF)0113$$2StatID$$aWoS$$bScience Citation Index Expanded$$d2022-11-18 000169351 915__ $$0StatID:(DE-HGF)0160$$2StatID$$aDBCoverage$$bEssential Science Indicators$$d2022-11-18 000169351 915__ $$0StatID:(DE-HGF)0420$$2StatID$$aNationallizenz$$d2023-10-21$$wger 000169351 915__ $$0StatID:(DE-HGF)0100$$2StatID$$aJCR$$bHUM MOL GENET : 2022$$d2023-10-21 000169351 915__ $$0StatID:(DE-HGF)0200$$2StatID$$aDBCoverage$$bSCOPUS$$d2023-10-21 000169351 915__ $$0StatID:(DE-HGF)0300$$2StatID$$aDBCoverage$$bMedline$$d2023-10-21 000169351 915__ $$0StatID:(DE-HGF)0320$$2StatID$$aDBCoverage$$bPubMed Central$$d2023-10-21 000169351 915__ $$0StatID:(DE-HGF)0199$$2StatID$$aDBCoverage$$bClarivate Analytics Master Journal List$$d2023-10-21 000169351 915__ $$0StatID:(DE-HGF)1050$$2StatID$$aDBCoverage$$bBIOSIS Previews$$d2023-10-21 000169351 915__ $$0StatID:(DE-HGF)0150$$2StatID$$aDBCoverage$$bWeb of Science Core Collection$$d2023-10-21 000169351 915__ $$0StatID:(DE-HGF)1030$$2StatID$$aDBCoverage$$bCurrent Contents - Life Sciences$$d2023-10-21 000169351 915__ $$0StatID:(DE-HGF)9900$$2StatID$$aIF < 5$$d2023-10-21 000169351 9201_ $$0I:(DE-2719)1013018$$kAG Tavosanis$$lDynamics of neuronal circuits$$x0 000169351 980__ $$ajournal 000169351 980__ $$aVDB 000169351 980__ $$aI:(DE-2719)1013018 000169351 980__ $$aUNRESTRICTED