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The N471D and several other point mutations of human strumpellin (Str; also known as WASHC5), a member of the Wiskott\u2013Aldrich syndrome protein and SCAR homologue (WASH) complex, have been shown to cause a form of HSP known as spastic paraplegia 8 (SPG8). To investigate the molecular functions of wild-type (WT) and N417D Str, we generated Dictyostelium Str\u2212 cells and ectopically expressed StrWT-GFP or StrN471D-GFP in Str\u2212 and WT cells. Overexpression of both proteins apparently caused a defect in cell division, as we observed a clear increase in multinucleate cells. Real-time PCR analyses revealed no transcriptional changes in WASH complex subunits in Str\u2212 cells, but western blots showed a twofold decrease in the SWIP subunit. GFP-trap experiments in conjunction with mass-spectrometric analysis revealed many previously known, as well as new, Str-interacting proteins, and also proteins that no longer bind to StrN471D. At the cellular level, Str\u2212 cells displayed defects in cell growth, phagocytosis, macropinocytosis, exocytosis and lysosomal function. Expression of StrWT-GFP in Str\u2212 cells rescued all observed defects. In contrast, expression of StrN471D-GFP could not rescue lysosome morphology and exocytosis of indigestible material. Our results underscore a key role for the WASH complex and its core subunit, Str, in the endolysosomal system, and highlight the fundamental importance of the Str N471 residue for maintaining lysosome morphology and dynamics. Our data indicate that the SPG8-causing N471D mutation leads to a partial loss of Str function in the endolysosomal system.<\/jats:p>\n               <jats:p>This article has an associated First Person interview with the first author of the paper.<\/jats:p>","DOI":"10.1242\/dmm.033449","type":"journal-article","created":{"date-parts":[[2018,7,30]],"date-time":"2018-07-30T09:35:47Z","timestamp":1532943347000},"source":"Crossref","is-referenced-by-count":15,"title":["Expression of N471D strumpellin leads to defects in the endolysosomal system"],"prefix":"10.1242","volume":"11","author":[{"given":"Lin","family":"Song","sequence":"first","affiliation":[{"name":"Center for Biochemistry, Institute of Biochemistry I 1 , Medical Faculty , , 50931 Cologne , Germany"},{"name":"University of Cologne 1 , Medical Faculty , , 50931 Cologne , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ramesh","family":"Rijal","sequence":"additional","affiliation":[{"name":"Center for Biochemistry, Institute of Biochemistry I 1 , Medical Faculty , , 50931 Cologne , Germany"},{"name":"University of Cologne 1 , Medical Faculty , , 50931 Cologne , Germany"},{"name":"Texas A&M University 2 Department of Biology , , College Station , TX 3258 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Malte","family":"Karow","sequence":"additional","affiliation":[{"name":"Center for Biochemistry, Institute of Biochemistry I 1 , Medical Faculty , , 50931 Cologne , Germany"},{"name":"University of Cologne 1 , Medical Faculty , , 50931 Cologne , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria","family":"Stumpf","sequence":"additional","affiliation":[{"name":"Center for Biochemistry, Institute of Biochemistry I 1 , Medical Faculty , , 50931 Cologne , Germany"},{"name":"University of Cologne 1 , Medical Faculty , , 50931 Cologne , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Oliver","family":"Hahn","sequence":"additional","affiliation":[{"name":"Max Planck Institute for Biology of Ageing 3 , Biological Mechanisms of Ageing , 50931 Cologne , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Park","sequence":"additional","affiliation":[{"name":"CR-UK Beatson Institute, Institute of Cancer Sciences 4 , Glasgow University , Glasgow G12 8QQ , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert","family":"Insall","sequence":"additional","affiliation":[{"name":"CR-UK Beatson Institute, Institute of Cancer Sciences 4 , Glasgow University , Glasgow G12 8QQ , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rolf","family":"Schr\u00f6der","sequence":"additional","affiliation":[{"name":"Institute of Neuropathology, University Hospital Erlangen 5 , 91054 Erlangen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andreas","family":"Hofmann","sequence":"additional","affiliation":[{"name":"Eskitis Institute, Griffith University 6 Structural Chemistry Program , , N75 Don Young Road, Nathan, QLD 4111 , Australia"},{"name":"The University of Melbourne 7 Faculty of Veterinary Science , , Parkville, VIC 3030 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christoph S.","family":"Clemen","sequence":"additional","affiliation":[{"name":"Center for Biochemistry, Institute of Biochemistry I 1 , Medical Faculty , , 50931 Cologne , Germany"},{"name":"University of Cologne 1 , Medical Faculty , , 50931 Cologne , Germany"},{"name":"Heimer Institute for Muscle Research, University Hospital Bergmannsheil, Ruhr-University Bochum 8 Department of Neurology , , 44789 Bochum , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1594-6117","authenticated-orcid":false,"given":"Ludwig","family":"Eichinger","sequence":"additional","affiliation":[{"name":"Center for Biochemistry, Institute of Biochemistry I 1 , Medical Faculty , , 50931 Cologne , Germany"},{"name":"University of Cologne 1 , Medical Faculty , , 50931 Cologne , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2018,9,13]]},"reference":[{"key":"2024061303515934800_DMM033449C1","doi-asserted-by":"publisher","first-page":"73","DOI":"10.1007\/s11010-009-0111-8","article-title":"Dictyostelium discoideum--a model for many reasons","volume":"329","author":"Annesley","year":"2009","journal-title":"Mol. 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