{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T15:02:02Z","timestamp":1791471722791,"version":"4.3.4"},"reference-count":28,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2018,10,11]],"date-time":"2018-10-11T00:00:00Z","timestamp":1539216000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001665","name":"Agence Nationale de la Recherche","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001665","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100009578","name":"Mizutani Foundation for Glycoscience","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100009578","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["faseb.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The FASEB Journal"],"published-print":{"date-parts":[[2019,2]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Congenital disorders of glycosylation are severe inherited diseases in which aberrant protein glycosylation is a hallmark. Transmembrane protein 165 (TMEM165) is a novel Golgi transmembrane protein involved in type II congenital disorders of glycosylation. Although its biologic function is still a controversial issue, we have demonstrated that the Golgi glycosylation defect due to TMEM165 deficiency resulted from a Golgi Mn\n                    <jats:sup>2+<\/jats:sup>\n                    homeostasis defect. The goal of this study was to delineate the cellular pathway by which extracellular Mn\n                    <jats:sup>2+<\/jats:sup>\n                    rescues\n                    <jats:italic>N<\/jats:italic>\n                    \u2010glycosylation in TMEM165 knockout (KO) cells. We first demonstrated that after extracellular exposure, Mn\n                    <jats:sup>2+<\/jats:sup>\n                    uptake by HEK293 cells at the plasma membrane did not rely on endocytosis but was likely done by plasma membrane transporters. Second, we showed that the secretory pathway Ca\n                    <jats:sup>2+<\/jats:sup>\n                    \u2010ATPase 1, also known to mediate the influx of cytosolic Mn\n                    <jats:sup>2+<\/jats:sup>\n                    into the lumen of the Golgi apparatus, is not crucial for the Mn\n                    <jats:sup>2+<\/jats:sup>\n                    \u2010induced rescue glycosylation of lysosomal\u2010associated membrane protein 2 (LAMP2). In contrast, our results demonstrate the involvement of cyclopiazonic acid\u2014and thapsigargin (Tg)\u2010sensitive pumps in the rescue of TMEM165\u2010associated glycosylation defects by Mn\n                    <jats:sup>2+<\/jats:sup>\n                    . Interestingly, overexpression of sarco\/endoplasmic reticulum Ca\n                    <jats:sup>2+<\/jats:sup>\n                    \u2010ATPase (SERCA) 2b isoform in TMEM165 KO cells partially rescues the observed LAMP2 glycosylation defect. Overall, this study indicates that the rescue of Golgi\n                    <jats:italic>N<\/jats:italic>\n                    \u2010glycosylation defects in TMEM165 KO cells by extracellular Mn\n                    <jats:sup>2+<\/jats:sup>\n                    involves the activity of Tg and cyclopiazonic acid\u2013sensitive pumps, probably the SERCA pumps.\u2014Houdou, M., Lebredonchel, E., Garat, A., Duvet, S., Legrand, D., Decool, V., Klein, A., Ouzzine, M., Gasnier, B., Potelle, S., Foulquier, F. Involvement of thapsigargin\u2014and cyclopiazonic acid\u2013sensitive pumps in the rescue of TMEM165\u2010associated glycosylation defects by Mn\n                    <jats:sup>2+<\/jats:sup>\n                    . FASEB J. 33, 2669\u20132679 (2019).\n                    <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" xlink:href=\"http:\/\/www.fasebj.org\">www.fasebj.org<\/jats:ext-link>\n                  <\/jats:p>","DOI":"10.1096\/fj.201800387r","type":"journal-article","created":{"date-parts":[[2018,10,11]],"date-time":"2018-10-11T14:02:04Z","timestamp":1539266524000},"page":"2669-2679","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":41,"title":["Involvement of thapsigargin\u2013 and cyclopiazonic acid\u2013sensitive pumps in the rescue of TMEM165\u2010associated glycosylation defects by Mn\n                    <sup>2+<\/sup>"],"prefix":"10.1096","volume":"33","author":[{"given":"Marine","family":"Houdou","sequence":"first","affiliation":[{"name":"Centre National de la Recherche Scientifique (CNRS) Unit\u00e9 Mixte de Recherche (UMR) 8576 Unit\u00e9 de Glycobiologie Structurale et Fonctionnelle (UGSF) University of Lille Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elodie","family":"Lebredonchel","sequence":"additional","affiliation":[{"name":"Centre National de la Recherche Scientifique (CNRS) Unit\u00e9 Mixte de Recherche (UMR) 8576 Unit\u00e9 de Glycobiologie Structurale et Fonctionnelle (UGSF) University of Lille Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne","family":"Garat","sequence":"additional","affiliation":[{"name":"Centre Hospitalier Universitaire (CHU) Lille Institut Pasteur de Lille, Equipe d'Accueil (EA) 4483 Impact de l'Environnement Chimique sur la Sant\u00e9 Humaine (IMPECS) University of Lille Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sandrine","family":"Duvet","sequence":"additional","affiliation":[{"name":"Centre National de la Recherche Scientifique (CNRS) Unit\u00e9 Mixte de Recherche (UMR) 8576 Unit\u00e9 de Glycobiologie Structurale et Fonctionnelle (UGSF) University of Lille Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dominique","family":"Legrand","sequence":"additional","affiliation":[{"name":"Centre National de la Recherche Scientifique (CNRS) Unit\u00e9 Mixte de Recherche (UMR) 8576 Unit\u00e9 de Glycobiologie Structurale et Fonctionnelle (UGSF) University of Lille Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Val\u00e9rie","family":"Decool","sequence":"additional","affiliation":[{"name":"Centre Hospitalier Universitaire (CHU) Lille Institut Pasteur de Lille, Equipe d'Accueil (EA) 4483 Impact de l'Environnement Chimique sur la Sant\u00e9 Humaine (IMPECS) University of Lille Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andr\u00e9","family":"Klein","sequence":"additional","affiliation":[{"name":"Centre National de la Recherche Scientifique (CNRS) Unit\u00e9 Mixte de Recherche (UMR) 8576 Unit\u00e9 de Glycobiologie Structurale et Fonctionnelle (UGSF) University of Lille Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mohamed","family":"Ouzzine","sequence":"additional","affiliation":[{"name":"UMR 7365 Centre National de la Recherche Scientifique (CNRS) Universit\u00e9 de Lorraine Biop\u00f4le-Facult\u00e9 de M\u00e9decine Vandoeuvre-l\u00e8s-Nancy France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bruno","family":"Gasnier","sequence":"additional","affiliation":[{"name":"Neurophotonics Laboratory CNRS UMR 8250 Paris Descartes University Sorbonne Paris Cit\u00e9 Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sven","family":"Potelle","sequence":"additional","affiliation":[{"name":"Centre National de la Recherche Scientifique (CNRS) Unit\u00e9 Mixte de Recherche (UMR) 8576 Unit\u00e9 de Glycobiologie Structurale et Fonctionnelle (UGSF) University of Lille Lille 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