{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T14:58:22Z","timestamp":1790953102178,"version":"4.1.0"},"reference-count":61,"publisher":"eLife Sciences Publications, Ltd","license":[{"start":{"date-parts":[[2021,11,1]],"date-time":"2021-11-01T00:00:00Z","timestamp":1635724800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2021,11,1]],"date-time":"2021-11-01T00:00:00Z","timestamp":1635724800000},"content-version":"am","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2021,11,1]],"date-time":"2021-11-01T00:00:00Z","timestamp":1635724800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100008731","name":"Association Europ\u00e9enne contre les Leucodystrophies","doi-asserted-by":"publisher","award":["ELA2012-014C2B"],"award-info":[{"award-number":["ELA2012-014C2B"]}],"id":[{"id":"10.13039\/501100008731","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100014865","name":"Fondation Maladies Rares","doi-asserted-by":"publisher","award":["20170603"],"award-info":[{"award-number":["20170603"]}],"id":[{"id":"10.13039\/100014865","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002915","name":"Fondation pour la Recherche M\u00e9dicale","doi-asserted-by":"publisher","award":["PLP20170939025p60"],"award-info":[{"award-number":["PLP20170939025p60"]}],"id":[{"id":"10.13039\/501100002915","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002915","name":"Fondation pour la Recherche M\u00e9dicale","doi-asserted-by":"publisher","award":["AJE20171039094"],"award-info":[{"award-number":["AJE20171039094"]}],"id":[{"id":"10.13039\/501100002915","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["elifesciences.org"],"crossmark-restriction":false},"short-container-title":[],"accepted":{"date-parts":[[2021,10,27]]},"abstract":"<jats:p>\n                    Absence of the astrocyte-specific membrane protein MLC1 is responsible for megalencephalic leukoencephalopathy with subcortical cysts (MLC), a rare type of leukodystrophy characterized by early-onset macrocephaly and progressive white matter vacuolation that lead to ataxia, spasticity, and cognitive decline. During postnatal development (from P5 to P15 in the mouse), MLC1 forms a membrane complex with GlialCAM (another astrocytic transmembrane protein) at the junctions between perivascular astrocytic processes. Perivascular astrocytic processes along with blood vessels form the gliovascular unit. It was not previously known how MLC1 influences the physiology of the gliovascular unit. Here, using the\n                    <jats:italic>Mlc1<\/jats:italic>\n                    knock-out mouse model of MLC, we demonstrated that MLC1 controls the postnatal development and organization of perivascular astrocytic processes, vascular smooth muscle cell contractility, neurovascular coupling, and intraparenchymal interstitial fluid clearance. Our data suggest that MLC is a developmental disorder of the gliovascular unit, and perivascular astrocytic processes and vascular smooth muscle cell maturation defects are primary events in the pathogenesis of MLC and therapeutic targets for this disease.\n                  <\/jats:p>","DOI":"10.7554\/elife.71379","type":"journal-article","created":{"date-parts":[[2021,11,1]],"date-time":"2021-11-01T07:15:12Z","timestamp":1635750912000},"update-policy":"https:\/\/doi.org\/10.7554\/elife.71379","source":"Crossref","is-referenced-by-count":43,"title":["Megalencephalic leukoencephalopathy with subcortical cysts is a developmental disorder of the gliovascular unit"],"prefix":"10.7554","volume":"10","author":[{"given":"Alice","family":"Gilbert","sequence":"first","affiliation":[{"name":"Physiology and Physiopathology of the Gliovascular Unit Research Group, Center for Interdisciplinary Research in Biology (CIRB), College de France, CNRS Research in Biology (CIRB), College de France, CNRS","place":["Paris, France"]},{"name":"\u00c9cole doctorale Cerveau Cognition Comportement \u201cED3C\u201d N\u00b0158, Pierre and Marie Curie University","place":["Paris, France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xabier","family":"Elorza-Vidal","sequence":"additional","affiliation":[{"name":"Physiology and Physiopathology of the Gliovascular Unit Research Group, Center for Interdisciplinary Research in Biology (CIRB), College de France, CNRS Research in Biology (CIRB), College de France, CNRS","place":["Paris, France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1085-5929","authenticated-orcid":true,"given":"Armelle","family":"Rancillac","sequence":"additional","affiliation":[{"name":"Neuroglial Interactions in Cerebral Physiopathology Research Group, Center for Interdisciplinary Research in Biology (CIRB), College de France, Labex Memolife, Universit\u00e9 PSL","place":["Paris, 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