{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T23:55:47Z","timestamp":1790639747268,"version":"4.1.0"},"reference-count":78,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2017,5,17]],"date-time":"2017-05-17T00:00:00Z","timestamp":1494979200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100002428","name":"Austrian Science Fund","doi-asserted-by":"publisher","award":["SFB021"],"award-info":[{"award-number":["SFB021"]}],"id":[{"id":"10.13039\/501100002428","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Traffic"],"published-print":{"date-parts":[[2017,7]]},"abstract":"<jats:p>\n                    Microvillus inclusion disease (\n                    <jats:styled-content style=\"fixed-case\">MVID<\/jats:styled-content>\n                    ) is a congenital enteropathy characterized by accumulation of vesiculo\u2010tubular endomembranes in the subapical cytoplasm of enterocytes, historically termed \u201csecretory granules.\u201d However, neither their identity nor pathophysiological significance is well defined. Using immunoelectron microscopy and tomography, we studied biopsies from\n                    <jats:styled-content style=\"fixed-case\">MVID<\/jats:styled-content>\n                    patients (3\u00d7 Myosin 5b mutations and 1\u00d7 Syntaxin3 mutation) and compared them to controls and genome\u2010edited\n                    <jats:styled-content style=\"fixed-case\">CaCo2<\/jats:styled-content>\n                    cell models, harboring relevant mutations. Duodenal biopsies from 2 patients with novel Myosin 5b mutations and typical clinical symptoms showed unusual ultrastructural phenotypes: aberrant subapical vesicles and tubules were prominent in the enterocytes, though other histological hallmarks of\n                    <jats:styled-content style=\"fixed-case\">MVID<\/jats:styled-content>\n                    were almost absent (ectopic intra\u2010\/intercellular microvilli, brush border atrophy). We identified these enigmatic vesiculo\u2010tubular organelles as Rab11\u2010Rab8\u2010positive recycling compartments of altered size, shape and location harboring the apical\n                    <jats:styled-content style=\"fixed-case\">SNARE<\/jats:styled-content>\n                    Syntaxin3, apical transporters sodium\u2010hydrogen exchanger 3 (\n                    <jats:styled-content style=\"fixed-case\">NHE3<\/jats:styled-content>\n                    ) and cystic fibrosis transmembrane conductance regulator. Our data strongly indicate that in\n                    <jats:styled-content style=\"fixed-case\">MVID<\/jats:styled-content>\n                    disrupted trafficking between cargo vesicles and the apical plasma membrane is the primary cause of a defect of epithelial polarity and subsequent facultative loss of brush border integrity, leading to malabsorption. Furthermore, they support the notion that mislocalization of transporters, such as\n                    <jats:styled-content style=\"fixed-case\">NHE3<\/jats:styled-content>\n                    substantially contributes to the reported sodium loss diarrhea.\n                  <\/jats:p>\n                  <jats:p>\n                    <jats:inline-graphic xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" xlink:href=\"graphic\/tra12486-gra-0001.png\" xlink:title=\"image\"\/>\n                  <\/jats:p>","DOI":"10.1111\/tra.12486","type":"journal-article","created":{"date-parts":[[2017,4,13]],"date-time":"2017-04-13T13:14:21Z","timestamp":1492089261000},"page":"453-464","source":"Crossref","is-referenced-by-count":54,"title":["Abnormal Rab11\u2010Rab8\u2010vesicles cluster in enterocytes of patients with microvillus inclusion disease"],"prefix":"10.1111","volume":"18","author":[{"given":"Georg F.","family":"Vogel","sequence":"first","affiliation":[{"name":"Division of Histology and 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