{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T07:39:51Z","timestamp":1791185991394,"version":"4.1.0"},"reference-count":28,"publisher":"National Academy of Sciences","issue":"7","content-domain":{"domain":["www.pnas.org"],"crossmark-restriction":true},"short-container-title":["Proc. Natl. Acad. Sci. U.S.A."],"published-print":{"date-parts":[[2002,4,2]]},"abstract":"<jats:p>\n                    Mammalian sulfoglycolipids comprise two major members, sulfatide (HSO\n                    <jats:sub>3<\/jats:sub>\n                    -3-galactosylceramide) and seminolipid (HSO\n                    <jats:sub>3<\/jats:sub>\n                    -3-monogalactosylalkylacylglycerol). Sulfatide is a major lipid component of the myelin sheath and serves as the epitope for the well known oligodendrocyte-marker antibody O4. Seminolipid is synthesized in spermatocytes and maintained in the subsequent germ cell stages. Both sulfoglycolipids can be synthesized\n                    <jats:italic>in vitro<\/jats:italic>\n                    by using the isolated cerebroside sulfotransferase. To investigate the physiological role of sulfoglycolipids and to determine whether sulfatide and seminolipid are biosynthesized\n                    <jats:italic>in vivo<\/jats:italic>\n                    by a single sulfotransferase,\n                    <jats:italic>Cst<\/jats:italic>\n                    -null mice were generated by gene targeting.\n                    <jats:italic>Cst<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice lacked sulfatide in brain and seminolipid in testis, proving that a single gene copy is responsible for their biosynthesis.\n                    <jats:italic>Cst<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice were born healthy, but began to display hindlimb weakness by 6 weeks of age and subsequently showed a pronounced tremor and progressive ataxia. Although compact myelin was preserved,\n                    <jats:italic>Cst<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice displayed abnormalities in paranodal junctions. On the other hand,\n                    <jats:italic>Cst<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    males were sterile because of a block in spermatogenesis before the first meiotic division, whereas females were able to breed. These data show a critical role for sulfoglycolipids in myelin function and spermatogenesis.\n                  <\/jats:p>","DOI":"10.1073\/pnas.032068299","type":"journal-article","created":{"date-parts":[[2002,7,26]],"date-time":"2002-07-26T10:37:36Z","timestamp":1027679856000},"page":"4227-4232","update-policy":"https:\/\/doi.org\/10.1073\/pnas.cm10313","source":"Crossref","is-referenced-by-count":300,"title":["Paranodal junction formation and spermatogenesis require sulfoglycolipids"],"prefix":"10.1073","volume":"99","author":[{"given":"Koichi","family":"Honke","sequence":"first","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yukie","family":"Hirahara","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey","family":"Dupree","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kinuko","family":"Suzuki","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brian","family":"Popko","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kikuro","family":"Fukushima","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Junko","family":"Fukushima","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takashi","family":"Nagasawa","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nobuaki","family":"Yoshida","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoshinao","family":"Wada","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; Research Institute, Osaka Medical Center for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka 594-1101, Japan; Department of Pathology and Laboratory Medicine and Neuroscience Center, University of North Carolina, Chapel Hill, NC 27599; Department of Physiology, Hokkaido University School of Medicine, Sapporo 060-8638, Japan; Department of Physical Therapy, College of Medical Technology,..."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naoyuki","family":"Taniguchi","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, Osaka University Medical School, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan; 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