{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,15]],"date-time":"2026-10-15T03:54:30Z","timestamp":1792036470380,"version":"4.4.0"},"reference-count":31,"publisher":"The Company of Biologists","issue":"6","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2011,11,1]]},"abstract":"<jats:title>SUMMARY<\/jats:title>\n               <jats:p>Zebrafish (Danio rerio) can serve as a model system to study heritable skin diseases. The skin is rapidly developed during the first 5\u20136 days of embryonic growth, accompanied by expression of skin-specific genes. Transmission electron microscopy (TEM) of wild-type zebrafish at day 5 reveals a two-cell-layer epidermis separated from the underlying collagenous stroma by a basement membrane with fully developed hemidesmosomes. Scanning electron microscopy (SEM) reveals an ordered surface contour of keratinocytes with discrete microridges. To gain insight into epidermal morphogenesis, we have employed morpholino-mediated knockdown of the abca12 and snap29 genes, which are crucial for secretion of lipids and intracellular trafficking of lamellar granules, respectively. Morpholinos, when placed on exon-intron junctions, were &amp;gt;90% effective in preventing the corresponding gene expression when injected into one- to four-cell-stage embryos. By day 3, TEM of abca12 morphants showed accumulation of lipid-containing electron-dense lamellar granules, whereas snap29 morphants showed the presence of apparently empty vesicles in the epidermis. Evaluation of epidermal morphogenesis by SEM revealed similar perturbations in both cases in the microridge architecture and the development of spicule-like protrusions on the surface of keratinocytes. These morphological findings are akin to epidermal changes in harlequin ichthyosis and CEDNIK syndrome, autosomal recessive keratinization disorders due to mutations in the ABCA12 and SNAP29 genes, respectively. The results indicate that interference of independent pathways involving lipid transport in the epidermis can result in phenotypically similar perturbations in epidermal morphogenesis, and that these fish mutants can serve as a model to study the pathomechanisms of these keratinization disorders.<\/jats:p>","DOI":"10.1242\/dmm.007146","type":"journal-article","created":{"date-parts":[[2011,8,5]],"date-time":"2011-08-05T02:38:52Z","timestamp":1312511932000},"page":"777-785","source":"Crossref","is-referenced-by-count":30,"title":["Abca12-mediated lipid transport and Snap29-dependent trafficking of lamellar granules are crucial for epidermal morphogenesis in a zebrafish model of ichthyosis"],"prefix":"10.1242","volume":"4","author":[{"given":"Qiaoli","family":"Li","sequence":"first","affiliation":[{"name":"Department of Dermatology and Cutaneous Biology, and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Frank","sequence":"additional","affiliation":[{"name":"Department of Dermatology and Cutaneous Biology, and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masashi","family":"Akiyama","sequence":"additional","affiliation":[{"name":"Department of Dermatology, Nagoya University Graduate School of Medicine, Nagoya 466-8550, Japan"},{"name":"Department of Dermatology, Hokkaido University Graduate School of Medicine, Sapporo 060-8638, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroshi","family":"Shimizu","sequence":"additional","affiliation":[{"name":"Department of Dermatology, Hokkaido University Graduate School of Medicine, Sapporo 060-8638, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shiu-Ying","family":"Ho","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, Jefferson Medical College, Thomas Jefferson University, Philadelphia, PA 19107, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christine","family":"Thisse","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, University of Virginia School of Medicine, Charlottesville, VA 22908, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bernard","family":"Thisse","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, University of Virginia School of Medicine, Charlottesville, VA 22908, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eli","family":"Sprecher","sequence":"additional","affiliation":[{"name":"Department of Dermatology, Tel Aviv Medical Center, Tel Aviv 64239, Israel"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jouni","family":"Uitto","sequence":"additional","affiliation":[{"name":"Department of Dermatology and Cutaneous Biology, and"},{"name":"Department of Biochemistry and Molecular Biology, Jefferson Medical College, Thomas Jefferson University, Philadelphia, PA 19107, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2021042500512518500_b1-0040777","doi-asserted-by":"crossref","first-page":"83","DOI":"10.1016\/j.jdermsci.2006.01.003","article-title":"Harlequin ichthyosis and other autosomal recessive congenital ichthyoses: the underlying genetic defects and pathomechanisms","volume":"42","author":"Akiyama","year":"2006","journal-title":"J. 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