KLEIP deficiency in mice causes progressive corneal neovascular dystrophy
The BTB-kelch protein KLEIP/KLHL20 is an actin binding protein that regulates cell-cell contact formation and cell migration. The aim of our study was to characterize KLEIP's function in ocular health and disease in mice. KLEIP(-/-) mice were generated, and corneas were examined histologically...
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| Veröffentlicht in: | Investigative ophthalmology & visual science 2012-05, Vol.53 (6), p.3260-3268 |
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| creator | Hahn, Nicole Dietz, Christian T Kühl, Sandra Vossmerbaeumer, Urs Kroll, Jens |
| description | The BTB-kelch protein KLEIP/KLHL20 is an actin binding protein that regulates cell-cell contact formation and cell migration. The aim of our study was to characterize KLEIP's function in ocular health and disease in mice.
KLEIP(-/-) mice were generated, and corneas were examined histologically and stained for keratin-1, loricrin, keratin-12, keratin-14, CD31, LYVE-1, F4/80, E-cadherin, and Ki67. Corneal abrasions were performed after eyelid opening.
Corneas of KLEIP(+/+) and KLEIP(-/-) mice were indistinguishable at birth. After eyelid opening corneal epithelial hyperplasia started to manifest in KLEIP(-/-) mice, showing a progressive epithelial metaplasia leading to total corneal opacity. In KLEIP(-/-) mice the initial stratified squamous corneal epithelium was altered to an epidermal histo-architecture showing several superficial keratinized cells, cell infiltrations into the stroma, and several apoptotic cells. Skin markers keratin 1 and loricrin were positive, and surface disease was accompanied by deep stromal vascularization. Expression analysis for E-cadherin in KLEIP(-/-) corneas showed acellular areas in the squamous epithelium, indicating a progressive fragile corneal integrity. Removal of the virgin epithelium accelerated strongly development of the epithelial and stromal alterations, identifying mechanical injuries as the major trigger for corneal dystrophy formation and scarification in KLEIP(-/-) mice.
The data identify KLEIP as an important molecule regulating corneal epithelial integrity. |
| doi_str_mv | 10.1167/iovs.12-9676 |
| format | Article |
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KLEIP(-/-) mice were generated, and corneas were examined histologically and stained for keratin-1, loricrin, keratin-12, keratin-14, CD31, LYVE-1, F4/80, E-cadherin, and Ki67. Corneal abrasions were performed after eyelid opening.
Corneas of KLEIP(+/+) and KLEIP(-/-) mice were indistinguishable at birth. After eyelid opening corneal epithelial hyperplasia started to manifest in KLEIP(-/-) mice, showing a progressive epithelial metaplasia leading to total corneal opacity. In KLEIP(-/-) mice the initial stratified squamous corneal epithelium was altered to an epidermal histo-architecture showing several superficial keratinized cells, cell infiltrations into the stroma, and several apoptotic cells. Skin markers keratin 1 and loricrin were positive, and surface disease was accompanied by deep stromal vascularization. Expression analysis for E-cadherin in KLEIP(-/-) corneas showed acellular areas in the squamous epithelium, indicating a progressive fragile corneal integrity. Removal of the virgin epithelium accelerated strongly development of the epithelial and stromal alterations, identifying mechanical injuries as the major trigger for corneal dystrophy formation and scarification in KLEIP(-/-) mice.
The data identify KLEIP as an important molecule regulating corneal epithelial integrity.</description><identifier>ISSN: 1552-5783</identifier><identifier>EISSN: 1552-5783</identifier><identifier>DOI: 10.1167/iovs.12-9676</identifier><identifier>PMID: 22511632</identifier><language>eng</language><publisher>United States</publisher><subject>Animals ; Carrier Proteins - biosynthesis ; Carrier Proteins - genetics ; Cornea - metabolism ; Cornea - pathology ; Corneal Neovascularization - complications ; Corneal Neovascularization - metabolism ; Corneal Neovascularization - pathology ; Corneal Opacity - etiology ; Corneal Opacity - genetics ; Corneal Opacity - pathology ; Disease Models, Animal ; Disease Progression ; Gene Expression Regulation ; Genotype ; Mice ; Mice, Inbred C57BL ; Mice, Transgenic ; Real-Time Polymerase Chain Reaction ; RNA, Messenger - genetics</subject><ispartof>Investigative ophthalmology & visual science, 2012-05, Vol.53 (6), p.3260-3268</ispartof><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c291t-429142637e21538bb6624eeb56bc39e11e7f0d1b26d405e5abc8e17d14ee26063</citedby></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27922,27923</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/22511632$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Hahn, Nicole</creatorcontrib><creatorcontrib>Dietz, Christian T</creatorcontrib><creatorcontrib>Kühl, Sandra</creatorcontrib><creatorcontrib>Vossmerbaeumer, Urs</creatorcontrib><creatorcontrib>Kroll, Jens</creatorcontrib><title>KLEIP deficiency in mice causes progressive corneal neovascular dystrophy</title><title>Investigative ophthalmology & visual science</title><addtitle>Invest Ophthalmol Vis Sci</addtitle><description>The BTB-kelch protein KLEIP/KLHL20 is an actin binding protein that regulates cell-cell contact formation and cell migration. The aim of our study was to characterize KLEIP's function in ocular health and disease in mice.
KLEIP(-/-) mice were generated, and corneas were examined histologically and stained for keratin-1, loricrin, keratin-12, keratin-14, CD31, LYVE-1, F4/80, E-cadherin, and Ki67. Corneal abrasions were performed after eyelid opening.
Corneas of KLEIP(+/+) and KLEIP(-/-) mice were indistinguishable at birth. After eyelid opening corneal epithelial hyperplasia started to manifest in KLEIP(-/-) mice, showing a progressive epithelial metaplasia leading to total corneal opacity. In KLEIP(-/-) mice the initial stratified squamous corneal epithelium was altered to an epidermal histo-architecture showing several superficial keratinized cells, cell infiltrations into the stroma, and several apoptotic cells. Skin markers keratin 1 and loricrin were positive, and surface disease was accompanied by deep stromal vascularization. Expression analysis for E-cadherin in KLEIP(-/-) corneas showed acellular areas in the squamous epithelium, indicating a progressive fragile corneal integrity. Removal of the virgin epithelium accelerated strongly development of the epithelial and stromal alterations, identifying mechanical injuries as the major trigger for corneal dystrophy formation and scarification in KLEIP(-/-) mice.
The data identify KLEIP as an important molecule regulating corneal epithelial integrity.</description><subject>Animals</subject><subject>Carrier Proteins - biosynthesis</subject><subject>Carrier Proteins - genetics</subject><subject>Cornea - metabolism</subject><subject>Cornea - pathology</subject><subject>Corneal Neovascularization - complications</subject><subject>Corneal Neovascularization - metabolism</subject><subject>Corneal Neovascularization - pathology</subject><subject>Corneal Opacity - etiology</subject><subject>Corneal Opacity - genetics</subject><subject>Corneal Opacity - pathology</subject><subject>Disease Models, Animal</subject><subject>Disease Progression</subject><subject>Gene Expression Regulation</subject><subject>Genotype</subject><subject>Mice</subject><subject>Mice, Inbred C57BL</subject><subject>Mice, Transgenic</subject><subject>Real-Time Polymerase Chain Reaction</subject><subject>RNA, Messenger - genetics</subject><issn>1552-5783</issn><issn>1552-5783</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2012</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpNkE1PwzAMhiMEYmNw44x65EBHnDRpd0TTBhOT4ADnKk1dCOrHiNdJ_fdk2kBcbMt6bL16GLsGPgXQ6b3rdjQFEc90qk_YGJQSsUozefpvHrELoi_OBYDg52wkhAq3UozZ6nm9WL1GJVbOOmztELk2apzFyJqekKKN7z48ErldWHW-RVNHLXY7Q7avjY_Kgba-23wOl-ysMjXh1bFP2Pty8TZ_itcvj6v5wzq2YgbbOAk1EVqmKEDJrCi0FglioXRh5QwBMK14CYXQZcIVKlPYDCEtIUBCcy0n7PbwNyT77pG2eePIYl2bEKunHDhkWmous4DeHVDrOyKPVb7xrjF-CFC-l5fv5eUg8r28gN8cP_dFg-Uf_GtL_gAf1Gqz</recordid><startdate>20120531</startdate><enddate>20120531</enddate><creator>Hahn, Nicole</creator><creator>Dietz, Christian T</creator><creator>Kühl, Sandra</creator><creator>Vossmerbaeumer, Urs</creator><creator>Kroll, Jens</creator><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope></search><sort><creationdate>20120531</creationdate><title>KLEIP deficiency in mice causes progressive corneal neovascular dystrophy</title><author>Hahn, Nicole ; Dietz, Christian T ; Kühl, Sandra ; Vossmerbaeumer, Urs ; Kroll, Jens</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c291t-429142637e21538bb6624eeb56bc39e11e7f0d1b26d405e5abc8e17d14ee26063</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2012</creationdate><topic>Animals</topic><topic>Carrier Proteins - biosynthesis</topic><topic>Carrier Proteins - genetics</topic><topic>Cornea - metabolism</topic><topic>Cornea - pathology</topic><topic>Corneal Neovascularization - complications</topic><topic>Corneal Neovascularization - metabolism</topic><topic>Corneal Neovascularization - pathology</topic><topic>Corneal Opacity - etiology</topic><topic>Corneal Opacity - genetics</topic><topic>Corneal Opacity - pathology</topic><topic>Disease Models, Animal</topic><topic>Disease Progression</topic><topic>Gene Expression Regulation</topic><topic>Genotype</topic><topic>Mice</topic><topic>Mice, Inbred C57BL</topic><topic>Mice, Transgenic</topic><topic>Real-Time Polymerase Chain Reaction</topic><topic>RNA, Messenger - genetics</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Hahn, Nicole</creatorcontrib><creatorcontrib>Dietz, Christian T</creatorcontrib><creatorcontrib>Kühl, Sandra</creatorcontrib><creatorcontrib>Vossmerbaeumer, Urs</creatorcontrib><creatorcontrib>Kroll, Jens</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Investigative ophthalmology & visual science</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Hahn, Nicole</au><au>Dietz, Christian T</au><au>Kühl, Sandra</au><au>Vossmerbaeumer, Urs</au><au>Kroll, Jens</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>KLEIP deficiency in mice causes progressive corneal neovascular dystrophy</atitle><jtitle>Investigative ophthalmology & visual science</jtitle><addtitle>Invest Ophthalmol Vis Sci</addtitle><date>2012-05-31</date><risdate>2012</risdate><volume>53</volume><issue>6</issue><spage>3260</spage><epage>3268</epage><pages>3260-3268</pages><issn>1552-5783</issn><eissn>1552-5783</eissn><abstract>The BTB-kelch protein KLEIP/KLHL20 is an actin binding protein that regulates cell-cell contact formation and cell migration. The aim of our study was to characterize KLEIP's function in ocular health and disease in mice.
KLEIP(-/-) mice were generated, and corneas were examined histologically and stained for keratin-1, loricrin, keratin-12, keratin-14, CD31, LYVE-1, F4/80, E-cadherin, and Ki67. Corneal abrasions were performed after eyelid opening.
Corneas of KLEIP(+/+) and KLEIP(-/-) mice were indistinguishable at birth. After eyelid opening corneal epithelial hyperplasia started to manifest in KLEIP(-/-) mice, showing a progressive epithelial metaplasia leading to total corneal opacity. In KLEIP(-/-) mice the initial stratified squamous corneal epithelium was altered to an epidermal histo-architecture showing several superficial keratinized cells, cell infiltrations into the stroma, and several apoptotic cells. Skin markers keratin 1 and loricrin were positive, and surface disease was accompanied by deep stromal vascularization. Expression analysis for E-cadherin in KLEIP(-/-) corneas showed acellular areas in the squamous epithelium, indicating a progressive fragile corneal integrity. Removal of the virgin epithelium accelerated strongly development of the epithelial and stromal alterations, identifying mechanical injuries as the major trigger for corneal dystrophy formation and scarification in KLEIP(-/-) mice.
The data identify KLEIP as an important molecule regulating corneal epithelial integrity.</abstract><cop>United States</cop><pmid>22511632</pmid><doi>10.1167/iovs.12-9676</doi><tpages>9</tpages></addata></record> |
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| subjects | Animals Carrier Proteins - biosynthesis Carrier Proteins - genetics Cornea - metabolism Cornea - pathology Corneal Neovascularization - complications Corneal Neovascularization - metabolism Corneal Neovascularization - pathology Corneal Opacity - etiology Corneal Opacity - genetics Corneal Opacity - pathology Disease Models, Animal Disease Progression Gene Expression Regulation Genotype Mice Mice, Inbred C57BL Mice, Transgenic Real-Time Polymerase Chain Reaction RNA, Messenger - genetics |
| title | KLEIP deficiency in mice causes progressive corneal neovascular dystrophy |
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