Clinical features of incontinentia pigmenti with emphasis on oral and dental abnormalities
One of interesting aspects in dermatology is the fact that skin may reflect the presence of anomalies in other organs and tissues. One such example is incontinentia pigmenti (IP), a rare, complex, X-linked genodermatosis. Clinical manifestations of IP according to evolution and prognosis can be cons...
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Veröffentlicht in: | Clinical oral investigations 2006-12, Vol.10 (4), p.343-347 |
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description | One of interesting aspects in dermatology is the fact that skin may reflect the presence of anomalies in other organs and tissues. One such example is incontinentia pigmenti (IP), a rare, complex, X-linked genodermatosis. Clinical manifestations of IP according to evolution and prognosis can be considered as skin, as well as dental, eye, and central nervous system, changes. We have investigated a total of nine families with 25 subjects, 23 females and 2 males. In 12 female and 2 male subjects, all of them with clinical characteristics of IP, we observed the following abnormalities: teeth-shape anomalies (coni- or peg-like teeth), the presence of numerous cariotic teeth, early dental loss, delayed eruption, partial anodontia, and gothic palate. To our knowledge, this is the first time that the presence of gothic palate in patients with IP has been documented. As we found out, in two female subjects and one male subject, in which nonrandomed X inactivation did not occur, gothic palate could be supposed as characteristic of IP. |
doi_str_mv | 10.1007/s00784-006-0066-z |
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One such example is incontinentia pigmenti (IP), a rare, complex, X-linked genodermatosis. Clinical manifestations of IP according to evolution and prognosis can be considered as skin, as well as dental, eye, and central nervous system, changes. We have investigated a total of nine families with 25 subjects, 23 females and 2 males. In 12 female and 2 male subjects, all of them with clinical characteristics of IP, we observed the following abnormalities: teeth-shape anomalies (coni- or peg-like teeth), the presence of numerous cariotic teeth, early dental loss, delayed eruption, partial anodontia, and gothic palate. To our knowledge, this is the first time that the presence of gothic palate in patients with IP has been documented. As we found out, in two female subjects and one male subject, in which nonrandomed X inactivation did not occur, gothic palate could be supposed as characteristic of IP.</description><identifier>ISSN: 1432-6981</identifier><identifier>EISSN: 1436-3771</identifier><identifier>DOI: 10.1007/s00784-006-0066-z</identifier><identifier>PMID: 16896835</identifier><language>eng</language><publisher>Germany: Springer Nature B.V</publisher><subject>Adult ; Child ; Child, Preschool ; Dentistry ; Female ; Humans ; Incontinentia Pigmenti - genetics ; Infant ; Infant, Newborn ; Male ; Middle Aged ; Palate - abnormalities ; Pedigree ; Tooth Abnormalities - genetics</subject><ispartof>Clinical oral investigations, 2006-12, Vol.10 (4), p.343-347</ispartof><rights>Springer-Verlag 2006</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c326t-c7fb434b7f73aaabf230ccbc0a016bc992851e2a06434e2ae504ea56d9a304983</citedby><cites>FETCH-LOGICAL-c326t-c7fb434b7f73aaabf230ccbc0a016bc992851e2a06434e2ae504ea56d9a304983</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/16896835$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Minić, Snezana</creatorcontrib><creatorcontrib>Novotny, Gerd E K</creatorcontrib><creatorcontrib>Trpinac, Dusan</creatorcontrib><creatorcontrib>Obradović, Miljana</creatorcontrib><title>Clinical features of incontinentia pigmenti with emphasis on oral and dental abnormalities</title><title>Clinical oral investigations</title><addtitle>Clin Oral Investig</addtitle><description>One of interesting aspects in dermatology is the fact that skin may reflect the presence of anomalies in other organs and tissues. One such example is incontinentia pigmenti (IP), a rare, complex, X-linked genodermatosis. Clinical manifestations of IP according to evolution and prognosis can be considered as skin, as well as dental, eye, and central nervous system, changes. We have investigated a total of nine families with 25 subjects, 23 females and 2 males. In 12 female and 2 male subjects, all of them with clinical characteristics of IP, we observed the following abnormalities: teeth-shape anomalies (coni- or peg-like teeth), the presence of numerous cariotic teeth, early dental loss, delayed eruption, partial anodontia, and gothic palate. To our knowledge, this is the first time that the presence of gothic palate in patients with IP has been documented. As we found out, in two female subjects and one male subject, in which nonrandomed X inactivation did not occur, gothic palate could be supposed as characteristic of IP.</description><subject>Adult</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Dentistry</subject><subject>Female</subject><subject>Humans</subject><subject>Incontinentia Pigmenti - genetics</subject><subject>Infant</subject><subject>Infant, Newborn</subject><subject>Male</subject><subject>Middle Aged</subject><subject>Palate - abnormalities</subject><subject>Pedigree</subject><subject>Tooth Abnormalities - genetics</subject><issn>1432-6981</issn><issn>1436-3771</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2006</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><sourceid>GNUQQ</sourceid><recordid>eNpdkE1LxDAQhoMorq7-AC8SPHir5qtpepTFL1jwohcvYZqmbpY2XZMWcX-9qbsgeJiZF-Z5h-FF6IKSG0pIcRtTUyIjRE4ls-0BOqGCy4wXBT381SyTpaIzdBrjmhAqZMGP0YxKVUrF8xP0vmiddwZa3FgYxmAj7hvsvOn94LxNDfDGfXSTwl9uWGHbbVYQXeI87kMygq9xnfaTrHwfOmjd4Gw8Q0cNtNGe7-ccvT3cvy6esuXL4_PibpkZzuSQmaKpBBdV0RQcAKqGcWJMZQgQKitTlkzl1DIgMlFp2pwIC7msS-BElIrP0fXu7ib0n6ONg-5cNLZtwdt-jFoqRgRRPIFX_8B1PwafftOMlZIzpmSC6A4yoY8x2EZvgusgfGtK9JS63qWuU-JTSb1Nnsv94bHqbP3n2MfMfwB6ln5v</recordid><startdate>20061201</startdate><enddate>20061201</enddate><creator>Minić, Snezana</creator><creator>Novotny, Gerd E K</creator><creator>Trpinac, Dusan</creator><creator>Obradović, Miljana</creator><general>Springer Nature B.V</general><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>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8C1</scope><scope>8FE</scope><scope>8FH</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BBNVY</scope><scope>BENPR</scope><scope>BHPHI</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>HCIFZ</scope><scope>K9.</scope><scope>LK8</scope><scope>M0S</scope><scope>M1P</scope><scope>M7P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope></search><sort><creationdate>20061201</creationdate><title>Clinical features of incontinentia pigmenti with emphasis on oral and dental abnormalities</title><author>Minić, Snezana ; Novotny, Gerd E K ; Trpinac, Dusan ; Obradović, Miljana</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c326t-c7fb434b7f73aaabf230ccbc0a016bc992851e2a06434e2ae504ea56d9a304983</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2006</creationdate><topic>Adult</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Dentistry</topic><topic>Female</topic><topic>Humans</topic><topic>Incontinentia Pigmenti - genetics</topic><topic>Infant</topic><topic>Infant, Newborn</topic><topic>Male</topic><topic>Middle Aged</topic><topic>Palate - abnormalities</topic><topic>Pedigree</topic><topic>Tooth Abnormalities - genetics</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Minić, Snezana</creatorcontrib><creatorcontrib>Novotny, Gerd E K</creatorcontrib><creatorcontrib>Trpinac, Dusan</creatorcontrib><creatorcontrib>Obradović, Miljana</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Public Health Database</collection><collection>ProQuest SciTech Collection</collection><collection>ProQuest Natural Science Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>Biological Science Collection</collection><collection>ProQuest Central</collection><collection>Natural Science Collection</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>SciTech Premium Collection</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>ProQuest Biological Science Collection</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Biological Science Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><jtitle>Clinical oral investigations</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Minić, Snezana</au><au>Novotny, Gerd E K</au><au>Trpinac, Dusan</au><au>Obradović, Miljana</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Clinical features of incontinentia pigmenti with emphasis on oral and dental abnormalities</atitle><jtitle>Clinical oral investigations</jtitle><addtitle>Clin Oral Investig</addtitle><date>2006-12-01</date><risdate>2006</risdate><volume>10</volume><issue>4</issue><spage>343</spage><epage>347</epage><pages>343-347</pages><issn>1432-6981</issn><eissn>1436-3771</eissn><abstract>One of interesting aspects in dermatology is the fact that skin may reflect the presence of anomalies in other organs and tissues. One such example is incontinentia pigmenti (IP), a rare, complex, X-linked genodermatosis. Clinical manifestations of IP according to evolution and prognosis can be considered as skin, as well as dental, eye, and central nervous system, changes. We have investigated a total of nine families with 25 subjects, 23 females and 2 males. In 12 female and 2 male subjects, all of them with clinical characteristics of IP, we observed the following abnormalities: teeth-shape anomalies (coni- or peg-like teeth), the presence of numerous cariotic teeth, early dental loss, delayed eruption, partial anodontia, and gothic palate. To our knowledge, this is the first time that the presence of gothic palate in patients with IP has been documented. As we found out, in two female subjects and one male subject, in which nonrandomed X inactivation did not occur, gothic palate could be supposed as characteristic of IP.</abstract><cop>Germany</cop><pub>Springer Nature B.V</pub><pmid>16896835</pmid><doi>10.1007/s00784-006-0066-z</doi><tpages>5</tpages></addata></record> |
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subjects | Adult Child Child, Preschool Dentistry Female Humans Incontinentia Pigmenti - genetics Infant Infant, Newborn Male Middle Aged Palate - abnormalities Pedigree Tooth Abnormalities - genetics |
title | Clinical features of incontinentia pigmenti with emphasis on oral and dental abnormalities |
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