Papillon–Lefèvre syndrome: a series of five cases among siblings
Identifieur interne : 000627 ( Main/Exploration ); précédent : 000626; suivant : 000628Papillon–Lefèvre syndrome: a series of five cases among siblings
Auteurs : Zyad M. Aibarrak [Arabie saoudite] ; Adel S. Alqarni ; Elna P. Chalisserry [Arabie saoudite] ; Sukumaran AnilSource :
- Journal of Medical Case Reports [ 1752-1947 ] ; 2016.
Abstract
Papillon–Lefèvre syndrome is a rare autosomal recessive disorder characterized by palmoplantar hyperkeratosis and aggressively progressing periodontitis leading to premature loss of deciduous and permanent dentition. The etiopathogenesis of the syndrome is relatively obscure, and immunologic, genetic, or possible bacterial etiologies have been proposed.
A series of five cases of Papillon–Lefèvre syndrome among the siblings in a family is presented here: a 3-year-old Arab girl, a 4-year-old Arab boy, a 11-year-old Arab boy, a 12-year-old Arab boy, and a 14-year-old Arab boy. The patients presented with severe gingival inflammation and mobility of teeth. The clinical manifestations were typical of Papillon–Lefèvre syndrome and the degree of involvement of the oral and skin conditions varied among them.
This case series stresses the consanguinity in the family as an etiologic factor. All siblings in the family were affected with Papillon–Lefèvre syndrome which makes this a rare case. A multidisciplinary approach with the active participation of a dental surgeon, dermatologist, and pediatrician is essential for the management of cases of Papillon–Lefèvre syndrome.
Url:
DOI: 10.1186/s13256-016-1051-z
PubMed: 27658951
PubMed Central: 5034476
Affiliations:
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Papillon–Lefèvre syndrome: a series of five cases among siblings</title><author><name sortKey="Aibarrak, Zyad M" sort="Aibarrak, Zyad M" uniqKey="Aibarrak Z" first="Zyad M." last="Aibarrak">Zyad M. Aibarrak</name><affiliation wicri:level="1"><nlm:aff id="Aff1">King Abdulaziz Medical City, Ministry of Health, Riyadh, Saudi Arabia</nlm:aff><country xml:lang="fr">Arabie saoudite</country><wicri:regionArea>King Abdulaziz Medical City, Ministry of Health, Riyadh</wicri:regionArea><wicri:noRegion>Riyadh</wicri:noRegion></affiliation></author><author><name sortKey="Alqarni, Adel S" sort="Alqarni, Adel S" uniqKey="Alqarni A" first="Adel S." last="Alqarni">Adel S. Alqarni</name><affiliation><nlm:aff id="Aff2">Department of Preventive Dental Sciences, College of Dentistry, Prince Sattam Bin Abdulaziz University, 153, AIkharj, 11942 Riyadh Saudi Arabia</nlm:aff><wicri:noCountry code="subfield">11942 Riyadh Saudi Arabia</wicri:noCountry></affiliation></author><author><name sortKey="Chalisserry, Elna P" sort="Chalisserry, Elna P" uniqKey="Chalisserry E" first="Elna P." last="Chalisserry">Elna P. Chalisserry</name><affiliation wicri:level="1"><nlm:aff id="Aff3">Department of Maxillofacial Surgery and Diagnostic Sciences, College of Dentistry, Jazan University, Jazan, 82943 Saudi Arabia</nlm:aff><country xml:lang="fr">Arabie saoudite</country><wicri:regionArea>Department of Maxillofacial Surgery and Diagnostic Sciences, College of Dentistry, Jazan University, Jazan</wicri:regionArea><wicri:noRegion>Jazan</wicri:noRegion></affiliation></author><author><name sortKey="Anil, Sukumaran" sort="Anil, Sukumaran" uniqKey="Anil S" first="Sukumaran" last="Anil">Sukumaran Anil</name><affiliation><nlm:aff id="Aff2">Department of Preventive Dental Sciences, College of Dentistry, Prince Sattam Bin Abdulaziz University, 153, AIkharj, 11942 Riyadh Saudi Arabia</nlm:aff><wicri:noCountry code="subfield">11942 Riyadh Saudi Arabia</wicri:noCountry></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">27658951</idno><idno type="pmc">5034476</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5034476</idno><idno type="RBID">PMC:5034476</idno><idno type="doi">10.1186/s13256-016-1051-z</idno><date when="2016">2016</date><idno type="wicri:Area/Pmc/Corpus">000982</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000982</idno><idno type="wicri:Area/Pmc/Curation">000982</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Curation">000982</idno><idno type="wicri:Area/Pmc/Checkpoint">000547</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Checkpoint">000547</idno><idno type="wicri:Area/Ncbi/Merge">006A54</idno><idno type="wicri:Area/Ncbi/Curation">006A54</idno><idno type="wicri:Area/Ncbi/Checkpoint">006A54</idno><idno type="wicri:Area/Main/Merge">000627</idno><idno type="wicri:Area/Main/Curation">000627</idno><idno type="wicri:Area/Main/Exploration">000627</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Papillon–Lefèvre syndrome: a series of five cases among siblings</title><author><name sortKey="Aibarrak, Zyad M" sort="Aibarrak, Zyad M" uniqKey="Aibarrak Z" first="Zyad M." last="Aibarrak">Zyad M. Aibarrak</name><affiliation wicri:level="1"><nlm:aff id="Aff1">King Abdulaziz Medical City, Ministry of Health, Riyadh, Saudi Arabia</nlm:aff><country xml:lang="fr">Arabie saoudite</country><wicri:regionArea>King Abdulaziz Medical City, Ministry of Health, Riyadh</wicri:regionArea><wicri:noRegion>Riyadh</wicri:noRegion></affiliation></author><author><name sortKey="Alqarni, Adel S" sort="Alqarni, Adel S" uniqKey="Alqarni A" first="Adel S." last="Alqarni">Adel S. Alqarni</name><affiliation><nlm:aff id="Aff2">Department of Preventive Dental Sciences, College of Dentistry, Prince Sattam Bin Abdulaziz University, 153, AIkharj, 11942 Riyadh Saudi Arabia</nlm:aff><wicri:noCountry code="subfield">11942 Riyadh Saudi Arabia</wicri:noCountry></affiliation></author><author><name sortKey="Chalisserry, Elna P" sort="Chalisserry, Elna P" uniqKey="Chalisserry E" first="Elna P." last="Chalisserry">Elna P. Chalisserry</name><affiliation wicri:level="1"><nlm:aff id="Aff3">Department of Maxillofacial Surgery and Diagnostic Sciences, College of Dentistry, Jazan University, Jazan, 82943 Saudi Arabia</nlm:aff><country xml:lang="fr">Arabie saoudite</country><wicri:regionArea>Department of Maxillofacial Surgery and Diagnostic Sciences, College of Dentistry, Jazan University, Jazan</wicri:regionArea><wicri:noRegion>Jazan</wicri:noRegion></affiliation></author><author><name sortKey="Anil, Sukumaran" sort="Anil, Sukumaran" uniqKey="Anil S" first="Sukumaran" last="Anil">Sukumaran Anil</name><affiliation><nlm:aff id="Aff2">Department of Preventive Dental Sciences, College of Dentistry, Prince Sattam Bin Abdulaziz University, 153, AIkharj, 11942 Riyadh Saudi Arabia</nlm:aff><wicri:noCountry code="subfield">11942 Riyadh Saudi Arabia</wicri:noCountry></affiliation></author></analytic><series><title level="j">Journal of Medical Case Reports</title><idno type="eISSN">1752-1947</idno><imprint><date when="2016">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><sec><title>Background</title><p>Papillon–Lefèvre syndrome is a rare autosomal recessive disorder characterized by palmoplantar hyperkeratosis and aggressively progressing periodontitis leading to premature loss of deciduous and permanent dentition. The etiopathogenesis of the syndrome is relatively obscure, and immunologic, genetic, or possible bacterial etiologies have been proposed.</p></sec><sec><title>Case presentation</title><p>A series of five cases of Papillon–Lefèvre syndrome among the siblings in a family is presented here: a 3-year-old Arab girl, a 4-year-old Arab boy, a 11-year-old Arab boy, a 12-year-old Arab boy, and a 14-year-old Arab boy. The patients presented with severe gingival inflammation and mobility of teeth. The clinical manifestations were typical of Papillon–Lefèvre syndrome and the degree of involvement of the oral and skin conditions varied among them.</p></sec><sec><title>Conclusions</title><p>This case series stresses the consanguinity in the family as an etiologic factor. All siblings in the family were affected with Papillon–Lefèvre syndrome which makes this a rare case. A multidisciplinary approach with the active participation of a dental surgeon, dermatologist, and pediatrician is essential for the management of cases of Papillon–Lefèvre syndrome.</p></sec></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Gorlin, Rj" uniqKey="Gorlin R">RJ Gorlin</name></author><author><name sortKey="Sedano, H" uniqKey="Sedano H">H Sedano</name></author><author><name sortKey="Anderson, Ve" uniqKey="Anderson V">VE Anderson</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hattab, Fn" uniqKey="Hattab F">FN Hattab</name></author><author><name sortKey="Rawashdeh, Ma" uniqKey="Rawashdeh M">MA Rawashdeh</name></author><author><name sortKey="Yassin, Om" uniqKey="Yassin O">OM Yassin</name></author><author><name sortKey="Al Momani, As" uniqKey="Al Momani A">AS al-Momani</name></author><author><name sortKey="Al Ubosi, Mm" uniqKey="Al Ubosi M">MM 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Adel S" uniqKey="Alqarni A" first="Adel S." last="Alqarni">Adel S. Alqarni</name><name sortKey="Anil, Sukumaran" sort="Anil, Sukumaran" uniqKey="Anil S" first="Sukumaran" last="Anil">Sukumaran Anil</name></noCountry><country name="Arabie saoudite"><noRegion><name sortKey="Aibarrak, Zyad M" sort="Aibarrak, Zyad M" uniqKey="Aibarrak Z" first="Zyad M." last="Aibarrak">Zyad M. Aibarrak</name></noRegion><name sortKey="Chalisserry, Elna P" sort="Chalisserry, Elna P" uniqKey="Chalisserry E" first="Elna P." last="Chalisserry">Elna P. Chalisserry</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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