Mal de Meleda with Lip Involvement: A Report of Two Cases
Identifieur interne : 004235 ( Ncbi/Merge ); précédent : 004234; suivant : 004236Mal de Meleda with Lip Involvement: A Report of Two Cases
Auteurs : Amiya Kumar Nath ; Sangita Chaudhuri ; Devinder Mohan ThappaSource :
- Indian Journal of Dermatology [ 0019-5154 ] ; 2012.
Abstract
Mal de Meleda is a rare autosomal recessive transgradient palmoplantar keratoderma characterized by transgradient keratoderma with associated scleroatrophy, nail changes, pseudoainhum around digits and perioral erythema, without a tendency for spontaneous resolution. Involvement of the lip by keratoderma has not been reported in the English literature. Here we present two cases of Mal de Meleda with unusual lip involvement. The first case was a 15-year-old girl, born of second-degree consanguineous marriage, who presented with transgradient palmoplantar keratoderma from 6 months of age, with lichenoid papules and plaques on the elbows and knees, conical tapering of the distal digits, flexion deformity of several fingers, digital constriction, knuckle pads and lip involvement. The second case was a 24-year-old male with transgradient palmoplantar keratoderma since birth. He also had scaly plaques on the extensors of bilateral knees and elbows, knuckle pads, pseudosclerodermatous fingers with conical tapering, digital constrictions at various places with mild flexion deformity and lip involvement. Both patients were otherwise normal without any family history.
Url:
DOI: 10.4103/0019-5154.100497
PubMed: 23112362
PubMed Central: 3482805
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<front><div type="abstract" xml:lang="en"><p>Mal de Meleda is a rare autosomal recessive transgradient palmoplantar keratoderma characterized by transgradient keratoderma with associated scleroatrophy, nail changes, pseudoainhum around digits and perioral erythema, without a tendency for spontaneous resolution. Involvement of the lip by keratoderma has not been reported in the English literature. Here we present two cases of Mal de Meleda with unusual lip involvement. The first case was a 15-year-old girl, born of second-degree consanguineous marriage, who presented with transgradient palmoplantar keratoderma from 6 months of age, with lichenoid papules and plaques on the elbows and knees, conical tapering of the distal digits, flexion deformity of several fingers, digital constriction, knuckle pads and lip involvement. The second case was a 24-year-old male with transgradient palmoplantar keratoderma since birth. He also had scaly plaques on the extensors of bilateral knees and elbows, knuckle pads, pseudosclerodermatous fingers with conical tapering, digital constrictions at various places with mild flexion deformity and lip involvement. Both patients were otherwise normal without any family history.</p>
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<front><journal-meta><journal-id journal-id-type="nlm-ta">Indian J Dermatol</journal-id>
<journal-id journal-id-type="iso-abbrev">Indian J Dermatol</journal-id>
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<article-id pub-id-type="pmc">3482805</article-id>
<article-id pub-id-type="publisher-id">IJD-57-390</article-id>
<article-id pub-id-type="doi">10.4103/0019-5154.100497</article-id>
<article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject>
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<title-group><article-title>Mal de Meleda with Lip Involvement: A Report of Two Cases</article-title>
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<contrib-group><contrib contrib-type="author"><name><surname>Nath</surname>
<given-names>Amiya Kumar</given-names>
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<contrib contrib-type="author"><name><surname>Chaudhuri</surname>
<given-names>Sangita</given-names>
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<xref ref-type="aff" rid="aff2">1</xref>
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<contrib contrib-type="author"><name><surname>Thappa</surname>
<given-names>Devinder Mohan</given-names>
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<xref ref-type="aff" rid="aff2">1</xref>
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<aff id="aff1"><italic>From the Department of Dermatology and STD, Indira Gandhi Medical College and Research Institute, Pondicherry, India</italic>
</aff>
<aff id="aff2"><label>1</label>
<italic>Department of Dermatology and STD, Jawaharlal Institute of Postgraduate Medical Education and Research (JIPMER), Pondicherry, India</italic>
</aff>
<author-notes><corresp id="cor1"><bold><italic>Address for correspondence:</italic>
</bold>
<italic>Dr. Devinder Mohan Thappa, Department of Dermatology and STD, JIPMER, Pondicherry - 605 006, India. E-mail: <email xlink:href="dmthappa@gmail.com">dmthappa@gmail.com</email>
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<pub-date pub-type="ppub"><season>Sep-Oct</season>
<year>2012</year>
</pub-date>
<volume>57</volume>
<issue>5</issue>
<fpage>390</fpage>
<lpage>393</lpage>
<history><date date-type="received"><month>9</month>
<year>2010</year>
</date>
<date date-type="accepted"><month>11</month>
<year>2010</year>
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<permissions><copyright-statement>Copyright: © Indian Journal of Dermatology</copyright-statement>
<copyright-year>2012</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc-sa/3.0"><license-p>This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
</license>
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<abstract><p>Mal de Meleda is a rare autosomal recessive transgradient palmoplantar keratoderma characterized by transgradient keratoderma with associated scleroatrophy, nail changes, pseudoainhum around digits and perioral erythema, without a tendency for spontaneous resolution. Involvement of the lip by keratoderma has not been reported in the English literature. Here we present two cases of Mal de Meleda with unusual lip involvement. The first case was a 15-year-old girl, born of second-degree consanguineous marriage, who presented with transgradient palmoplantar keratoderma from 6 months of age, with lichenoid papules and plaques on the elbows and knees, conical tapering of the distal digits, flexion deformity of several fingers, digital constriction, knuckle pads and lip involvement. The second case was a 24-year-old male with transgradient palmoplantar keratoderma since birth. He also had scaly plaques on the extensors of bilateral knees and elbows, knuckle pads, pseudosclerodermatous fingers with conical tapering, digital constrictions at various places with mild flexion deformity and lip involvement. Both patients were otherwise normal without any family history.</p>
</abstract>
<kwd-group><kwd><italic>Lip</italic>
</kwd>
<kwd><italic>Mal de Meleda</italic>
</kwd>
<kwd><italic>palmoplantar keratoderma</italic>
</kwd>
<kwd><italic>transgradient</italic>
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<tree><noCountry><name sortKey="Chaudhuri, Sangita" sort="Chaudhuri, Sangita" uniqKey="Chaudhuri S" first="Sangita" last="Chaudhuri">Sangita Chaudhuri</name>
<name sortKey="Nath, Amiya Kumar" sort="Nath, Amiya Kumar" uniqKey="Nath A" first="Amiya Kumar" last="Nath">Amiya Kumar Nath</name>
<name sortKey="Thappa, Devinder Mohan" sort="Thappa, Devinder Mohan" uniqKey="Thappa D" first="Devinder Mohan" last="Thappa">Devinder Mohan Thappa</name>
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