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<article xml:lang="EN" article-type="case-report">

<front>
<journal-meta>
<journal-id journal-id-type="nlm-ta">Ann Dermatol</journal-id>
<journal-id journal-id-type="publisher-id">AD</journal-id>
<journal-title>Annals of Dermatology</journal-title>
<issn pub-type="ppub">1013-9087</issn>
<issn pub-type="epub">2005-3894</issn>
<publisher>
<publisher-name>Korean Dermatological Association; The Korean Society for Investigative Dermatology</publisher-name>
</publisher>
</journal-meta>

<article-meta>
<article-id pub-id-type="doi">10.5021/ad.2010.22.3.323</article-id>

<article-categories>
<subj-group>
<subject>Case Report</subject>
</subj-group>
</article-categories>
<title-group>
<article-title>A Case of Linear Lichen Planus Pigmentosus</article-title>
</title-group>

<contrib-group>

<contrib contrib-type="author">
<name>
<surname>Seo</surname>
<given-names>Jong Keun</given-names>
</name>
<degrees>M.D.</degrees>
<xref ref-type="aff" rid="A1"></xref>
</contrib>

<contrib contrib-type="author">
<name>
<surname>Lee</surname>
<given-names>Hyun Jae</given-names>
</name>
<degrees>M.D.</degrees>
<xref ref-type="aff" rid="A1"></xref>
</contrib>

<contrib contrib-type="author" corresp="yes">
<name>
<surname>Lee</surname>
<given-names>Deborah</given-names>
</name>
<degrees>M.D.</degrees>
<xref ref-type="aff" rid="A1"></xref>
</contrib>

<contrib contrib-type="author">
<name>
<surname>Choi</surname>
<given-names>Joon Hee</given-names>
</name>
<degrees>M.D.</degrees>
<xref ref-type="aff" rid="A2">1</xref>
</contrib>

<contrib contrib-type="author">
<name>
<surname>Sung</surname>
<given-names>Ho-Suck</given-names>
</name>
<degrees>M.D.</degrees>
<xref ref-type="aff" rid="A1"></xref>
</contrib>

</contrib-group>

<aff id="A1">Department of Dermatology, Busan Paik Hospital, College of Medicine, Inje University, Busan, Korea.</aff>
<aff id="A2"><label>1</label>Department of Dermatology, Maryknoll Medical Center, Busan, Korea.</aff>

<author-notes>
<corresp>Corresponding author: Deborah Lee, M.D., Department of Dermatology, Busan Paik Hospital, College of Medicine, Inje University, 633-165 Gaegum-dong, Busanjin-gu, Busan 614-735, Korea. Tel: 82-51-890-6135, Fax: 82-51-897-6391, <email>btyouth@hanmail.net</email>
</corresp>
</author-notes>

<pub-date pub-type="ppub">
<month>08</month>
<year>2010</year>
</pub-date>
<pub-date pub-type="epub">
<day>05</day>
<month>08</month>
<year>2010</year>
</pub-date>
<volume>22</volume>
<issue>3</issue>
<fpage>323</fpage>
<lpage>325</lpage>
<history>
<date date-type="received">
<day>20</day>
<month>07</month>
<year>2009</year>
</date>
<date date-type="rev-recd">
<day>09</day>
<month>11</month>
<year>2009</year>
</date>
<date date-type="accepted">
<day>09</day>
<month>11</month>
<year>2009</year>
</date>
</history>
<permissions>
<copyright-statement>Copyright &#x00A9; 2010 Korean Dermatological Association; The Korean Society for Investigative Dermatology</copyright-statement>
<copyright-year>2010</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/3.0">
<p>This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (<ext-link ext-link-type="uri" xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="http://creativecommons.org/licenses/by-nc/3.0">http://creativecommons.org/licenses/by-nc/3.0</ext-link>) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.</p>
</license>
</permissions>

<abstract>
<p>Lichen planus pigmentosus (LPP) is chronic pigmentary disorder that shows diffuse or reticulated hyperpigmented, dark brown macules on the sun-exposed areas such as the face, neck and other flexural folds. Clinically, it is different from classical lichen planus because LPP has a longer clinical course and it manifests with dark brown macules. In case of LPP, involvement of the scalp, nail or mucosal area is rare. The histopathological findings of the lesions show an atrophic epidermis, the presence of melanophages and a vacuolar alteration of the basal cell layer with a sparse lymphohistiocytic lichenoid infiltration. Although there have been a few reports of LPP, there have only 3 cases of linear LPP along the lines of Blaschko in the Korean dermatologic literature. Our patient had lesions on the neck and chin with a linear pattern. In this report, we describe a very rare case of LPP with a linear distribution related to Blaschko's lines on the neck and chin areas.</p>
</abstract>

<kwd-group>
<kwd>Blaschko's lines</kwd>
<kwd>Lichen planus pigmentosus</kwd>
<kwd>Linear</kwd>
</kwd-group>

</article-meta>
</front>

<body>
<sec sec-type="intro">
<title>INTRODUCTION</title>
  <p>Lichen planus pigmentosus (LPP) was first described by Bhutani et al.<xref ref-type="bibr" rid="B1">1</xref>, and this a rare variant of lichen planus, and LPP appears as mottled or reticulated hyperpigmented, dark brown macules or papules on the sun exposed areas and flexural folds<xref ref-type="bibr" rid="B2">2</xref>,<xref ref-type="bibr" rid="B3">3</xref>. Linear lichen planus, which shows a linear distribution following Blaschko's lines, is considered a subtype of lichen planus<xref ref-type="bibr" rid="B3">3</xref>-<xref ref-type="bibr" rid="B5">5</xref>.</p>
  <p>The histologic findings of LPP show hyperkeratosis and atrophy of the epidermis with vacuolar degeneration of the basal layer. A perivascular lymphohistiocytic infiltration and pigmentary incontinence in the dermis are also noted<xref ref-type="bibr" rid="B2">2</xref>,<xref ref-type="bibr" rid="B6">6</xref>.</p>
  <p>Linear LPP is considered as a combined type of linear lichen planus and LPP, and there have been only a few reports on this in the dermatologic literature<xref ref-type="bibr" rid="B3">3</xref>. Only 3 such cases have been reported in the Korean dermatologic literature<xref ref-type="bibr" rid="B2">2</xref>,<xref ref-type="bibr" rid="B7">7</xref>. Here we report on a case of linear LPP in a 60-year-old male.</p>
</sec>

<sec sec-type="cases">
<title>CASE REPORT</title>
  <p>A 60-year-old man presented with an asymptomatic hyperpigmented linear patch on the anterior neck and chin and he'd had this patch for 8 month. One dark brown macule first appeared on the neck, and it gradually spread to the chin. There was no history of excessive sun exposure or trauma on that site. On examination, he had a linear streak of dark brown patches from the chin and across the neck following one of Blaschko's lines (<xref ref-type="fig" rid="F1">Fig. 1</xref>). There was no other skin lesion and the oral mucosa and nails were not involved. He had previously suffered with gastric cancer and he had undergone gastrectomy at the age of 47 years.</p>
  <p>The routine laboratory data, including the peripheral blood cells and liver function tests, was normal. Skin biopsy showed epidermal thinning, basal cell degeneration, some dyskeratotic cells with pigment incontinence and a lymphohistiocytic infiltration in the dermis (<xref ref-type="fig" rid="F2">Fig. 2</xref>). He was treated with systemic and topical steroid, but the lesion still remained unchanged for 7 months.</p>
</sec>

<sec sec-type="discussion">
<title>DISCUSSION</title>
  <p>LPP is characterized by mottled or reticulated hyperpigmented, dark brown macules on the sun exposure skin areas and flexural folds on the face and neck, and the axillae and inguinal regions<xref ref-type="bibr" rid="B1">1</xref>-<xref ref-type="bibr" rid="B3">3</xref>.</p>
  <p>The cause of LPP is unknown, but an immunologic mechanism mediates its development, as well as that of lichen planus<xref ref-type="bibr" rid="B4">4</xref>. Based on the distinctive lymphocytic inflammatory response of the lichenoid reactions, cell mediated immunity seems to play a pivotal role in triggering the clinical expression of the disease. T-lymphocytes are pivotal as they regulate epidermal cell recognition, the lichenoid response and epithelial destruction<xref ref-type="bibr" rid="B2">2</xref>-<xref ref-type="bibr" rid="B4">4</xref>,<xref ref-type="bibr" rid="B8">8</xref>. Clinically, LPP differs from classical lichen planus by exhibiting dark brown macules and/or papules and a longer clinical course<xref ref-type="bibr" rid="B2">2</xref>.</p>
  <p>Kanwar et al.<xref ref-type="bibr" rid="B6">6</xref> described flexural involvement over the axillae (8.9&#x0025;), inflamed skin folds (6.5&#x0025;) and groin (3.2&#x0025;) in 123 Indian patients with LPP. The histopathological findings of LPP consist of hyperkeratosis, atrophic epidermis with vacuolar alteration of the basal layer and scarce lymphohistiocytic or lichenoid infiltrates in the dermis with pigment incontinence<xref ref-type="bibr" rid="B1">1</xref>-<xref ref-type="bibr" rid="B3">3</xref>,<xref ref-type="bibr" rid="B8">8</xref>.</p>
  <p>Although there have been a few reports of LPP, there have been only 2 reports of LPP with a linear pattern in the Korean dermatologic literature<xref ref-type="bibr" rid="B2">2</xref>. Our patient had lesion on the neck and chin with a linear pattern. The linearity of the lesion is probably related to Blaschko's lines, which suggests that the predisposition to develop LPP might be determined during embryogenesis<xref ref-type="bibr" rid="B5">5</xref>.</p>
  <p>Blaschko's lines are thought to reflect T-lymphocytic migration and the clonal expression during embryogenesis of the skin. The genetic mosaicism in an acquired Blaschko-linear inflammatory dermatosis could be responsible for the cutaneous antigenic mosaicism that may induce a mosaic T-cell response according to the trigger of viral infection or drugs. Some apoptotic change in our cases may be responsible for the response of the mosaic T cells that are present along Blaschko's lines<xref ref-type="bibr" rid="B2">2</xref>,<xref ref-type="bibr" rid="B5">5</xref>.</p>
  <p>The differential diagnosis of our case includes linear dermatosis such as lichen striatus, linear and whorled nevoid hypermelanosis, ashy dermatosis and postinflammatory hyperpigmentation. Lichen striatus almost always has preceding inflammatory papules or a scaly eruption, which last for 4 months to 4 years and a perivascular and periadnexal inflammatory cell infiltration<xref ref-type="bibr" rid="B4">4</xref>,<xref ref-type="bibr" rid="B8">8</xref>. There were no previous papules on our patient and the histological findings were more consistent with LPP. Linear and whorled nevoid hypermelanosis usually presents within a few weeks of birth and it is associated with congenital anomalies. The histologic findings have shown basal pigmentation without pigment incontinence<xref ref-type="bibr" rid="B3">3</xref>,<xref ref-type="bibr" rid="B4">4</xref>. In ashy dermatosis and postinflammatory hyperpigmentation, the histopathologic findings show a slightly smoothed epidermis with a scanty perivascular lymphocytic infiltration. However, there was a dense lymphohistiocytic infiltration in our case. Although erythema multiforme and fixed drug eruptions show histopathological findings similar to those of our case, there was no apparent epidermal necrosis in our case.</p>
  <p>In conclusion, LPP can present along the lines of Blaschko. Therefore, we should consider LPP in the differential diagnosis of a linear pigmentary disorder.</p>
</sec>
</body>

<back>

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<floats-wrap>

<fig position="float" id="F1">
<label>Fig. 1</label>
<caption>
  <p>Linear streaks of dark brown patches from the chin and across the neck.</p>
</caption>
<graphic xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="ad-22-323-g001" alt-version="no"></graphic>
</fig>

<fig position="float" id="F2">
<label>Fig. 2</label>
<caption>
  <p>Epidermal thinning, basal cell degeneration, some dyskeratotic cells, pigment incontinence and a lymphohistiocytic infiltration in the dermis (A: H&#x0026;E stain, &#x00D7;40, B: H&#x0026;E stain, &#x00D7;200).</p>
</caption>
<graphic xmlns:xlink="http://www.w3.org/1999/xlink" xlink:href="ad-22-323-g002" alt-version="no"></graphic>
</fig>

</floats-wrap>
</article>