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Neutrophilic Dermatosis Confined to the Lymphedematous Area

Identifieur interne : 002540 ( Pmc/Corpus ); précédent : 002539; suivant : 002541

Neutrophilic Dermatosis Confined to the Lymphedematous Area

Auteurs : Ji-Youn Park ; Hee Young Kang ; You Chan Kim

Source :

RBID : PMC:4069661
Url:
DOI: 10.5021/ad.2014.26.3.411
PubMed: 24966650
PubMed Central: 4069661

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PMC:4069661

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<name sortKey="Kang, Hee Young" sort="Kang, Hee Young" uniqKey="Kang H" first="Hee Young" last="Kang">Hee Young Kang</name>
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<name sortKey="Kim, You Chan" sort="Kim, You Chan" uniqKey="Kim Y" first="You Chan" last="Kim">You Chan Kim</name>
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<name sortKey="Kim, You Chan" sort="Kim, You Chan" uniqKey="Kim Y" first="You Chan" last="Kim">You Chan Kim</name>
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<name sortKey="Mortimer, P" uniqKey="Mortimer P">P Mortimer</name>
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<name sortKey="Ryan, Tj" uniqKey="Ryan T">TJ Ryan</name>
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<name sortKey="Demitsu, T" uniqKey="Demitsu T">T Demitsu</name>
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<name sortKey="Tadaki, T" uniqKey="Tadaki T">T Tadaki</name>
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<name sortKey="Garcia Rio, I" uniqKey="Garcia Rio I">I García-Río</name>
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<name sortKey="Perez Gala, S" uniqKey="Perez Gala S">S Pérez-Gala</name>
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<name sortKey="Aragues, M" uniqKey="Aragues M">M Aragüés</name>
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<name sortKey="Fernandez Herrera, J" uniqKey="Fernandez Herrera J">J Fernández-Herrera</name>
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<name sortKey="Fraga, J" uniqKey="Fraga J">J Fraga</name>
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<pmc-dir>properties open_access</pmc-dir>
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<journal-meta>
<journal-id journal-id-type="nlm-ta">Ann Dermatol</journal-id>
<journal-id journal-id-type="iso-abbrev">Ann Dermatol</journal-id>
<journal-id journal-id-type="publisher-id">AD</journal-id>
<journal-title-group>
<journal-title>Annals of Dermatology</journal-title>
</journal-title-group>
<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>
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<article-id pub-id-type="pmid">24966650</article-id>
<article-id pub-id-type="pmc">4069661</article-id>
<article-id pub-id-type="doi">10.5021/ad.2014.26.3.411</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Letter to the Editor</subject>
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<title-group>
<article-title>Neutrophilic Dermatosis Confined to the Lymphedematous Area</article-title>
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<contrib-group>
<contrib contrib-type="author">
<name>
<surname>Park</surname>
<given-names>Ji-Youn</given-names>
</name>
<xref ref-type="aff" rid="A1"></xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Kang</surname>
<given-names>Hee Young</given-names>
</name>
<xref ref-type="aff" rid="A1"></xref>
</contrib>
<contrib contrib-type="author" corresp="yes">
<name>
<surname>Kim</surname>
<given-names>You Chan</given-names>
</name>
<xref ref-type="aff" rid="A1"></xref>
</contrib>
</contrib-group>
<aff id="A1">Department of Dermatology, Ajou University School of Medicine, Suwon, Korea.</aff>
<author-notes>
<corresp>Corresponding author: You Chan Kim, Department of Dermatology, Ajou University School of Medicine, 206 WorldCup-ro, Yeongtong-gu, Suwon 443-749, Korea. Tel: 82-31-219-5190, Fax: 82-31-219-5189,
<email>maychan@ajou.ac.kr</email>
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<pub-date pub-type="ppub">
<month>6</month>
<year>2014</year>
</pub-date>
<pub-date pub-type="epub">
<day>12</day>
<month>6</month>
<year>2014</year>
</pub-date>
<volume>26</volume>
<issue>3</issue>
<fpage>411</fpage>
<lpage>413</lpage>
<history>
<date date-type="received">
<day>04</day>
<month>6</month>
<year>2013</year>
</date>
<date date-type="accepted">
<day>14</day>
<month>6</month>
<year>2014</year>
</date>
</history>
<permissions>
<copyright-statement>Copyright © 2014 The Korean Dermatological Association and The Korean Society for Investigative Dermatology</copyright-statement>
<copyright-year>2014</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/">
<license-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" 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.</license-p>
</license>
</permissions>
</article-meta>
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<body>
<p>Dear Editor:</p>
<p>Lymphedema is a common sequelae after cancer surgery with lymph node dissection
<xref rid="B1" ref-type="bibr">1</xref>
. A lymphedematous limb, which is prone to the development of infections or tumors, suggests an alteration in regional immune competence
<xref rid="B2" ref-type="bibr">2</xref>
. Neutrophilic dermatosis on postmastectomy lymphedema (NDPL) is a newly suggested disease by Demitsu and Tadaki
<xref rid="B3" ref-type="bibr">3</xref>
It is also referred to as localized Sweet's syndrome (SS) because of the histological similarities between the two conditions
<xref rid="B4" ref-type="bibr">4</xref>
. Herein, we report additional cases of neutrophilic dermatosis confined within a lymphedematous site and a review of the disease entity.</p>
<p>All three cases involved female patients, two of whom were breast cancer patients who underwent modified radical mastectomy with axillary lymph node dissections (
<xref ref-type="table" rid="T1">Table 1</xref>
). The third patient had undergone radical hysterectomy with pelvic lymph node dissection for cervical cancer. Lymphedema was confined to the lymphnode-dissected limb developed in all three patients after the surgery. All of them rapidly developed erythematous rashes on their lymphedematous limb (
<xref ref-type="fig" rid="F1">Fig. 1A, B</xref>
). Skin biopsy revealed marked papillary dermal edema and dense dermal neutrophil infiltrates, consistent with the histopathologic features of SS (
<xref ref-type="fig" rid="F1">Fig. 1C, D</xref>
). The laboratory findings, including white blood cell count, percentage of neutrophils, erythrocyte sedimentation rate (ESR), and C-reactive protein (CRP), were within the normal ranges, except in case 3 (ESR: 35 mm/h; CRP: 4.09 mg/dl). Each case was treated with topical or oral corticosteroid, or oral antibiotics. In case 3, the lesion recurred rapidly after initially being treated with oral antibiotics. After the administration of oral corticosteroid, the lesion rapidly resolved within 1 week. On the basis of the characteristic distribution of the lesion and the histopathologic features, a diagnosis of 'neutrophilic dermatosis on the lymphedematous area' was made.</p>
<p>Neutrophilic dermatosis or SS localized on the area of lymphedema is rare, and only 11 cases have been reported
<xref rid="B1" ref-type="bibr">1</xref>
,
<xref rid="B3" ref-type="bibr">3</xref>
,
<xref rid="B4" ref-type="bibr">4</xref>
. The clinical presentations of previously reported cases were erythematous papules, plaques, and vesicles on the lymphedematous arm after a mastectomy, which are consistent with our cases
<xref rid="B1" ref-type="bibr">1</xref>
,
<xref rid="B3" ref-type="bibr">3</xref>
,
<xref rid="B4" ref-type="bibr">4</xref>
. The pathomechanism cannot be fully demonstrated; however, the vulnerability of the lymphedematous area seems to be the main factor
<xref rid="B1" ref-type="bibr">1</xref>
,
<xref rid="B2" ref-type="bibr">2</xref>
,
<xref rid="B5" ref-type="bibr">5</xref>
. The stasis of protein-rich lymphatic fluid contains numerous cytokines that might attract neutrophils and also result in the impairment of immune surveillance
<xref rid="B2" ref-type="bibr">2</xref>
,
<xref rid="B5" ref-type="bibr">5</xref>
. Because this dermatosis shows typical clinicohistopathological findings of SS, they are considered a localized variant of SS
<xref rid="B4" ref-type="bibr">4</xref>
. However, other systemic presentations, such as leukocytosis, neutrophilia, or fever, were less frequent than in typical SS. Therefore, several other reports suggested the use of the new term NDPL
<xref rid="B1" ref-type="bibr">1</xref>
,
<xref rid="B3" ref-type="bibr">3</xref>
. Other clinical differential diagnosis included cellulitis or erysipelas. Contrary to cellulitis or erysipelas, the lesion was confined only to the lymphedematous area and was well treated with oral or topical corticosteroid
<xref rid="B1" ref-type="bibr">1</xref>
,
<xref rid="B4" ref-type="bibr">4</xref>
. Although the 11 reports to date were cases of a lesion on a lymphedematous arm after a mastectomy, our report includes one case of a lesion that had developed on the lymphedematous leg after a hysterectomy for cervical cancer. Consequently, our cases suggest a novel point that a lesion could develop on any lymphedematous limb after lymph node dissection. Therefore, we suggest the term 'neutrophilic dermatosis on the lymphedematous area' rather than NDPL.</p>
</body>
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<floats-group>
<fig id="F1" orientation="portrait" position="float">
<label>Fig. 1</label>
<caption>
<p>(A) Case 1. Multiple variable-sized erythematous papules, vesicles, and plaques localized on the right lymphedematous arm. (B) Case 3. Multiple erythematous papules and patches confined on the left lymphedematous leg. (C, D) Histopathological findings. Skin biopsy taken from the patient of case 1, showing marked papillary dermal edema and dense dermal neutrophil infiltrates (H&E; C: ×40, D: ×200).</p>
</caption>
<graphic xlink:href="ad-26-411-g001"></graphic>
</fig>
<table-wrap id="T1" orientation="portrait" position="float">
<label>Table 1</label>
<caption>
<p>Clinical characteristics of the three cases</p>
</caption>
<graphic xlink:href="ad-26-411-i001"></graphic>
<table-wrap-foot>
<fn>
<p>F: female, LND: lymph node dissection, CRTx: chemoradiotherapy, CBC: complete blood cell count, WNL: within normal limit, ESR: erythrocyte sedimentation rate, CRP: C-reactive protein, ND: not done, (-): negative, (+): positive.</p>
</fn>
</table-wrap-foot>
</table-wrap>
</floats-group>
</pmc>
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