<?xml version="1.0" encoding="utf-8"?>
<!DOCTYPE article PUBLIC "-//NLM//DTD JATS (Z39.96) Journal Publishing DTD v1.0 20120330//EN" "JATS-journalpublishing1.dtd">
<article article-type="case-report" dtd-version="1.0" xml:lang="en" xmlns:mml="http://www.w3.org/1998/Math/MathML" xmlns:xlink="http://www.w3.org/1999/xlink" xmlns:xsi="http://www.w3.org/2001/XMLSchema-instance">
<front>
<journal-meta>
<journal-id journal-id-type="publisher-id">JKOS</journal-id>
<journal-title-group>
<journal-title>Journal of the Korean Ophthalmological Society</journal-title>
<abbrev-journal-title>J Korean Ophthalmol Soc</abbrev-journal-title>
</journal-title-group>
<issn pub-type="ppub">0378-6471</issn>
<issn pub-type="epub">2092-9374</issn>
<publisher>
<publisher-name>Korean Ophthalmological Society</publisher-name>
</publisher>
</journal-meta>
<article-meta>
<article-id pub-id-type="doi">10.3341/jkos.2018.59.9.876</article-id>
<article-id pub-id-type="publisher-id">jkos-59-876</article-id>
<article-categories>
<subj-group>
<subject>Case Report</subject>
</subj-group>
</article-categories>
<title-group>
<article-title>Bilateral Frosted Branch Angiitis in Kikuchi-Fujimoto Disease</article-title>
</title-group>
<contrib-group>
<contrib contrib-type="author">
<name name-style="western" xml:lang="en"><surname>Kim</surname><given-names>Hun Min</given-names></name>
<degrees>MD</degrees>
<xref ref-type="aff" rid="aff1-jkos-59-876"><sup>1</sup></xref>
</contrib>
<contrib contrib-type="author">
<name name-style="western" xml:lang="en"><surname>Choi</surname><given-names>Young Je</given-names></name>
<degrees>MD</degrees>
<xref ref-type="aff" rid="aff2-jkos-59-876"><sup>2</sup></xref>
</contrib>
<contrib contrib-type="author">
<name name-style="western" xml:lang="en"><surname>Kim</surname><given-names>Seong Taeck</given-names></name>
<degrees>MD, PhD</degrees>
<xref ref-type="aff" rid="aff1-jkos-59-876"><sup>1</sup></xref>
<xref ref-type="corresp" rid="c1-jkos-59-876"/>
</contrib>
<aff id="aff1-jkos-59-876"><label>1</label>Department of Ophthalmology, Chosun University School of Medicine, Gwangju, <country>Korea</country></aff>
<aff id="aff2-jkos-59-876"><label>2</label>Gangjin Medical Center, Sinan, <country>Korea</country></aff>
</contrib-group>
<author-notes>
<corresp id="c1-jkos-59-876">Address reprint requests to <bold>Seong Taeck Kim, MD, PhD</bold> Department of Ophthalmology, Chosun University Hospital, &#x0023;365 Pilmun-daero, Dong-gu, Gwangju 61453, Korea Tel: 82-62-220-3190, Fax: 82-62-225-9839 E-mail: <email>s20age@hanmail.net</email></corresp>
</author-notes>
<pub-date pub-type="ppub">
<month>09</month>
<year>2018</year>
</pub-date>
<pub-date pub-type="epub">
<day>25</day>
<month>09</month>
<year>2018</year>
</pub-date>
<volume>59</volume><issue>9</issue>
<fpage>876</fpage>
<lpage>880</lpage>
<history>
<date date-type="received"><day>17</day><month>05</month><year>2018</year></date>
<date date-type="rev-recd"><day>22</day><month>06</month><year>2018</year></date>
<date date-type="accepted"><day>28</day><month>08</month><year>2018</year></date>
</history>
<permissions>
<copyright-statement>Copyright &#x00A9; 2018 Korean Ophthalmological Society</copyright-statement>
<copyright-year>2018</copyright-year>
<license><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>
<abstract xml:lang="en">
<title>Abstract</title>
<sec><title>Purpose</title>
<p>A case of frosted branch angiitis in Kikuchi-Fujimoto disease is reported.</p>
</sec>
<sec><title>Case summary</title>
<p>A 33-year-old male complained of a sudden decrease in visual acuity that developed in both eyes 5 days prior. He suffered from a headache, chills, myalgia, and flank pain 1 week before. The initial best-corrected visual acuity (BCVA) was 0.1 in the right eye and 0.2 in the left eye. On slit lamp examination, no inflammatory finding was observed in the anterior chamber and vitreous body of both eyes. On fundus examination, a diffuse vascular sheathing-like frosted branch was found in the retinal vessels, and retinal hemorrhage was observed. Fluorescein angiography showed staining and leakage of dye along the vascular sheathing. Serological findings were negative, showing no evidence of an autoimmune disease or viral infection. Neck ultrasonography revealed non-tender left cervical lymph node enlargement &#x003E;1 cm in diameter. Ultrasound-guided fine needle aspiration cytology showed findings compatible with Kikuchi-Fujimoto disease, including necrotic changes and pronounced karyorrhexis, plus histiocyte and lymphocyte infiltration without neutrophils. We started systemic steroid therapy. One month after treatment, the BCVA of both eyes improved to 1.0.</p>
</sec>
<sec><title>Conclusions</title>
<p>In patients with frosted branch angiitis, systemic disease such as Kikuchi-Fujimoto disease should be considered.</p>
</sec>
</abstract>
<kwd-group xml:lang="en">
<kwd>Frosted branch angiitis</kwd>
<kwd>Kikuchi-Fujimoto disease</kwd>
<kwd>Necrotizing lymphadenitis</kwd>
</kwd-group>
</article-meta>
</front>
<back>
<ref-list>
<title>References</title>
<ref id="b1-jkos-59-876"><label>1</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Norris</surname> <given-names>AH</given-names></name> <name><surname>Krasinskas</surname> <given-names>AM</given-names></name> <name><surname>Salhany</surname> <given-names>KE</given-names></name> <name><surname>Gluckmman</surname> <given-names>SJ</given-names></name></person-group> <article-title>KikuchiFujimoto disease: a benign cause of fever and lymphadenopathy</article-title> <source>Am J Med</source> <year>1996</year> <volume>101</volume><fpage>401</fpage><lpage>5</lpage></element-citation></ref>
<ref id="b2-jkos-59-876"><label>2</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Rocher</surname> <given-names>F</given-names></name> <name><surname>Pelosse</surname> <given-names>B</given-names></name> <name><surname>Momtchilova</surname> <given-names>M</given-names></name> <name><surname>Laroche</surname> <given-names>L</given-names></name></person-group> <article-title>Kikuchi&#x0027;s abdominal and ocular manifestation</article-title> <source>J Fr Ophtalmal</source> <year>2006</year> <volume>29</volume><fpage>932</fpage><lpage>6</lpage></element-citation></ref>
<ref id="b3-jkos-59-876"><label>3</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Ito</surname> <given-names>Y</given-names></name></person-group> <article-title>Frosted branch angiitis in a child</article-title> <source>Jpn J Clin Ophthalmol</source> <year>1976</year> <volume>30</volume><fpage>797</fpage><lpage>803</lpage></element-citation></ref>
<ref id="b4-jkos-59-876"><label>4</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Bosch</surname> <given-names>X</given-names></name> <name><surname>Guilabert</surname> <given-names>A</given-names></name> <name><surname>Miquel</surname> <given-names>R</given-names></name> <etal/></person-group> <article-title>Enigmatic Kikuchi-Fujimoto disease: a comprehensive review</article-title> <source>Am J Clin Pathol</source> <year>2004</year> <volume>122</volume><fpage>141</fpage><lpage>52</lpage></element-citation></ref>
<ref id="b5-jkos-59-876"><label>5</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Famularo</surname> <given-names>G</given-names></name> <name><surname>Giustiniani</surname> <given-names>MC</given-names></name> <name><surname>Marasco</surname> <given-names>A</given-names></name> <etal/></person-group> <article-title>De Simone C. Kikuchi Fujimoto lymphadenitis: case report and literature review</article-title> <source>AM J Hematol</source> <year>2003</year> <volume>74</volume><fpage>60</fpage><lpage>3</lpage></element-citation></ref>
<ref id="b6-jkos-59-876"><label>6</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Cheng</surname> <given-names>CY</given-names></name> <name><surname>Sheng</surname> <given-names>WH</given-names></name> <name><surname>Lo</surname> <given-names>YC</given-names></name> <etal/></person-group> <article-title>Clinical presentations, abdominal results and outcomes of patients with Kikuchi&#x0027;s disease: abdominal on the association between recurrent Kikuchi&#x0027;s disease and autoimmune diseases</article-title> <source>J Microbiol Immunol Infect</source> <year>2010</year> <volume>43</volume><fpage>366</fpage><lpage>71</lpage></element-citation></ref>
<ref id="b7-jkos-59-876"><label>7</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Kim</surname> <given-names>SH</given-names></name> <name><surname>Kim</surname> <given-names>SJ</given-names></name> <name><surname>Chung</surname> <given-names>H</given-names></name> <etal/></person-group> <article-title>Bilateral anterior uveitis as an abdominal manifestation of Kikuchi-Fujimoto disease</article-title> <source>Rheumatology (Oxford)</source> <year>2004</year> <volume>43</volume><fpage>1056</fpage><lpage>7</lpage></element-citation></ref>
<ref id="b8-jkos-59-876"><label>8</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Taguri</surname> <given-names>AH</given-names></name> <name><surname>Mcllwaine</surname> <given-names>GG</given-names></name></person-group> <article-title>Bilateral panuveitis: a possible abdominal with Kikuchi&#x2013;Fujimoto disease</article-title> <source>Am J Ophthalmol</source> <year>2001</year> <volume>132</volume><fpage>419</fpage><lpage>21</lpage></element-citation></ref>
<ref id="b9-jkos-59-876"><label>9</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Zou</surname> <given-names>W</given-names></name> <name><surname>Wen</surname> <given-names>F</given-names></name></person-group> <article-title>Bilateral occlusive retinal vasculitis in Kikuchi-Fujumoto disease</article-title> <source>Clin Exp Ophthalmol</source> <year>2007</year> <volume>35</volume><fpage>875</fpage><lpage>7</lpage></element-citation></ref>
<ref id="b10-jkos-59-876"><label>10</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Galor</surname> <given-names>A</given-names></name> <name><surname>Georgy</surname> <given-names>M</given-names></name> <name><surname>Leder</surname> <given-names>HA</given-names></name> <etal/></person-group> <article-title>Papillary conjunctivitis abdominal with Kikuchi disease</article-title> <source>Cornea</source> <year>2008</year> <volume>27</volume><fpage>944</fpage><lpage>6</lpage></element-citation></ref>
<ref id="b11-jkos-59-876"><label>11</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Biswas</surname> <given-names>J</given-names></name> <name><surname>Fogla</surname> <given-names>R</given-names></name> <name><surname>Madhavan</surname> <given-names>HN</given-names></name></person-group> <article-title>Bilateral frosted branchangiitis in an 8-year-old indian girl</article-title> <source>Retina</source> <year>1996</year> <volume>16</volume><fpage>444</fpage><lpage>5</lpage></element-citation></ref>
<ref id="b12-jkos-59-876"><label>12</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Masuda</surname> <given-names>K</given-names></name> <name><surname>Ueno</surname> <given-names>M</given-names></name> <name><surname>Watanabe</surname> <given-names>I</given-names></name></person-group> <article-title>A case of frosted branch angiitis with yellowish-white placoid lesions: fluorescein and indocyanine green angiography findings</article-title> <source>Jpn J Ophthalmol</source> <year>1998</year> <volume>42</volume><fpage>484</fpage><lpage>9</lpage></element-citation></ref>
<ref id="b13-jkos-59-876"><label>13</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Duker</surname> <given-names>JS</given-names></name> <name><surname>Brown</surname> <given-names>GC</given-names></name> <name><surname>Brooks</surname> <given-names>L</given-names></name></person-group> <article-title>Retinal vasculitis in Crohn&#x0027;s disease</article-title> <source>Am J Ophthalmol</source> <year>1987</year> <volume>103</volume><fpage>664</fpage><lpage>8</lpage></element-citation></ref>
<ref id="b14-jkos-59-876"><label>14</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Dorfman</surname> <given-names>RF</given-names></name> <name><surname>Berry</surname> <given-names>GJ</given-names></name></person-group> <article-title>Kikuchi&#x0027;s histiocytic necrotizing lympha-denitis: an analysis of 108 cases with emphasis of differential diagnosis</article-title> <source>Semin Diagn Pathol</source> <year>1988</year> <volume>5</volume><fpage>329</fpage><lpage>45</lpage></element-citation></ref>
<ref id="b15-jkos-59-876"><label>15</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Imamura</surname> <given-names>M</given-names></name> <name><surname>Ueno</surname> <given-names>H</given-names></name> <name><surname>Matsuura</surname> <given-names>A</given-names></name> <etal/></person-group> <article-title>An ultrastructural study of subacute necrotizing lymphadenitis</article-title> <source>Am J Pathol</source> <year>1982</year> <volume>107</volume><fpage>292</fpage><lpage>9</lpage></element-citation></ref>
</ref-list>
<sec sec-type="display-objects">
<title>Figures</title>
<fig id="f1-jkos-59-876" position="anchor">
<label>Figure 1.</label>
<caption xml:lang="en"><p>Fundus exam at the initial visit. Fundus photograph shows sheathing of retinal vessels and multiple retinal hemorrhages in the right (A) and the left (B) eye. Fundus fluorescein angiography at the initial examination shows dye leakages from the sheathed retinal vessels with normal blood flow in the right (C) and the left (D) eye.</p></caption>
<graphic xlink:href="jkos-59-876f1.tif"/>
</fig>
<fig id="f2-jkos-59-876" position="anchor">
<label>Figure 2.</label>
<caption xml:lang="en"><p>Histopathological finding of the cervical lymph node. Numerous phagocytic histiocytes and prominent karyorrhexis are identified (hematoxylin and eosin stain [H&#x0026;E] stain, &#x00D7;100).</p></caption>
<graphic xlink:href="jkos-59-876f2.tif"/>
</fig>
<fig id="f3-jkos-59-876" position="anchor">
<label>Figure 3.</label>
<caption xml:lang="en"><p>Fundus photographs at 1 month after systemic steroid therapy. Fundus photograph shows almost normal without any sheathing of retinal vessels in the right (A) and the left (B) eye.</p></caption>
<graphic xlink:href="jkos-59-876f3.tif"/>
</fig>
</sec>
</back>
</article>
