<?xml version="1.0" encoding="UTF-8"?><!DOCTYPE article PUBLIC "-//NLM//DTD JATS (Z39.96) Journal Publishing DTD v1.2 20190208//EN" "http://jats.nlm.nih.gov/publishing/1.2/JATS-journalpublishing1.dtd"><article xmlns:mml="http://www.w3.org/1998/Math/MathML" xmlns:xlink="http://www.w3.org/1999/xlink" article-type="case-report" dtd-version="1.2" xml:lang="en">
    <front>
        <journal-meta>
            <journal-id journal-id-type="pmc">F1000Research</journal-id>
            <journal-title-group>
                <journal-title>F1000Research</journal-title>
            </journal-title-group>
            <issn pub-type="epub">2046-1402</issn>
            <publisher>
                <publisher-name>F1000 Research Limited</publisher-name>
                <publisher-loc>London, UK</publisher-loc>
            </publisher>
        </journal-meta>
        <article-meta>
            <article-id pub-id-type="doi">10.12688/f1000research.147343.1</article-id>
            <article-categories>
                <subj-group subj-group-type="heading">
                    <subject>Case Report</subject>
                </subj-group>
                <subj-group>
                    <subject>Articles</subject>
                </subj-group>
            </article-categories>
            <title-group>
                <article-title>Case Report: A case of metastasis to branchial cleft cyst from papillary thyroid cancer</article-title>
                <fn-group content-type="pub-status">
                    <fn>
                        <p>[version 1; peer review: 1 approved with reservations]</p>
                    </fn>
                </fn-group>
            </title-group>
            <contrib-group>
                <contrib contrib-type="author" corresp="yes">
                    <name>
                        <surname>Shelar</surname>
                        <given-names>Sheetal</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Funding Acquisition</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Software</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="corresp" rid="c1">a</xref>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Gupta</surname>
                        <given-names>Roohi</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Funding Acquisition</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Software</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Dhande</surname>
                        <given-names>Rajasbala</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Funding Acquisition</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Software</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Parihar</surname>
                        <given-names>P. H.</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Funding Acquisition</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Software</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Validation</role>
                    <role content-type="http://credit.niso.org/">Visualization</role>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <aff id="a1">
                    <label>1</label>RADIODIAGNOSIS, JAWAHARLAL NEHRU MEDICAL COLLEGE, WARDHA, MAHARASHTRA, 442001, India</aff>
            </contrib-group>
            <author-notes>
                <corresp id="c1">
                    <label>a</label>
                    <email xlink:href="mailto:sheetal.shelar@dmiher.edu.in">sheetal.shelar@dmiher.edu.in</email>
                </corresp>
                <fn fn-type="conflict">
                    <p>No competing interests were disclosed.</p>
                </fn>
            </author-notes>
            <pub-date pub-type="epub">
                <day>6</day>
                <month>6</month>
                <year>2024</year>
            </pub-date>
            <pub-date pub-type="collection">
                <year>2024</year>
            </pub-date>
            <volume>13</volume>
            <elocation-id>586</elocation-id>
            <history>
                <date date-type="accepted">
                    <day>14</day>
                    <month>5</month>
                    <year>2024</year>
                </date>
            </history>
            <permissions>
                <copyright-statement>Copyright: &#x00a9; 2024 Shelar S et al.</copyright-statement>
                <copyright-year>2024</copyright-year>
                <license xlink:href="https://creativecommons.org/licenses/by/4.0/">
                    <license-p>This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
                </license>
            </permissions>
            <self-uri content-type="pdf" xlink:href="https://f1000research.com/articles/13-586/pdf"/>
            <abstract>
                <p>Branchial cleft cysts are frequently encountered congenital anomalies, arising from the first to fourth pharyngeal clefts and second branchial cleft anomalies are the most common. These anomalies, even though present from birth, become symptomatic only later in life. Majority of them are benign in nature. However, these cysts can get secondarily infected or can harbour secondary metastases and sometimes even primary malignancy in very rare cases. Here we discuss a case of a middle-aged female presenting with a gradually increasing branchial cleft cyst with incidental thyroid lesion on ultrasonography, later proven to be papillary thyroid carcinoma with metastatic spread to the brachial cleft cyst.</p>
            </abstract>
            <kwd-group kwd-group-type="author">
                <kwd>Branchial cleft cyst</kwd>
                <kwd>ultrasonography</kwd>
                <kwd>thyroid gland</kwd>
                <kwd>papillary carcinoma</kwd>
                <kwd>metastases</kwd>
            </kwd-group>
            <funding-group>
                <funding-statement>The author(s) declared that no grants were involved in supporting this work.</funding-statement>
            </funding-group>
        </article-meta>
    </front>
    <body>
        <sec id="sec1" sec-type="intro">
            <title>Introduction</title>
            <p>Incomplete obliteration of the first four pharyngeal arches gives rise to branchial cleft anomalies in the form of cysts, sinus or fistulae formation depending on the degree of obliteration during embryonic development.
                <sup>
                    <xref ref-type="bibr" rid="ref1">1</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref2">2</xref>
                </sup> The most common branchial cleft anomaly is the branchial cleft cyst arising from the second pharyngeal cleft.
                <sup>
                    <xref ref-type="bibr" rid="ref1">1</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref3">3</xref>
                </sup> Their most common location is below the mandible just anterior to sternocleidomastoid but can occur at any location along the path of second branchial apparatus.
                <sup>
                    <xref ref-type="bibr" rid="ref4">4</xref>
                </sup> Since most of them are benign, their presentation to a clinician is usually when it increases in size or post-infection when it becomes tender with or without surrounding inflammatory skin changes.
                <sup>
                    <xref ref-type="bibr" rid="ref5">5</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref6">6</xref>
                </sup> Investigating a symptomatic branchial cleft cyst becomes important to rule out neoplastic etiology and for early management.</p>
        </sec>
        <sec id="sec2">
            <title>Case Presentation</title>
            <p>A 35-year-old female presented to the surgery OPD with a 2-3 cm swelling on the left side of the neck along the middle third of sternocleidomastoid muscle (
                <xref ref-type="fig" rid="f1">Figure 1</xref>). The patient said that the swelling was present since many years. However, in the past three months there was gradual increase in the size of the swelling. It was round to oval in shape, soft in consistency, non-tender and freely movable over the underlying muscle. There was no evidence of adjacent skin changes or any other neck swelling on physical examination.</p>
            <fig fig-type="figure" id="f1" orientation="portrait" position="float">
                <label>Figure 1. </label>
                <caption>
                    <title>Clinical image of the patient with a localized left sided anterior neck swelling.</title>
                </caption>
                <graphic id="gr1" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/161530/8e6ad066-fe4a-4f75-ba12-83521e0a0988_figure1.gif"/>
            </fig>
            <p>The patient had no complains of difficulty in breathing or deglutition and no restriction in movement of the neck. She had no history of trauma, fever or any event of tuberculosis. Her routine blood investigations as well as thyroid profile was within normal ranges.</p>
            <p>On ultrasound of the neck, the lesion measured 2 x 1.5 cm, was thin walled solid-cystic (cystic component&gt; solid) with echogenic debris and abutting sternocleidomastoid muscle (
                <xref ref-type="fig" rid="f2">Figure 2a</xref>). The solid component showed internal vascularity on color doppler (
                <xref ref-type="fig" rid="f2">Figure 2b</xref>).</p>
            <fig fig-type="figure" id="f2" orientation="portrait" position="float">
                <label>Figure 2. </label>
                <caption>
                    <title>Grey scale ultrasound images of the swelling showing the solid cystic nature of the lesion (a) and evident vascularity within the solid component on color doppler (b).</title>
                </caption>
                <graphic id="gr2" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/161530/8e6ad066-fe4a-4f75-ba12-83521e0a0988_figure2.gif"/>
            </fig>
            <p>Thyroid gland was further examined on ultrasonography and appeared normal in maximum dimensions. The isthmus and right lobe were normal in echotexture (
                <xref ref-type="fig" rid="f3">Figure 3</xref>).</p>
            <fig fig-type="figure" id="f3" orientation="portrait" position="float">
                <label>Figure 3. </label>
                <caption>
                    <title>Grey scale ultrasound images with linear probe showing normal isthmus (a) and right lobe (b) of the thyroid gland.</title>
                </caption>
                <graphic id="gr3" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/161530/8e6ad066-fe4a-4f75-ba12-83521e0a0988_figure3.gif"/>
            </fig>
            <p>However, the left lobe of thyroid revealed an ill-defined round (1 x 1 cm) hypoechoic solid lesion with punctate microcalcifications and increased vascularity within (
                <xref ref-type="fig" rid="f4">Figure 4a</xref>). Rest of the left lobe appeared normal in echotexture as well as vascularity (
                <xref ref-type="fig" rid="f4">Figure 4b</xref>). The lesion was graded BIRADS-4 on ultrasound indicating moderate suspicion for malignancy.</p>
            <fig fig-type="figure" id="f4" orientation="portrait" position="float">
                <label>Figure 4. </label>
                <caption>
                    <title>Grey scale ultrasound images of the left lobe of thyroid showing a hypoechoic lesion with punctate microcalcifications (a) with peripheral vascularity in color doppler (b).</title>
                </caption>
                <graphic id="gr4" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/161530/8e6ad066-fe4a-4f75-ba12-83521e0a0988_figure4.gif"/>
            </fig>
            <p>The patient was then further evaluated on computed tomography before subjecting the patient to FNAC (fine needle aspiration cytology) of the neck lesion. On contrast enhanced CT of the neck, there is a well-defined round solid-cystic lesion lying posterior to middle one-third of the left sternocleidomastoid muscle and focally abutting the left jugular vein. The lesion has thin enhancing wall and enhancing internal solid component (
                <xref ref-type="fig" rid="f5">Figure 5</xref>).</p>
            <fig fig-type="figure" id="f5" orientation="portrait" position="float">
                <label>Figure 5. </label>
                <caption>
                    <title>Contrast CT axial (a), coronal (b) and sagittal (c) images showing a well-defined cystic lesion with thin wall and enhancing solid component.</title>
                </caption>
                <graphic id="gr5" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/161530/8e6ad066-fe4a-4f75-ba12-83521e0a0988_figure5.gif"/>
            </fig>
            <p>After these radiological investigations, the patient was then taken for FNAC of the swelling. The first sample that was fluid aspirate came out to be indeterminate in nature. However, in the next sampling, the solid component was targeted and it came out to be positive for malignant cytology. Since the patient had concurrent BIRADS-4 thyroid lesion, there was suspicion of thyroid malignancy with metastasis to the branchial cleft cyst. The thyroid lesion was then taken up for aspiration and was proven to be papillary thyroid carcinoma.</p>
        </sec>
        <sec id="sec3" sec-type="discussion">
            <title>Discussion</title>
            <p>Branchial cleft cysts are one of the most commonly encountered lateral neck masses.
                <sup>
                    <xref ref-type="bibr" rid="ref6">6</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref7">7</xref>
                </sup> These are embryological remnants that occur due of failure of closure of the pharyngeal arches before birth.
                <sup>
                    <xref ref-type="bibr" rid="ref8">8</xref>
                </sup> Metastases to the branchial cleft cyst is rare and identifying, differentiating it from cystic metastatic lymph nodes is important to plan treatment.
                <sup>
                    <xref ref-type="bibr" rid="ref9">9</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref10">10</xref>
                </sup> Papillary thyroid carcinoma frequently metastasizes and therefore it is necessary to evaluate the thyroid gland in such instances to narrow down the diagnoses.
                <sup>
                    <xref ref-type="bibr" rid="ref11">11</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref12">12</xref>
                </sup>
            </p>
        </sec>
        <sec id="sec4">
            <title>Consent</title>
            <p>Written informed consent for publication of their clinical details and clinical images was obtained from the patient.</p>
        </sec>
    </body>
    <back>
        <sec id="sec7" sec-type="data-availability">
            <title>Data Availability</title>
            <p>No data associated with this article.</p>
        </sec>
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    </back>
    <sub-article article-type="reviewer-report" id="report307444">
        <front-stub>
            <article-id pub-id-type="doi">10.5256/f1000research.161530.r307444</article-id>
            <title-group>
                <article-title>Reviewer response for version 1</article-title>
            </title-group>
            <contrib-group>
                <contrib contrib-type="author">
                    <name>
                        <surname>Salahuddin</surname>
                        <given-names>Nor Azirah</given-names>
                    </name>
                    <xref ref-type="aff" rid="r307444a1">1</xref>
                    <role>Referee</role>
                </contrib>
                <aff id="r307444a1">
                    <label>1</label>Universiti Sains Islam Malaysia, Nilai, Malaysia</aff>
            </contrib-group>
            <author-notes>
                <fn fn-type="conflict">
                    <p>
                        <bold>Competing interests: </bold>No competing interests were disclosed.</p>
                </fn>
            </author-notes>
            <pub-date pub-type="epub">
                <day>12</day>
                <month>8</month>
                <year>2024</year>
            </pub-date>
            <permissions>
                <copyright-statement>Copyright: &#x00a9; 2024 Salahuddin NA</copyright-statement>
                <copyright-year>2024</copyright-year>
                <license xlink:href="https://creativecommons.org/licenses/by/4.0/">
                    <license-p>This is an open access peer review report distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
                </license>
            </permissions>
            <related-article ext-link-type="doi" id="relatedArticleReport307444" related-article-type="peer-reviewed-article" xlink:href="10.12688/f1000research.147343.1"/>
            <custom-meta-group>
                <custom-meta>
                    <meta-name>recommendation</meta-name>
                    <meta-value>approve-with-reservations</meta-value>
                </custom-meta>
            </custom-meta-group>
        </front-stub>
        <body>
            <p>Major comment 
                <list list-type="order">
                    <list-item>
                        <p>The discussion is too short. As we know branchial cyst can represent primary malignancy or secondary mets. How the author differentiates the initial diagnosis of branchial&#x00a0;cystic vs malignant cystic swelling/secondary metastasis cystic neck swelling (ref 1). Is there any cytology diagnosis which suggests the swelling is a branchial cyst e,g cholesterol crystal. Suggest to elaborate the discussion how this case is differ from reported cases and ass learning points to others so that it will be more valuable for the indexing.&#x00a0;</p>
                    </list-item>
                </list> &#x00a0; &#x00a0; &#x00a0; &#x00a0; &#x00a0; &#x00a0; Ref: Mc Loughlin L, Elsafty N, Kavanagh F, Gillanders S, et al.: AB087. 192. Branchial cleft cyst&#x2014;&#x00a0; really?. 
                <italic>Mesentery and Peritoneum</italic>. 2018; 2.&#x00a0;</p>
            <p> </p>
            <p> Minor comment 
                <list list-type="order">
                    <list-item>
                        <p>The case presentation was presented well but the author did not highlight the management and outcome of the case. We would like to know further the management eg surgical intervention, oncology treatment etc</p>
                    </list-item>
                    <list-item>
                        <p>The second FNAC was done to target the solid component. Is the repeated FNAC done under ultrasound guided?</p>
                    </list-item>
                    <list-item>
                        <p>References -some of the references cited was too old (&gt;20 years).</p>
                    </list-item>
                </list>
            </p>
            <p>Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?</p>
            <p>Partly</p>
            <p>Is the case presented with sufficient detail to be useful for other practitioners?</p>
            <p>Partly</p>
            <p>Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?</p>
            <p>No</p>
            <p>Is the background of the case&#x2019;s history and progression described in sufficient detail?</p>
            <p>Yes</p>
            <p>Reviewer Expertise:</p>
            <p>Otorhinolaryngology, head and neck surgery</p>
            <p>I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above.</p>
        </body>
        <back>
            <ref-list>
                <title>References</title>
                <ref id="rep-ref-307444-1">
                    <label>1</label>
                    <mixed-citation publication-type="journal">
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                        <article-title>AB087. 192. Branchial cleft cyst&#x2014;really?</article-title>.
                        <source>
                            <italic>Mesentery and Peritoneum</italic>
                        </source>.<year>2018</year>;<volume>2</volume>:
                        <elocation-id>10.21037/map.2018.AB087</elocation-id>
                        <pub-id pub-id-type="doi">10.21037/map.2018.AB087</pub-id>
                    </mixed-citation>
                </ref>
            </ref-list>
        </back>
    </sub-article>
</article>
