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  <front>
    <journal-meta>
      <journal-id journal-id-type="publisher-id">87</journal-id>
      <journal-id journal-id-type="index">urn:lsid:arphahub.com:pub:A116C711-4C18-5A38-8F1E-5E97753A8A64</journal-id>
      <journal-title-group>
        <journal-title xml:lang="en">Folia Medica</journal-title>
        <abbrev-journal-title xml:lang="en">FM</abbrev-journal-title>
      </journal-title-group>
      <issn pub-type="ppub">0204-8043</issn>
      <issn pub-type="epub">1314-2143</issn>
      <publisher>
        <publisher-name>Plovdiv Medical University</publisher-name>
      </publisher>
    </journal-meta>
    <article-meta>
      <article-id pub-id-type="doi">10.3897/folmed.65.e68256</article-id>
      <article-id pub-id-type="publisher-id">68256</article-id>
      <article-categories>
        <subj-group subj-group-type="heading">
          <subject>Case Report</subject>
        </subj-group>
        <subj-group subj-group-type="scientific_subject">
          <subject>Oncology</subject>
          <subject>Pediatrics &amp; Genetic diseases</subject>
          <subject>Surgery &amp; Invasive treatment</subject>
        </subj-group>
      </article-categories>
      <title-group>
        <article-title>Giant cell tumor of the patellar tendon sheath in childhood: case report</article-title>
      </title-group>
      <contrib-group content-type="authors">
        <contrib contrib-type="author" corresp="yes">
          <name name-style="western">
            <surname>Skarentzos</surname>
            <given-names>Konstantinos</given-names>
          </name>
          <email xlink:type="simple">k.skarentzos@gmail.com</email>
          <uri content-type="orcid">https://orcid.org/0000-0003-0994-4959</uri>
          <xref ref-type="aff" rid="A1">1</xref>
        </contrib>
        <contrib contrib-type="author" corresp="no">
          <name name-style="western">
            <surname>Panteli</surname>
            <given-names>Dimitra</given-names>
          </name>
          <xref ref-type="aff" rid="A1">1</xref>
        </contrib>
        <contrib contrib-type="author" corresp="no">
          <name name-style="western">
            <surname>Moustafa</surname>
            <given-names>Reichan Molla</given-names>
          </name>
          <xref ref-type="aff" rid="A1">1</xref>
        </contrib>
        <contrib contrib-type="author" corresp="no">
          <name name-style="western">
            <surname>Tottas</surname>
            <given-names>Stylianos</given-names>
          </name>
          <xref ref-type="aff" rid="A1">1</xref>
        </contrib>
        <contrib contrib-type="author" corresp="no">
          <name name-style="western">
            <surname>Kougioumtzis</surname>
            <given-names>Ioannis E.</given-names>
          </name>
          <uri content-type="orcid">https://orcid.org/0000-0003-2022-2818</uri>
          <xref ref-type="aff" rid="A1">1</xref>
        </contrib>
        <contrib contrib-type="author" corresp="no">
          <name name-style="western">
            <surname>Drosos</surname>
            <given-names>Georgios I.</given-names>
          </name>
          <xref ref-type="aff" rid="A1">1</xref>
        </contrib>
      </contrib-group>
      <aff id="A1">
        <label>1</label>
        <addr-line content-type="verbatim">Department of Medicine, Democritus University of Thrace, Alexandroupolis, Greece</addr-line>
        <institution>Democritus University of Thrace</institution>
        <addr-line content-type="city">Alexandroupolis</addr-line>
        <country>Greece</country>
      </aff>
      <aff id="A2">
        <label>2</label>
        <addr-line content-type="verbatim">Department of Orthopaedic and Trauma Surgery, Democritus University of Thrace, University General Hospital of Alexandroupolis, Alexandroupolis, Greece</addr-line>
        <institution>Democritus University of Thrace</institution>
        <addr-line content-type="city">Alexandroupolis</addr-line>
        <country>Greece</country>
      </aff>
      <author-notes>
        <fn fn-type="corresp">
          <p>Corresponding author: Konstantinos Skarentzos, Department of Medicine, Democritus University of Thrace, Alexandroupolis, Greece; Email: <email xlink:type="simple">k.skarentzos@gmail.com</email></p>
        </fn>
      </author-notes>
      <pub-date pub-type="collection">
        <year>2023</year>
      </pub-date>
      <pub-date pub-type="epub">
        <day>30</day>
        <month>04</month>
        <year>2023</year>
      </pub-date>
      <volume>65</volume>
      <issue>2</issue>
      <fpage>301</fpage>
      <lpage>304</lpage>
      <uri content-type="arpha" xlink:href="http://openbiodiv.net/E945FC2D-2D0C-59E9-85C3-C788D8EDEB04">E945FC2D-2D0C-59E9-85C3-C788D8EDEB04</uri>
      <history>
        <date date-type="received">
          <day>04</day>
          <month>05</month>
          <year>2021</year>
        </date>
        <date date-type="accepted">
          <day>07</day>
          <month>10</month>
          <year>2022</year>
        </date>
      </history>
      <permissions>
        <copyright-statement>Konstantinos Skarentzos, Dimitra Panteli, Reichan Molla Moustafa, Stylianos Tottas, Ioannis E. Kougioumtzis, Georgios I. Drosos</copyright-statement>
        <license license-type="creative-commons-attribution" xlink:href="http://creativecommons.org/licenses/by/4.0/" xlink:type="simple">
          <license-p>This is an open access article distributed under the terms of the Creative Commons Attribution License (CC BY 4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.</license-p>
        </license>
      </permissions>
      <abstract>
        <label>Abstract</label>
        <p>﻿The aim of this study was to report a rare case of a giant cell tumor of the patellar tendon sheath. This indicates the diagnostic procedures and treatment options for giant cell tumors of the patellar tendon. This study reported a case of a 13-year-old male patient with a giant cell tumor of the tendon sheath. In our case, open arthrotomy was performed with complete surgical excision of the lesion. Histopathological examination revealed a giant cell tumor. At the last follow-up, 2 years after surgery, no complications were reported. The giant cell tumor of the patellar tendon sheath is an uncommon benign tumor. It mimics common knee symptoms. A differential diagnosis is definitely a challenge. Available operation approaches have demonstrated similar results, which lead to symptom relief and a low recurrence rate.</p>
      </abstract>
      <kwd-group>
        <label>Keywords</label>
        <kwd>arthroscopy</kwd>
        <kwd>child</kwd>
        <kwd>open arthrotomy</kwd>
      </kwd-group>
    </article-meta>
    <notes>
      <sec sec-type="Citation" id="SECID0ERE">
        <title>Citation</title>
        <p>Skarentzos K, Panteli D, Moustafa RM, Tottas S, Kougioumtzis IE, Drosos GI. Giant cell tumor of the patellar tendon sheath in childhood: case report. Folia Med (Plovdiv) 2023;65(2):301-304. doi: <ext-link xlink:type="simple" ext-link-type="doi" xlink:href="10.3897/folmed.65.e68256">10.3897/folmed.65.e68256</ext-link>.</p>
      </sec>
    </notes>
  </front>
  <body>
    <sec sec-type="Introduction" id="SECID0E4E">
      <title>Introduction</title>
      <p>The WHO classified two types of giant cell lesions originating from the tendon and synovium. Giant cell tumors of the patellar tendon sheath (GCT-TS) can be classified as localized (L-) or diffuse (D-) type. L-GCT-TS initially occurs in the tendon sheaths of the hand and foot and is characterized by a nodular mass. In comparison, D-GCT-TS occurs in large joints with increased growth and recurrence rates. It is common for the knee joint to invade the outer part of the joint capsule. The prevalence of GCTs is 1/50,000 per population. Females tend to be affected 1.6 times more than males. Tissues around the knee were affected in only 2 of 71 patients in the study by Monaghan et al.<sup>[<xref ref-type="bibr" rid="B1">1</xref>]</sup></p>
      <p>The exact etiology of GCTTS remains unknown. Clinical characteristics are proportional to the location; however, differential diagnosis remains challenging. The predominant symptom is painless soft tissue masses, accompanied by discomfort, swelling, and restriction of the range of motion.<sup>[<xref ref-type="bibr" rid="B2">2</xref>]</sup></p>
      <p>The aim of this study was to report a rare case of GCT-TS of the patellar tendon. This indicates the diagnostic procedures and treatment options for GCT-TS of the patellar tendon.</p>
    </sec>
    <sec sec-type="Case report" id="SECID0ESF">
      <title>Case report</title>
      <p><italic>A 13-year-old boy presented to our hospital complaining of chronic, continuous, worsening pain in the anterior surface of the right knee for 2 years. There was no history of trauma during the study period. On clinical evaluation, no knee instability was observed, and all the meniscal evaluation tests were negative. A hard-textured, immobile, palpable mass was found on the inner side of the patellar tendon. Full flexion was limited by 10 degrees compared to the left knee. Radiographic examination revealed no abnormalities. Magnetic resonance imaging (MRI) revealed an extra-articular formation 40×20 mm on the inner half of the patellar tendon, which had a high-intensity signal in T2 sequences</italic><bold><italic>(Fig. <xref ref-type="fig" rid="F1">1</xref>)</italic></bold> . <italic>The formation was initially considered a hemorrhagic collection. Open arthrotomy was performed with complete surgical excision of the lesion</italic><bold><italic>(Fig. <xref ref-type="fig" rid="F2">2</xref>)</italic></bold> . <italic>The histopathological examination revealed a giant cell tumor. The tumor was slightly brownish in color, capsulated, 50×33×25 mm in size, and rubbery in consistency. Postoperative assessment revealed no complications or symptom reappearance. The last follow-up was 2 years after surgery.</italic></p>
      <fig id="F1" position="float" orientation="portrait">
        <object-id content-type="arpha">07A82202-1850-5ED0-BE22-422A300D9392</object-id>
        <label>Figure 1.</label>
        <caption>
          <p>T-2 weighted MR1, coronal and sagittal planes, depicting the giant cell tumor of the patellar tendon sheath.</p>
        </caption>
        <graphic xlink:href="foliamedica-65-2-e68256-g001.jpg" position="float" orientation="portrait" xlink:type="simple" id="oo_848102.jpg">
          <uri content-type="original_file">https://binary.pensoft.net/fig/848102</uri>
        </graphic>
      </fig>
      <fig id="F2" position="float" orientation="portrait">
        <object-id content-type="arpha">8C64D84D-CE50-5879-9FD8-722960681E55</object-id>
        <label>Figure 2.</label>
        <caption>
          <p>The excised lesion of the giant cell tumor.</p>
        </caption>
        <graphic xlink:href="foliamedica-65-2-e68256-g002.jpg" position="float" orientation="portrait" xlink:type="simple" id="oo_848103.jpg">
          <uri content-type="original_file">https://binary.pensoft.net/fig/848103</uri>
        </graphic>
      </fig>
    </sec>
    <sec sec-type="Discussion" id="SECID0ENG">
      <title>Discussion</title>
      <p>The patellar tendon is considered a rare location for manifestation of GCTTS. This condition is mostly localized in the hands (93%) and rarely around the knee (2.82%). The mean age at presentation is 46.3 years.<sup>[<xref ref-type="bibr" rid="B1">1</xref>]</sup> It seems that the juvenile incidence of GCT-TS around the knee is low. This makes our case even more uncommon. However, the exact etiology remains unclear. However, there are some theories that include several factors that might contribute to this rare condition, including inflammation, neoplasia, trauma, toxic substances, allergies, and genetic factors.<sup>[<xref ref-type="bibr" rid="B3">3</xref>]</sup></p>
      <p>The most predominant symptom is a painless soft-tissue mass, accompanied by pain, swelling, and ROM limitation.<sup>[<xref ref-type="bibr" rid="B2">2</xref>]</sup> The clinical manifestations in our patient were quite similar. Soft tissue masses may be detected by palpation or imaging techniques. The differential diagnosis includes intra-articular and extra-articular lesions, including PVNS, synovial cyst, and synovial sarcoma.<sup>[<xref ref-type="bibr" rid="B4">4</xref>]</sup> The most important imaging examination is magnetic resonance imaging (MRI). GCT-TS is characterized by a weak or intermediate signal on the T1 and T2 sequences. Synovial cysts show high T2 signals, but other pathologies, including xanthoma and Morton neuroma, usually show intermediate T2 signals. In these cases, the diagnosis is guided by the location, symptomatology, and clinical findings. Ultrasound can be indicative of a hypervascularized mass of variable aspect but with a suggestive location, which optimally guides synovial biopsy.<sup>[<xref ref-type="bibr" rid="B5">5</xref>]</sup></p>
      <p>Surgical treatment involves an assorted approach. Abdullah et al. treated a patient with arthrotomy.<sup>[<xref ref-type="bibr" rid="B3">3</xref>]</sup> In contrast, Chechik et al. treated patients with arthroscopy with similar results.<sup>[<xref ref-type="bibr" rid="B4">4</xref>]</sup> In all cases, the examination was unremarkable at the last follow-up. No recurrence was observed.</p>
      <p>Histopathological examination confirmed the diagnosis after tumor excision. Typically, the tumor is soft, slightly brownish, or reddish-tan. Common findings include yellow and white areas due to xanthomatous changes and fibrous tissue. Microscopical elements are addressed into distinction of forms including the presence of foam cells, multinucleated giant cells, stromal cells with or without hemosiderin deposits and collagen.<sup>[<xref ref-type="bibr" rid="B6">6</xref>]</sup> GCT is associated with mutations in H3F3A (G34W or G34L).<sup>[<xref ref-type="bibr" rid="B7">7</xref>]</sup></p>
      <p>Open arthrotomy and arthroscopic recession are considered acceptable treatment options. According to the cases, both interventions showed similar results. Moreover, they showed no complications, pain relief, or ROM recovery. Minimally invasive approaches, such as arthroscopy, allow for the effective examination of all compartments of the knee with compelling excision of the lesion. The recurrence rate is independent of mitotic activity and incomplete resection. <sup>[<xref ref-type="bibr" rid="B2">2</xref>]</sup> As a result, radiotherapy may play a decisive role in these cases.<sup>[<xref ref-type="bibr" rid="B6">6</xref>]</sup> GCT-TS, as a benign lesion, showed no recurrence in our case. Ushijima et al. reported local postoperative recurrence rates of 10%-20% within 24 months, particularly in non-removable satellite nodules.<sup>[<xref ref-type="bibr" rid="B8">8</xref>]</sup> Thus, postoperative follow-up can be considered as devoid of lesions.</p>
    </sec>
    <sec sec-type="Conclusions" id="SECID0ENBAC">
      <title>Conclusions</title>
      <p>GCT-TS is an uncommon benign tumor. It mimics common knee symptoms. A differential diagnosis is definitely a challenge. It is crucial to indicate patient history, meticulous clinical examination, and cutting-edge imaging techniques; however, the definitive diagnosis is confirmed by histopathological examination. Available operation approaches have demonstrated similar results, which lead to symptom relief and a low recurrence rate.</p>
    </sec>
    <sec sec-type="Source of funding" id="SECID0ESBAC">
      <title>Source of funding</title>
      <p>This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Written consent was obtained from all the patients.</p>
    </sec>
    <sec sec-type="Declaration of Competing Interest" id="SECID0EXBAC">
      <title>Declaration of Competing Interest</title>
      <p>All authors have declared no conﬂict of interest.</p>
    </sec>
  </body>
  <back>
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      <title>References</title>
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</article>
