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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.66.e116865</article-id>
      <article-id pub-id-type="publisher-id">116865</article-id>
      <article-categories>
        <subj-group subj-group-type="heading">
          <subject>Case Report</subject>
        </subj-group>
        <subj-group subj-group-type="scientific_subject">
          <subject>Obstetrics &amp; Gynecology</subject>
          <subject>Oncology</subject>
          <subject>Radiology &amp; Imaging</subject>
          <subject>Surgery &amp; Invasive treatment</subject>
          <subject>Women health</subject>
        </subj-group>
      </article-categories>
      <title-group>
        <article-title>Paraovarian tumor of borderline malignancy: A case report</article-title>
      </title-group>
      <contrib-group content-type="authors">
        <contrib contrib-type="author" corresp="yes">
          <name name-style="western">
            <surname>Bairaktaris</surname>
            <given-names>Dimitrios</given-names>
          </name>
          <email xlink:type="simple">mpairaktarisdim@gmail.com</email>
          <uri content-type="orcid">https://orcid.org/0009-0000-2709-333X</uri>
          <xref ref-type="aff" rid="A1">1</xref>
        </contrib>
        <contrib contrib-type="author" corresp="no">
          <name name-style="western">
            <surname>Tsoplaktsoglou</surname>
            <given-names>Stefania</given-names>
          </name>
          <xref ref-type="aff" rid="A2">2</xref>
        </contrib>
        <contrib contrib-type="author" corresp="no">
          <name name-style="western">
            <surname>Souka</surname>
            <given-names>Efthymia</given-names>
          </name>
          <uri content-type="orcid">https://orcid.org/0000-0002-5298-1926</uri>
          <xref ref-type="aff" rid="A1">1</xref>
        </contrib>
        <contrib contrib-type="author" corresp="no">
          <name name-style="western">
            <surname>Kalmantis</surname>
            <given-names>Konstantinos</given-names>
          </name>
          <uri content-type="orcid">https://orcid.org/0000-0002-5758-8840</uri>
          <xref ref-type="aff" rid="A3">3</xref>
        </contrib>
        <contrib contrib-type="author" corresp="no">
          <name name-style="western">
            <surname>Iavazzo</surname>
            <given-names>Christos</given-names>
          </name>
          <uri content-type="orcid">https://orcid.org/0000-0003-2119-4599</uri>
          <xref ref-type="aff" rid="A1">1</xref>
        </contrib>
      </contrib-group>
      <aff id="A1">
        <label>1</label>
        <addr-line>Department of Gynecological Oncology, Metaxa Memorial Cancer Hospital, Piraeus, Athens, Greece</addr-line>
      </aff>
      <aff id="A2">
        <label>2</label>
        <addr-line>Service de Gynécologie / Obstétrique, CHI André Grégoire, Paris, France</addr-line>
      </aff>
      <aff id="A3">
        <label>3</label>
        <addr-line>Department of Pathology, Metaxa Memorial Cancer Hospital, Piraeus, Athens, Greece</addr-line>
      </aff>
      <aff id="A4">
        <label>4</label>
        <addr-line>Department of Obstetrics and Gynecology, Iaso, Maternity Hospital, Athens, Greece</addr-line>
      </aff>
      <author-notes>
        <fn fn-type="corresp">
          <p>Corresponding author: Dimitrios Bairaktaris, Department of Gynecological Oncology, Metaxa Memorial Cancer Hospital, Charilaou Trikoupi 39, 18536, Piraeus, Athens, Greece; Email: <email xlink:type="simple">mpairaktarisdim@gmail.com</email>; Tel.: +306976679837</p>
        </fn>
      </author-notes>
      <pub-date pub-type="collection">
        <year>2024</year>
      </pub-date>
      <pub-date pub-type="epub">
        <day>29</day>
        <month>02</month>
        <year>2024</year>
      </pub-date>
      <volume>66</volume>
      <issue>1</issue>
      <fpage>128</fpage>
      <lpage>131</lpage>
      <uri content-type="arpha" xlink:href="http://openbiodiv.net/3C0750D2-D754-599D-91B8-E62E5555A62C">3C0750D2-D754-599D-91B8-E62E5555A62C</uri>
      <history>
        <date date-type="received">
          <day>05</day>
          <month>12</month>
          <year>2023</year>
        </date>
        <date date-type="accepted">
          <day>19</day>
          <month>12</month>
          <year>2023</year>
        </date>
      </history>
      <permissions>
        <copyright-statement>Dimitrios Bairaktaris, Stefania Tsoplaktsoglou, Efthymia Souka, Konstantinos Kalmantis, Christos Iavazzo</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>Paraovarian tumors of borderline malignancy (<abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0ELE">PTBM</abbrev>) are exceedingly rare, with only slightly over 60 cases reported worldwide. This report presents the case of a 22-year-old nulliparous patient who incidentally discovered a left paraovarian mass during a routine abdominal ultrasound. Subsequent MRI revealed a 2.5×2.1 cm cystic lesion located in close proximity to, but outside of, the left ovary, with no other pathological findings. A laparoscopic cystectomy was performed with meticulous care to prevent tumor spillage, and the patient experienced an uneventful recovery. Histopathological examination unveiled irregularly shaped tissue measuring 2.2×1.2×1 cm, characterized by fibrous tissue/wall with spindle cell stroma and an epithelium displaying features consistent with a serous borderline tumor. Our multidisciplinary team recommended diligent follow-up. This case contributes to the existing literature on <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0EPE">PTBM</abbrev> and highlights the imperative for additional cases to enhance our comprehension of the optimal management of these exceedingly rare tumors.</p>
      </abstract>
      <kwd-group>
        <label>Keywords</label>
        <kwd>adnexal mass</kwd>
        <kwd>fertility-sparing</kwd>
        <kwd>low malignant potential</kwd>
        <kwd>paratubal</kwd>
        <kwd>paraovarian</kwd>
      </kwd-group>
    </article-meta>
    <notes>
      <sec sec-type="Citation" id="SECID0E2E">
        <title>Citation</title>
        <p>Bairaktaris D, Tsoplaktsoglou S, Souka E, Kalmantis K, Iavazzo C. Paraovarian tumor of borderline malignancy: A case report. Folia Med (Plovdiv) 2024;66(1):128-131. doi: <ext-link xlink:type="simple" ext-link-type="doi" xlink:href="10.3897/folmed.66.e116865">10.3897/folmed.66.e116865</ext-link>.</p>
      </sec>
    </notes>
  </front>
  <body>
    <sec sec-type="Introduction" id="SECID0EHF">
      <title>Introduction</title>
      <p>Paraovarian cysts (<abbrev xlink:title="Paraovarian cysts" id="ABBRID0ENF">POCs</abbrev>), located between the fallopian tube and the ovary, comprise around 10% of adnexal masses.<sup>[<xref ref-type="bibr" rid="B1">1</xref>]</sup> While their precise occurrence remains uncertain, they are estimated at roughly 3% in the general population, peaking in the third and fourth decades of life.<sup>[<xref ref-type="bibr" rid="B1">1</xref>]</sup> Predominantly benign, these cysts are often asymptomatic and discovered incidentally during clinical examinations.<sup>[<xref ref-type="bibr" rid="B1">1</xref>]</sup> Treatment usually involves laparoscopic cystectomy, guided by factors like cyst size and patient age.<sup>[<xref ref-type="bibr" rid="B1">1</xref>, <xref ref-type="bibr" rid="B2">2</xref>]</sup> However, rare cases of malignant or borderline tumors exist, posing diagnostic challenges due to similarities in symptoms and imaging features with benign cysts. Given the lack of specific treatment guidelines, reporting such cases becomes crucial. This report presents a case of a 22-year-old woman with a pelvic mass near the left ovary.</p>
    </sec>
    <sec sec-type="Case report" id="SECID0ERG">
      <title>Case report</title>
      <p>
        <italic>A 22-year-old nulliparous female presented as an outpatient at our institution with a pelvic mass located in close proximity to the left ovary. The patient did not report any symptoms, had no significant medical history and had a negative family history for malignancy.</italic>
      </p>
      <p><italic>The discovery of the mass was incidental during a routine outpatient abdominal ultrasound. Outpatient transvaginal ultrasound (<abbrev xlink:title="transvaginal ultrasound" id="ABBRID0E4G">TVUS</abbrev>) revealed a cystic lesion measuring 2.5 cm in size, closely associated with the left ovary. The lesion exhibited numerous echogenic papillary projections on the cyst wall. Subsequent MRI of the lower abdomen confirmed the presence of a cystic lesion measuring 2.5×2.1 cm, located in close proximity to the left ovary but outside of it</italic><bold><italic>(Figs <xref ref-type="fig" rid="F1">1</xref>–<xref ref-type="fig" rid="F4">4</xref>)</italic></bold> . <italic>This lesion contained smaller nodules inside, with a maximum diameter of 4 mm. Laboratory assessments, including tumor markers, yielded normal results (CA-125: 10.7 U/mL, CEA: 0.97 ng/mL, CA 15-3: 11 U/mL, AFP: 2.83 IU/ml, CA 19-9: 3.30 U/mL, HE-4: 47.11 pmol/l).</italic></p>
      <p><italic>The patient underwent laparoscopic cystectomy, during which a laparoscopic bag was used and no tumor spillage was observed within the abdominal cavity. Subsequently, the patient experienced an uneventful postoperative recovery and was discharged from the hospital on the first postoperative day. The pathology report described a grayish mass with irregular dimensions of 2.2×1.2×1 cm, identified as the wall of a ruptured cystic lesion (macroscopic report). Microscopically, examination revealed the presence of fibrous tissue with occasional spindle cell stroma, as well as an epithelium displaying histopathological features consistent with a serous borderline tumor (World Health Organization 2020 <sup>[<xref ref-type="bibr" rid="B3">3</xref>]</sup>)</italic><bold><italic>(Figs <xref ref-type="fig" rid="F5">5</xref>–<xref ref-type="fig" rid="F7">7</xref></italic></bold>) .</p>
      <fig id="F1" position="float" orientation="portrait">
        <object-id content-type="arpha">11095E0E-F044-50A2-B6AD-0E806BE465B2</object-id>
        <label>Figure 1.</label>
        <caption>
          <p>MRI T2-weighted examination of small pelvis. Right and left ovaries appear normal (blue arrows). The <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0EHFAC">PTBM</abbrev> is in contact with, but outside of, the left ovary (yellow arrow, axial plane).</p>
        </caption>
        <graphic xlink:href="foliamedica-66-1-e116865-g001.jpg" position="float" orientation="portrait" xlink:type="simple" id="oo_994869.jpg">
          <uri content-type="original_file">https://binary.pensoft.net/fig/994869</uri>
        </graphic>
      </fig>
      <fig id="F2" position="float" orientation="portrait">
        <object-id content-type="arpha">1E2E103E-0678-59E1-834F-AFFCA7A23D73</object-id>
        <label>Figure 2.</label>
        <caption>
          <p>MRI T2-weighted examination of <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0EYFAC">PTBM</abbrev>. Right normal ovary (blue arrow) and the <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0E3FAC">PTBM</abbrev> measuring 2.52 cm (red arrow, axial plane).</p>
        </caption>
        <graphic xlink:href="foliamedica-66-1-e116865-g002.jpg" position="float" orientation="portrait" xlink:type="simple" id="oo_994870.jpg">
          <uri content-type="original_file">https://binary.pensoft.net/fig/994870</uri>
        </graphic>
      </fig>
      <fig id="F3" position="float" orientation="portrait">
        <object-id content-type="arpha">35158305-32A8-5ECA-BD85-7B84F75917D0</object-id>
        <label>Figure 3.</label>
        <caption>
          <p>MRI T2-weighted examination of uterus, left ovary and <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0ENGAC">PTBM</abbrev>. The <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0ERGAC">PTBM</abbrev> (blue arrow) appears to be in contact with, but outside of the left normal ovary (yellow arrow, coronal plane).</p>
        </caption>
        <graphic xlink:href="foliamedica-66-1-e116865-g003.jpg" position="float" orientation="portrait" xlink:type="simple" id="oo_994871.jpg">
          <uri content-type="original_file">https://binary.pensoft.net/fig/994871</uri>
        </graphic>
      </fig>
      <fig id="F4" position="float" orientation="portrait">
        <object-id content-type="arpha">7F21C792-9E84-590A-92B1-E6634A5412E9</object-id>
        <label>Figure 4.</label>
        <caption>
          <p>MRI T1-weighted examination of small pelvis. <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0ECHAC">PTBM</abbrev> (blue arrow, axial plane).</p>
        </caption>
        <graphic xlink:href="foliamedica-66-1-e116865-g004.jpg" position="float" orientation="portrait" xlink:type="simple" id="oo_994872.jpg">
          <uri content-type="original_file">https://binary.pensoft.net/fig/994872</uri>
        </graphic>
      </fig>
      <fig id="F5" position="float" orientation="portrait">
        <object-id content-type="arpha">10F4B30F-BDF9-5286-9B86-08C01DD20FE6</object-id>
        <label>Figure 5.</label>
        <caption>
          <p>Hematoxylin and eosin staining (×40). Fibrous cystic wall with complex branching papillary stromal proliferation lined by epithelium with areas of epithelial proliferation (&gt;10% of the tumor epithelium).</p>
        </caption>
        <graphic xlink:href="foliamedica-66-1-e116865-g005.jpg" position="float" orientation="portrait" xlink:type="simple" id="oo_994873.jpg">
          <uri content-type="original_file">https://binary.pensoft.net/fig/994873</uri>
        </graphic>
      </fig>
      <fig id="F6" position="float" orientation="portrait">
        <object-id content-type="arpha">B0034E34-363A-516B-A1C3-AF148684ABC6</object-id>
        <label>Figure 6.</label>
        <caption>
          <p>Hematoxylin and eosin staining (×200). Papillary architecture with cuboidal to columnar serous type cells with tufting and nuclear atypia.</p>
        </caption>
        <graphic xlink:href="foliamedica-66-1-e116865-g006.jpg" position="float" orientation="portrait" xlink:type="simple" id="oo_994874.jpg">
          <uri content-type="original_file">https://binary.pensoft.net/fig/994874</uri>
        </graphic>
      </fig>
      <fig id="F7" position="float" orientation="portrait">
        <object-id content-type="arpha">A27CB87D-CA09-564B-94DC-909F6A149641</object-id>
        <label>Figure 7.</label>
        <caption>
          <p>Hematoxylin and eosin staining (×400). Uniform mild cytologic atypia, epithelial pseudostratification and tufting, without prominent mitotic activity.</p>
        </caption>
        <graphic xlink:href="foliamedica-66-1-e116865-g007.jpg" position="float" orientation="portrait" xlink:type="simple" id="oo_994875.jpg">
          <uri content-type="original_file">https://binary.pensoft.net/fig/994875</uri>
        </graphic>
      </fig>
      <p>
        <italic>The patient’s case was thoroughly reviewed by our multidisciplinary team, and a decision was made to proceed with close follow-up examinations. No further interventions were planned unless remaining disease was detected. Remarkably, the patient remained disease-free at the 12-month follow-up mark. Images in this publication have been used with the explicit written consent of the patient involved, ensuring adherence to ethical standards and patient confidentiality.</italic>
      </p>
    </sec>
    <sec sec-type="Discussion" id="SECID0EQAAC">
      <title>Discussion</title>
      <p>While <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0EWAAC">PTBM</abbrev> are rare and often asymptomatic, their diagnosis poses challenges. Imaging techniques, such as ultrasound, CT, and MRI, provide limited preoperative specificity.<sup>[<xref ref-type="bibr" rid="B4">4</xref>]</sup> Histologically, <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0EBBAC">PTBM</abbrev> exhibit distinct features, resembling ovarian borderline tumors.<sup>[<xref ref-type="bibr" rid="B2">2</xref>]</sup> Yet, due to their infrequency, establishing standardized guidelines for their treatment remains challenging.</p>
      <p>The range of surgical interventions for <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0EOBAC">PTBM</abbrev> is guided by factors such as patient age and fertility considerations. These procedures span from fertility-preserving surgeries to more extensive interventions like hysterectomy.<sup>[<xref ref-type="bibr" rid="B4">4</xref>, <xref ref-type="bibr" rid="B5">5</xref>]</sup> The absence of standardized protocols for specific procedures in managing <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0E4BAC">PTBM</abbrev>, such as lymphadenectomy or omentectomy, highlights the need for clearer guidelines in this domain.</p>
      <p>Given the rarity of <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0EDCAC">PTBM</abbrev>, devising specific treatment guidelines presents a significant challenge. Typically, due to patients’ age demographics, there’s a preference for fertility-preserving surgeries along with vigilant postoperative surveillance.<sup>[<xref ref-type="bibr" rid="B5 B6 B7 B8">5–8</xref>]</sup> Conversely, patients without the intent to maintain fertility may undergo more comprehensive surgical measures, such as hysterectomy along with bilateral salpingo-oophorectomy. Considering the young age of our patient, we opted for a fertility-sparing approach, coupled with careful monitoring. Our case highlights the intricacies in diagnosing <abbrev xlink:title="Paraovarian tumors of borderline malignancy" id="ABBRID0EOCAC">PTBM</abbrev> and emphasizes the urgent need for standardized management protocols for these uncommon conditions.</p>
    </sec>
  </body>
  <back>
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</article>
