<?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="research-article" dtd-version="1.0" 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">CE</journal-id>
<journal-title-group>
<journal-title>Clinical Endoscopy</journal-title><abbrev-journal-title>Clin Endosc</abbrev-journal-title></journal-title-group>
<issn pub-type="ppub">2234-2400</issn>
<issn pub-type="epub">2234-2443</issn>
<publisher>
<publisher-name>Korean Society of Gastrointestinal Endoscopy</publisher-name></publisher></journal-meta>
<article-meta>
<article-id pub-id-type="doi">10.5946/ce.2023.231</article-id>
<article-id pub-id-type="publisher-id">ce-2023-231</article-id>
<article-categories>
<subj-group>
<subject>Brief Report</subject></subj-group></article-categories>
<title-group>
<article-title>Jejunal Dieulafoy’s lesion resembling subepithelial mass resulting in profound gastrointestinal hemorrhage</article-title>
<alt-title alt-title-type="right-running-head">Jejunal Dieulafoy's lesion resembling mass</alt-title>
</title-group>
<contrib-group>
<contrib contrib-type="author">
<contrib-id contrib-id-type="orcid">http://orcid.org/0009-0001-3408-7277</contrib-id>
<name><surname>Chaemsupaphan</surname><given-names>Thanaboon</given-names></name>
<xref ref-type="aff" rid="af1-ce-2023-231"><sup>1</sup></xref>
</contrib>
<contrib contrib-type="author">
<contrib-id contrib-id-type="orcid">http://orcid.org/0000-0002-8346-7487</contrib-id>
<name><surname>Geeratragool</surname><given-names>Tanawat</given-names></name>
<xref ref-type="aff" rid="af1-ce-2023-231"><sup>1</sup></xref>
</contrib>
<contrib contrib-type="author">
<contrib-id contrib-id-type="orcid">http://orcid.org/0000-0001-7831-2475</contrib-id>
<name><surname>Angkathunyakul</surname><given-names>Napat</given-names></name>
<xref ref-type="aff" rid="af2-ce-2023-231"><sup>2</sup></xref>
</contrib>
<contrib contrib-type="author">
<contrib-id contrib-id-type="orcid">http://orcid.org/0009-0005-9452-1986</contrib-id>
<name><surname>Phothisirisakulwong</surname><given-names>Arissa</given-names></name>
<xref ref-type="aff" rid="af3-ce-2023-231"><sup>3</sup></xref>
</contrib>
<contrib contrib-type="author">
<contrib-id contrib-id-type="orcid">http://orcid.org/0000-0002-7556-3540</contrib-id>
<name><surname>Maneerattanaporn</surname><given-names>Monthira</given-names></name>
<xref ref-type="corresp" rid="c1-ce-2023-231"/>
<xref ref-type="aff" rid="af1-ce-2023-231"><sup>1</sup></xref>
</contrib>
<aff id="af1-ce-2023-231">
<label>1</label>Division of Gastroenterology, Department of Medicine, Siriraj Hospital, Mahidol University, Bangkok, <country>Thailand</country></aff>
<aff id="af2-ce-2023-231">
<label>2</label>Department of Pathology, Siriraj Hospital, Mahidol University, Bangkok, <country>Thailand</country></aff>
<aff id="af3-ce-2023-231">
<label>3</label>Department of Radiology, Siriraj Hospital, Mahidol University, Bangkok, <country>Thailand</country></aff>
</contrib-group>
<author-notes>
<corresp id="c1-ce-2023-231">Correspondence: Monthira Maneerattanaporn Division of Gastroenterology, Department of Medicine, Siriraj Hospital, Mahidol University, 2 Wanglang Road, Bangkoknoi, Bangkok 10700, Thailand E-mail: <email>monthira.man@mahidol.ac.th</email></corresp>
</author-notes>
<pub-date pub-type="ppub">
<month>7</month>
<year>2024</year></pub-date>
<pub-date pub-type="epub">
<day>29</day>
<month>3</month>
<year>2024</year></pub-date>
<volume>57</volume>
<issue>4</issue>
<fpage>552</fpage>
<lpage>554</lpage>
<history>
<date date-type="received">
<day>14</day>
<month>09</month>
<year>2023</year></date>
<date date-type="rev-recd">
<day>26</day>
<month>09</month>
<year>2023</year></date>
<date date-type="accepted">
<day>30</day>
<month>09</month>
<year>2023</year></date>
</history>
<permissions>
<copyright-statement>&#x000A9; 2024 Korean Society of Gastrointestinal Endoscopy</copyright-statement>
<copyright-year>2024</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="https://creativecommons.org/licenses/by-nc/4.0/">https://creativecommons.org/licenses/by-nc/4.0/</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></front>
<body>
<p>Dieulafoy&#x02019;s lesion (DL) is a vascular malformation that causes profound gastrointestinal (GI) bleeding. The incidence, accounting for approximately 1% to 2%, is relatively uncommon.<xref ref-type="bibr" rid="b1-ce-2023-231">1</xref> Approximately 70% of these lesions are located in the proximal stomach, within 6 cm of the esophagogastric junction. Other common locations include the duodenum and esophagus. DL usually manifests as a small vascular protrusion without significant mucosal defect.<xref ref-type="bibr" rid="b2-ce-2023-231">2</xref></p>
<p>A 73-year-old Thai man with a history of diabetes mellitus, hypertension, and a minor ischemic stroke presented with hematemesis. He was admitted for the treatment of coronavirus disease 2019 (COVID-19) pneumonia. During his hospitalization, he received oxygen support, intravenous remdesivir, systemic corticosteroids, and enoxaparin dose for 1 week for thrombotic prophylaxis. His overall condition gradually improved within a week without evidence of GI bleeding. In the 2nd week of his admission, he developed hypovolemic shock, evidenced by the passage of copious melena. The patient was resuscitated and finally stabilized after fluid therapy and blood transfusion. He denied administration of over-the-counter drugs and had a history of abdominal surgery. His hemoglobin level decreased from 11.9 to 8 g/dL, whereas his platelet count and coagulogram were within normal limits. His blood urea nitrogen level was 40.3 mg/dL, with a creatinine level of 0.7 mg/dL. Esophagogastroduodenoscopy was performed that did not lead to significant lesions, except for minimal blood-stained mucosa at the second part of the duodenum. Hence, push enteroscopy was performed, which revealed a 2.5 cm, round, subepithelial mass with a large central defect covered by an adherent clot in the proximal jejunum (<xref rid="f1-ce-2023-231" ref-type="fig">Fig. 1</xref>). The initial impression was gastrointestinal stromal tumor (GIST) with suspicion of ongoing bleeding. Thus, an anatomical landmark was located by applying a hemostatic clip adjacent to the lesion, and then abdominal computed tomographic angiography (CTA) was promptly performed. Radiological examination revealed a round-shaped arterial enhancing lesion in the jejunum without active contrast extravasation. This lesion was persistently enhanced in the portal venous phase, suggesting a vascular lesion or tumor (<xref rid="f2-ce-2023-231" ref-type="fig">Fig. 2A</xref>). This imaging revealed no evidence of abnormal vessels or lymphadenopathy. An interventional radiologist was consulted due to the continued passage of maroon stool and hemodynamic instability. Angiography was performed with the intention of angioembolization. After super-selection angiography into the jejunal branch of the superior mesenteric artery, an accumulation of contrast media, supplied by multiple small jejunal branches, was identified (<xref rid="f2-ce-2023-231" ref-type="fig">Fig. 2B</xref>). Embolization was not performed as active contrast extravasation could not be demonstrated. Subsequently, surgery was performed to stop the bleeding. Exploratory laparotomy revealed a non-active bleeding subepithelial mass, excised along with 20 cm of small bowel. The bleeding stopped immediately after the surgery. The lesion revealed a large thrombus within the submucosa (<xref rid="f3-ce-2023-231" ref-type="fig">Fig. 3A</xref>). The histopathological examination revealed a submucosal persistent-caliber artery that was dilated with a recent thrombus, and the lesion was connected to the overlying mucosal defect (<xref rid="f3-ce-2023-231" ref-type="fig">Fig. 3B</xref>). The vascular wall of the lesion was exceedingly thickened compared to the normal submucosal artery (<xref rid="f3-ce-2023-231" ref-type="fig">Fig. 3C</xref>). The overall pathological finding is consistent with DL. The postoperative course of the patient was uneventful, without further bleeding.</p>
<p>DL is a condition characterized by an enlarged blood vessel beneath the GI mucosa that erodes the overlying tissue, resulting in bleeding without ulcers or erosion.<xref ref-type="bibr" rid="b3-ce-2023-231">3</xref> Some predisposing factors have been identified, including male sex, advanced age, administration of antithrombotic drugs, and comorbidities, such as cardiovascular disease and chronic renal insufficiency despite the lack of knowledge regarding its exact cause.<xref ref-type="bibr" rid="b4-ce-2023-231">4</xref> One case report recently described a patient who experienced rectal bleeding due to DL while hospitalized for severe COVID-19 pneumonia and respiratory failure.<xref ref-type="bibr" rid="b5-ce-2023-231">5</xref> This indicates that severe medical conditions, including SARS-CoV-2 infection, may precede this vascular bleeding. The jejunal location is extremely uncommon and predominantly observed in patients aged 70&#x02013;80 years. The proposed mechanism for this occurrence is related to age-related wear and tear of the submucosal vessels.<xref ref-type="bibr" rid="b6-ce-2023-231">6</xref> DL typically presents as a small pigmented protuberance, measuring &#x0003c;10 mm, with minimal mucosal defect. It often exhibits active bleeding during endoscopy.<xref ref-type="bibr" rid="b2-ce-2023-231">2</xref> We demonstrated an atypical appearance of DL, characterized by a large subepithelial mass with a significant central defect. This unique manifestation indicates a tumor rather than a vascular lesion. GIST was the common differential diagnosis for the bleeding subepithelial mass according to the current endoscopic evaluation. Few cases of DL mimicking GIST have been reported in the literature, with an average size of 2 cm in the stomach and 1 cm in jejunum (<xref rid="t1-ce-2023-231" ref-type="table">Table 1</xref>).<xref ref-type="bibr" rid="b7-ce-2023-231">7</xref>-<xref ref-type="bibr" rid="b9-ce-2023-231">9</xref> Other potential rare causes of bleeding subepithelial mass include glomus tumor and arteriovenous malformation.<xref ref-type="bibr" rid="b10-ce-2023-231">10</xref> Although endoscopic findings can make definitive diagnosis difficult, the management approach is consistent. Direct endoscopic intervention is not the preferred method, as clot removal or biopsy might exacerbate bleeding, posing a life-threatening risk. Patients with subepithelial mass bleeding should undergo CTA followed by embolization, with surgical resection as the definitive treatment. Informed consent was obtained for this case report.</p>
</body>
<back>
<fn-group>
<fn fn-type="conflict"><p><bold>Conflicts of Interest</bold></p>
<p>The authors have no potential conflicts of interest.</p></fn>
<fn fn-type="financial-disclosure"><p><bold>Funding</bold></p>
<p>None.</p></fn>
<fn fn-type="participating-researchers"><p><bold>Author Contributions</bold></p>
<p>Conceptualization: MM, TC; Data curation: all authors; Investigations: TC, NA, AP; Writing–original draft: TC; Writing–review &amp; editing: TG, MM, NA, AP.</p></fn>
</fn-group>
<ref-list>
<title>REFERENCES</title>
<ref id="b1-ce-2023-231">
<label>1</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name><surname>Kim</surname><given-names>SE</given-names></name>
<name><surname>Kim</surname><given-names>HJ</given-names></name>
<name><surname>Koh</surname><given-names>M</given-names></name>
<etal/>
</person-group>
<article-title>A practical approach for small bowel bleeding</article-title>
<source>Clin Endosc</source>
<year>2023</year>
<volume>56</volume>
<fpage>283</fpage>
<lpage>289</lpage>
</element-citation></ref>
<ref id="b2-ce-2023-231">
<label>2</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name><surname>Baxter</surname><given-names>M</given-names></name>
<name><surname>Aly</surname><given-names>EH</given-names></name>
</person-group>
<article-title>Dieulafoy&#x00027;s lesion: current trends in diagnosis and management</article-title>
<source>Ann R Coll Surg Engl</source>
<year>2010</year>
<volume>92</volume>
<fpage>548</fpage>
<lpage>554</lpage>
</element-citation></ref>
<ref id="b3-ce-2023-231">
<label>3</label>
<element-citation publication-type="book">
<person-group person-group-type="author">
<name><surname>Malik</surname><given-names>TF</given-names></name>
<name><surname>Anjum</surname><given-names>F</given-names></name>
</person-group>
<source>Dieulafoys Lesion causing gastrointestinal bleeding</source>
<publisher-name>StatPearls</publisher-name>
<year>2023</year>
</element-citation></ref>
<ref id="b4-ce-2023-231">
<label>4</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name><surname>Sakai</surname><given-names>E</given-names></name>
<name><surname>Ohata</surname><given-names>K</given-names></name>
<name><surname>Nakajima</surname><given-names>A</given-names></name>
<etal/>
</person-group>
<article-title>Diagnosis and therapeutic strategies for small bowel vascular lesions</article-title>
<source>World J Gastroenterol</source>
<year>2019</year>
<volume>25</volume>
<fpage>2720</fpage>
<lpage>2733</lpage>
</element-citation></ref>
<ref id="b5-ce-2023-231">
<label>5</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name><surname>Tahir</surname><given-names>MM</given-names></name>
<name><surname>Saeed</surname><given-names>S</given-names></name>
<name><surname>Hlaing</surname><given-names>SS</given-names></name>
<etal/>
</person-group>
<article-title>Dieulafoy lesion causing lower GI bleeding: a case of COVID-19 critical illness prompting unusual presentation of a more rare condition</article-title>
<source>Critical Care</source>
<year>2023</year>
<volume>162</volume>
<fpage>A926</fpage>
<lpage>A927</lpage>
</element-citation></ref>
<ref id="b6-ce-2023-231">
<label>6</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name><surname>Malik</surname><given-names>A</given-names></name>
<name><surname>Inayat</surname><given-names>F</given-names></name>
<name><surname>Goraya</surname><given-names>MH</given-names></name>
<etal/>
</person-group>
<article-title>Jejunal Dieulafoy&#x00027;s lesion: a systematic review of evaluation, diagnosis, and management</article-title>
<source>J Investig Med High Impact Case Rep</source>
<year>2021</year>
<volume>9</volume>
<fpage>2324709620987703</fpage>
</element-citation></ref>
<ref id="b7-ce-2023-231">
<label>7</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name><surname>Badwal</surname><given-names>S</given-names></name>
<name><surname>Jain</surname><given-names>M</given-names></name>
<name><surname>Rastogi</surname><given-names>A</given-names></name>
<etal/>
</person-group>
<article-title>Dieulafoy disease of the stomach presenting as mass lesion: a case report</article-title>
<source>Indian J Pathol Microbiol</source>
<year>2005</year>
<volume>48</volume>
<fpage>211</fpage>
<lpage>213</lpage>
</element-citation></ref>
<ref id="b8-ce-2023-231">
<label>8</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name><surname>Jeong</surname><given-names>HW</given-names></name>
<name><surname>Kim</surname><given-names>JY</given-names></name>
<name><surname>Kim</surname><given-names>SJ</given-names></name>
<etal/>
</person-group>
<article-title>A case of a Jejunal Dieulafoy&#x00027;s lesion mimicking a submucosal tumor</article-title>
<source>Korean J Gastrointest Endosc</source>
<year>2008</year>
<volume>37</volume>
<fpage>438</fpage>
<lpage>442</lpage>
</element-citation></ref>
<ref id="b9-ce-2023-231">
<label>9</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name><surname>Zhao</surname><given-names>J</given-names></name>
<name><surname>Sun</surname><given-names>Z</given-names></name>
<name><surname>Zhang</surname><given-names>X</given-names></name>
</person-group>
<article-title>A Jejunal Dieulafoy&#x00027;s lesion mimicking a gastrointestinal stromal tumor</article-title>
<source>Clin Gastroenterol Hepatol</source>
<year>2019</year>
<volume>17</volume>
<fpage>A19</fpage>
</element-citation></ref>
<ref id="b10-ce-2023-231">
<label>10</label>
<element-citation publication-type="journal">
<person-group person-group-type="author">
<name><surname>Shrestha</surname><given-names>S</given-names></name>
<name><surname>Pradhan</surname><given-names>S</given-names></name>
<name><surname>Kc</surname><given-names>A</given-names></name>
<etal/>
</person-group>
<article-title>Arteriovenous malformation of the Jejunum, causing massive gastrointestinal bleeding, treated with intraoperative enteroscopy guidance: a case report</article-title>
<source>Cureus</source>
<year>2023</year>
<volume>15</volume>
<fpage>e39940</fpage>
</element-citation></ref>
</ref-list>
<sec sec-type="display-objects">
<title>Figures and Table</title>
<fig id="f1-ce-2023-231" position="float">
<label>Fig. 1.</label><caption><p>Push enteroscopy reveals a 2.5 cm round subepithelial mass with a large central defect covered by an adherent clot in the proximal part of the jejunum.</p></caption>
<graphic xlink:href="ce-2023-231f1.tif"/>
</fig>
<fig id="f2-ce-2023-231" position="float">
<label>Fig. 2.</label><caption><p>(A) Arterial phase computed tomographic angiography shows a round-shaped 2×1.4 cm arterial enhancing lesion in the proximal jejunum (arrow) adjacent to hemostatic clip (*) without active contrast extravasation within bowel loops. (B) The angiography of the jejunal branch of the superior mesenteric artery reveals an accumulation of contrast media at the proximal jejunum (arrow) near the hemostatic clip (*), which is supplied by multiple small jejunal branches. No demonstrable active contrast extravasation is seen in the study.</p></caption>
<graphic xlink:href="ce-2023-231f2.tif"/>
</fig>
<fig id="f3-ce-2023-231" position="float">
<label>Fig. 3.</label><caption><p>(A) The gross lesion reveals a large thrombus within the submucosa. (B) The histopathology reveals a submucosal persistent-caliber artery, dilated with a recent thrombus (*) (hematoxylin and eosin stain, ×10). The overlying mucosal defect (arrow) is connected to the lesion. (C) The vascular wall of the lesion (dotted line) is exceedingly thickened compared to the normal submucosal artery (*) (hematoxylin and eosin stain, ×200).</p></caption>
<graphic xlink:href="ce-2023-231f3.tif"/>
</fig>
<table-wrap id="t1-ce-2023-231" position="float">
<label>Table 1.</label>
<caption><p>Published case reports of Dieulafoy’s lesion resembling a gastrointestinal stromal tumor</p></caption>
<table rules="groups" frame="hsides">
<thead>
<tr>
<th valign="middle">Study</th>
<th valign="middle" align="center">Sex/age (yr)</th>
<th valign="middle" align="center">Comorbidity</th>
<th valign="middle" align="center">Presentation</th>
<th valign="middle" align="center">Location, diameter</th>
<th valign="middle" align="center">Diagnostic methods</th>
<th valign="middle" align="center">Treatment</th>
<th valign="middle" align="center">Outcome</th>
</tr>
</thead>
<tbody>
<tr>
<td valign="top" align="left">Badwal et al.<xref ref-type="bibr" rid="b7-ce-2023-231">7</xref></td>
<td valign="top" align="left">Female/55</td>
<td valign="top" align="left">None</td>
<td valign="top" align="left">Hematemesis</td>
<td valign="top" align="left">Fundus of stomach, 2 cm</td>
<td valign="top" align="left">Esophagogastro-duodenoscopy</td>
<td valign="top" align="left">Surgical resection</td>
<td valign="top" align="left">Uneventful</td>
</tr>
<tr>
<td valign="top" align="left">Jeong et al.<xref ref-type="bibr" rid="b8-ce-2023-231">8</xref></td>
<td valign="top" align="left">Male/32</td>
<td valign="top" align="left">None</td>
<td valign="top" align="left">Hematochezia</td>
<td valign="top" align="left">Proximal jejunum, 1.5 cm</td>
<td valign="top" align="left">Double balloon enteroscopy</td>
<td valign="top" align="left">Surgical resection</td>
<td valign="top" align="left">Uneventful</td>
</tr>
<tr>
<td valign="top" align="left">Zhao et al.<xref ref-type="bibr" rid="b9-ce-2023-231">9</xref></td>
<td valign="top" align="left">Male/41</td>
<td valign="top" align="left">None</td>
<td valign="top" align="left">Hematochezia</td>
<td valign="top" align="left">Proximal jejunum, 1 cm</td>
<td valign="top" align="left">Single balloon enteroscopy</td>
<td valign="top" align="left">Surgical resection</td>
<td valign="top" align="left">Uneventful</td>
</tr>
<tr>
<td valign="top" align="left">This case</td>
<td valign="top" align="left">Male/73</td>
<td valign="top" align="left">Hypertension, diabetes mellitus, ischemic stroke</td>
<td valign="top" align="left">Hematemesis</td>
<td valign="top" align="left">Proximal jejunum, 2.5 cm</td>
<td valign="top" align="left">Push enteroscopy </td>
<td valign="top" align="left">Surgical resection</td>
<td valign="top" align="left">Uneventful</td>
</tr>
</tbody>
</table>
</table-wrap>
</sec>
</back></article>