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<article xml:lang="EN" xmlns:mml="http://www.w3.org/1998/Math/MathML" xmlns:xlink="http://www.w3.org/1999/xlink" article-type="case-report">
<front>
<journal-meta>
<journal-id journal-id-type="publisher-id">Front. Med.</journal-id>
<journal-title>Frontiers in Medicine</journal-title>
<abbrev-journal-title abbrev-type="pubmed">Front. Med.</abbrev-journal-title>
<issn pub-type="epub">2296-858X</issn>
<publisher>
<publisher-name>Frontiers Media S.A.</publisher-name>
</publisher>
</journal-meta>
<article-meta>
<article-id pub-id-type="doi">10.3389/fmed.2022.918748</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Medicine</subject>
<subj-group>
<subject>Case Report</subject>
</subj-group>
</subj-group>
</article-categories>
<title-group>
<article-title>Case Report: Spontaneous Rupture of Hepatic Hemangioma</article-title>
</title-group>
<contrib-group>
<contrib contrib-type="author">
<name><surname>Pan</surname> <given-names>Bing</given-names></name>
</contrib>
<contrib contrib-type="author">
<name><surname>Lyu</surname> <given-names>Shao-Cheng</given-names></name>
<uri xlink:href="http://loop.frontiersin.org/people/1376205/overview"/>
</contrib>
<contrib contrib-type="author" corresp="yes">
<name><surname>He</surname> <given-names>Qiang</given-names></name>
<xref ref-type="corresp" rid="c001"><sup>&#x002A;</sup></xref>
<uri xlink:href="http://loop.frontiersin.org/people/1753402/overview"/>
</contrib>
</contrib-group>
<aff><institution>Department of Hepatobiliary Surgery, Beijing Chaoyang Hospital, Capital Medical University</institution>, <addr-line>Beijing</addr-line>, <country>China</country></aff>
<author-notes>
<fn fn-type="edited-by"><p>Edited by: Christophe Moreno, Universit&#x00E9; Libre de Bruxelles, Belgium</p></fn>
<fn fn-type="edited-by"><p>Reviewed by: Muhammad Hashim Hayat, Vanderbilt University Medical Center, United States; Yuichiro Ikebuchi, Tottori University Hospital, Japan</p></fn>
<corresp id="c001">&#x002A;Correspondence: Qiang He, <email>heqiang349@163.com</email></corresp>
<fn fn-type="other" id="fn004"><p>This article was submitted to Hepatology, a section of the journal Frontiers in Medicine</p></fn>
</author-notes>
<pub-date pub-type="epub">
<day>13</day>
<month>07</month>
<year>2022</year>
</pub-date>
<pub-date pub-type="collection">
<year>2022</year>
</pub-date>
<volume>9</volume>
<elocation-id>918748</elocation-id>
<history>
<date date-type="received">
<day>12</day>
<month>04</month>
<year>2022</year>
</date>
<date date-type="accepted">
<day>21</day>
<month>06</month>
<year>2022</year>
</date>
</history>
<permissions>
<copyright-statement>Copyright &#x00A9; 2022 Pan, Lyu and He.</copyright-statement>
<copyright-year>2022</copyright-year>
<copyright-holder>Pan, Lyu and He</copyright-holder>
<license xlink:href="http://creativecommons.org/licenses/by/4.0/"><p>This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.</p></license>
</permissions>
<abstract>
<sec>
<title>Background</title>
<p>Hepatic hemangioma (HH) is a congenital vasal malformation that seemed like the most probable benign liver neoplasm, composed of masses of blood vessels, which are anomalous in arrangement and size. In most cases, HH is asymptomatic, and patients have an excellent prognosis. According to research, the location and size of the mass are correlated with the symptoms and complications. Reports of spontaneous rupture of HH have been less reported in the literature. In this emergency condition, dynamic contrast-enhanced CT scanning, especially triple-phase computed tomography (CT) with delayed imaging, is preferred.</p>
</sec>
<sec>
<title>Case Presentation</title>
<p>Here, we presented two middle-aged female patients with spontaneous rupture of HH in our hospital. Following an accurate diagnosis of enhanced CT and emergency surgery, patients recovered well and were discharged from the hospital.</p>
</sec>
<sec>
<title>Conclusion</title>
<p>Appropriate imaging studies, especially enhanced CT, and emergency surgery are indispensable for patients with spontaneous rupture of HH. As a surgeon, we need to pay attention to the asymptomatic patient with HH.</p>
</sec>
</abstract>
<kwd-group>
<kwd>hepatic hemangioma</kwd>
<kwd>spontaneous rupture</kwd>
<kwd>surgery</kwd>
<kwd>emergency</kwd>
<kwd>intra-abdominal hemorrhage</kwd>
</kwd-group>
<counts>
<fig-count count="2"/>
<table-count count="1"/>
<equation-count count="0"/>
<ref-count count="22"/>
<page-count count="5"/>
<word-count count="2564"/>
</counts>
</article-meta>
</front>
<body>
<sec id="S1" sec-type="intro">
<title>Introduction</title>
<p>Hepatic hemangioma (HH) is the most probable benign tumor of the liver, and often asymptomatic, can be found accidentally during the imagological examination, has a prevalence of approximately 20%, and is more frequent in women. Clinically, the most common type is a cavernous hemangioma, and most patients have an excellent prognosis because of the benign nature of hemangioma (<xref ref-type="bibr" rid="B1">1</xref>). So, most scholars propose that surgery should be restricted to the specific situation. The incidence of HH with rupture is low (1&#x2013;4%). However, the mortality is rather high. Thus, spontaneous rupture is the most severe complication (<xref ref-type="bibr" rid="B2">2</xref>). There is no scientific evidence correlating the size of the hemangioma with the risk of rupture. In this emergency condition, dynamic contrast-enhanced computed tomography (CT) scanning, especially triple-phase CT with delayed imaging, is preferred. Herein, we presented two patients in our hospital, whose HHs were spontaneously ruptured, and whose hemangiomas are less than 10 cm in diameter. Following an accurate diagnosis of enhanced CT and emergency surgery, patients recovered well and were discharged from the hospital.</p>
</sec>
<sec id="S2">
<title>Case Presentation</title>
<sec id="S2.SS1">
<title>Case 1</title>
<p>A 33-years-old woman was admitted to the emergency department with persistent hurt in the upper quadrant of the abdomen, which had occurred suddenly, and the pain was not relieved after resting or changing position. She denied a history of any medical condition (including hepatitis), recent trauma, or oral contraceptive use, and a family history of hepatic disease. On admission physical examination (PE), the following results are observed: temperature (T) is 36.5&#x00B0;C, heart rate (HR) 75 beats/min, respiratory rate (RR) is 20 times/min, and blood pressure (BP) is 110/75 mmHg. Tenderness in the right upper quadrant of the abdomen was noted. Cardiopulmonary function examination showed no obvious abnormalities. Laboratory examination results showed a hemoglobin (Hb) level of 98 g/L (normal range: 120&#x2013;160 g/L). The rest of the laboratory tests, including liver function and coagulation function, are normal. The abdominal enhanced-CT result (<xref ref-type="fig" rid="F1">Figure 1A</xref>) showed a large round shape with a slightly lower confounding density shadow in the right hepatic. During the venous phase and the delayed phase, the enhancement is obvious, showing a tendency for inward filling, the local capsule is blurred, and a small amount of effusion is seen around the liver. According to the imaging study, we considered a diagnosis of rupture and hemorrhage of HH. Considering the threat of further rupture of the hemangioma, the patient underwent an emergency exploratory operation. During the operation, a dark red mass of about 10 cm was seen in the right liver, which was tough in quality. The mass slightly adheres to the diaphragm and lateral abdominal wall, and a small amount of hemorrhage can be seen around the liver. So, HH resection was performed. The findings of pathology indicated rupture hemorrhage of HH (<xref ref-type="fig" rid="F1">Figure 1B</xref>). The patient recovered well (<xref ref-type="fig" rid="F1">Figure 1C</xref>) and was discharged 5 days post-surgery.</p>
<fig id="F1" position="float">
<label>FIGURE 1</label>
<caption><p>Case 1: <bold>(A)</bold> Pre-operative imaging examination, <bold>(B)</bold> pathological findings, and <bold>(C)</bold> post-operative imaging review.</p></caption>
<graphic mimetype="image" mime-subtype="tiff" xlink:href="fmed-09-918748-g001.tif"/>
</fig>
</sec>
<sec id="S2.SS2">
<title>Case 2</title>
<p>A 36-year-old woman was admitted to the clinic with a chief complaint of &#x201C;sudden right the upper quadrant pain for 14 h.&#x201D; PE indicated abdominal pain, especially in the upper right abdomen. There was no obvious muscle tension and rebound pain. The results of the laboratory were normal. The enhanced abdominal CT said a mass in the right liver lobe, which was a patchy progressive enhancement shadow, a fusiform shape was visible in the adjacent liver capsule, and there was a small amount of fluid around the liver (<xref ref-type="fig" rid="F2">Figure 2A</xref>). The patient underwent exploratory laparotomy followed by liver segmentectomy. The pathology results revealed rupture hemorrhage of HH (<xref ref-type="fig" rid="F2">Figure 2B</xref>). The post-operative outcome was favorable (<xref ref-type="fig" rid="F2">Figure 2C</xref>).</p>
<fig id="F2" position="float">
<label>FIGURE 2</label>
<caption><p>Case 2: <bold>(A)</bold> Pre-operative imaging examination, <bold>(B)</bold> pathological findings, and <bold>(C)</bold> post-operative imaging review.</p></caption>
<graphic mimetype="image" mime-subtype="tiff" xlink:href="fmed-09-918748-g002.tif"/>
</fig>
</sec>
</sec>
<sec id="S3" sec-type="discussion">
<title>Discussion</title>
<p>Hepatic hemangioma is the most probable benign neoplasm and is often found in women. The majority of HH is a cavernous hemangioma, with little chance of malignant transformation (<xref ref-type="bibr" rid="B3">3</xref>). After improving people&#x2019;s health consciousness, more and more patients with hemangioma have been found. HHs grow slowly, and the course often lasts for many years. In most cases, the diameter of HH (&#x003C;4 cm) is usually asymptomatic and easy to ignore, being discovered only as an incidental imaging finding. At &#x003E;4-cm diameter, HHs are considered gigantic in size and might result in symptoms, such as abdominal discomfort, rupture, internal hemorrhage, coagulation disorder, etc. (<xref ref-type="bibr" rid="B4">4</xref>).</p>
<p>Reports of HH with spontaneous hemorrhage are less, causing a misunderstanding and ignorance of its serious complications (<xref ref-type="bibr" rid="B5">5</xref>). The incidence of abdominal bleeding caused by ruptured HH is low (1&#x2013;4%), the mortality ranges from 60 to 70% (<xref ref-type="bibr" rid="B2">2</xref>), and the surgery mortality rate from this complication is 36.4% (<xref ref-type="bibr" rid="B6">6</xref>). There has been no scientific evidence correlating the size of the hemangioma with the risk of rupture. The first spontaneous rupture case of HH was reported by Van Haefen in 1898 (<xref ref-type="bibr" rid="B7">7</xref>). Yamamoto et al. documented 28 cases of ruptured tumors, with sizes ranging from 3 to 25 cm (<xref ref-type="bibr" rid="B8">8</xref>). To more comprehensively understand this disease, we searched PubMed and found 13 cases reported in recent 20 years (<xref ref-type="table" rid="T1">Table 1</xref>). These previous reports, combined with our case report, will significantly contribute to the diagnosis and treatment of spontaneous rupture of HH.</p>
<table-wrap position="float" id="T1">
<label>TABLE 1</label>
<caption><p>Other case reports of spontaneous rupture of hepatic hemangioma (HH) reported in PubMed in recent 20 years.</p></caption>
<table cellspacing="5" cellpadding="5" frame="hsides" rules="groups">
<thead>
<tr>
<td valign="top" align="left">Age</td>
<td valign="top" align="left">Gender</td>
<td valign="top" align="left">Size<break/> (cm)</td>
<td valign="top" align="left">Hemorrhage type</td>
<td valign="top" align="left">Management</td>
<td valign="top" align="left">outcome</td>
<td valign="top" align="left">Author</td>
<td valign="top" align="left">Title</td>
<td valign="top" align="left">Journal</td>
<td valign="top" align="left">year</td>
</tr>
</thead>
<tbody>
<tr>
<td valign="top" align="left">37</td>
<td valign="top" align="left">Female</td>
<td valign="top" align="left">15&#x00D7;12</td>
<td valign="top" align="left">Intratumor bleed</td>
<td valign="top" align="left">Hepatectomy</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Zhao (<xref ref-type="bibr" rid="B12">12</xref>)</td>
<td valign="top" align="left">Spontaneous rupture of hepatic hemangioma: a case report and literature review</td>
<td valign="top" align="left">Int J Clin Exp Pathol</td>
<td valign="top" align="left">2015</td>
</tr>
<tr>
<td valign="top" align="left">56</td>
<td valign="top" align="left">Female</td>
<td valign="top" align="left">10&#x00D7;6</td>
<td valign="top" align="left">Extratumor bleed</td>
<td valign="top" align="left">Hepatectomy</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Zhai (<xref ref-type="bibr" rid="B13">13</xref>)</td>
<td valign="top" align="left">Spontaneous rupture of giant hepatic hemangioma: misdiagnosis as gastrointestinal perforation</td>
<td valign="top" align="left">J Int Med Res</td>
<td valign="top" align="left">2019</td>
</tr>
<tr>
<td valign="top" align="left">35</td>
<td valign="top" align="left">Female</td>
<td valign="top" align="left">12&#x00D7;10</td>
<td valign="top" align="left">Extratumor bleed</td>
<td valign="top" align="left">TACE</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Cao (<xref ref-type="bibr" rid="B14">14</xref>)</td>
<td valign="top" align="left">A case of spontaneous hepatic hemangioma rupture: Successful management with transarterial chemoembolization alone</td>
<td valign="top" align="left">J Interv Med</td>
<td valign="top" align="left">2019</td>
</tr>
<tr>
<td valign="top" align="left">65</td>
<td valign="top" align="left">Male</td>
<td valign="top" align="left">11&#x00D7;8</td>
<td valign="top" align="left">Intratumor bleed</td>
<td valign="top" align="left">Hepatectomy</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Yang (<xref ref-type="bibr" rid="B15">15</xref>)</td>
<td valign="top" align="left">Spontaneous intracapsular hemorrhage of a giant hepatic cavernous hemangioma: a rare case report and literature review</td>
<td valign="top" align="left">BMC Gastroenterol</td>
<td valign="top" align="left">2021</td>
</tr>
<tr>
<td valign="top" align="left">25</td>
<td valign="top" align="left">Male</td>
<td valign="top" align="left">12&#x00D7;9</td>
<td valign="top" align="left">Extratumor bleed</td>
<td valign="top" align="left">Hepatectomy</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Gupta (<xref ref-type="bibr" rid="B16">16</xref>)</td>
<td valign="top" align="left">Spontaneous rupture of a giant hepatic hemangioma-report of a case</td>
<td valign="top" align="left">Indian J Surg</td>
<td valign="top" align="left">2012</td>
</tr>
<tr>
<td valign="top" align="left">76</td>
<td valign="top" align="left">Female</td>
<td valign="top" align="left">9&#x00D7;9</td>
<td valign="top" align="left">Extratumor bleed</td>
<td valign="top" align="left">Hepatectomy</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Nguyen (<xref ref-type="bibr" rid="B17">17</xref>)</td>
<td valign="top" align="left">Giant hepatic hemangioma rupture in a patient on direct oral anticoagulant therapy</td>
<td valign="top" align="left">J Surg Case Rep</td>
<td valign="top" align="left">2021</td>
</tr>
<tr>
<td valign="top" align="left">46</td>
<td valign="top" align="left">Female</td>
<td valign="top" align="left"><xref ref-type="table-fn" rid="t1fns1">&#x002A;</xref></td>
<td valign="top" align="left"><xref ref-type="table-fn" rid="t1fns1">&#x002A;</xref></td>
<td valign="top" align="left">Untreated</td>
<td valign="top" align="left">Dead</td>
<td valign="top" align="left">Bel Hadj et al. (<xref ref-type="bibr" rid="B5">5</xref>)</td>
<td valign="top" align="left">Spontaneous rupture of a hepatic cavernous hemangioma: A rare case of sudden unexpected death</td>
<td valign="top" align="left">Am J Forensic Med pathol</td>
<td valign="top" align="left">2020</td>
</tr>
<tr>
<td valign="top" align="left">64</td>
<td valign="top" align="left">Male</td>
<td valign="top" align="left">5</td>
<td valign="top" align="left">Extratumor bleed</td>
<td valign="top" align="left">TACE</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Rossi (<xref ref-type="bibr" rid="B18">18</xref>)</td>
<td valign="top" align="left">Spontaneous hepatic hemangioma rupture and hemoperitoneum: a double problem with a single stage interventional radiology solution</td>
<td valign="top" align="left">Clin Exp Emerg Med</td>
<td valign="top" align="left">2019</td>
</tr>
<tr>
<td valign="top" align="left">52</td>
<td valign="top" align="left">Female</td>
<td valign="top" align="left">16</td>
<td valign="top" align="left">Intratumor bleed</td>
<td valign="top" align="left">Hepatectomy</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Hao (<xref ref-type="bibr" rid="B19">19</xref>)</td>
<td valign="top" align="left">Spontaneous internal hemorrhage of a giant hepatic hemangioma: A case report</td>
<td valign="top" align="left">Medicine (Baltimore)</td>
<td valign="top" align="left">2017</td>
</tr>
<tr>
<td valign="top" align="left">31</td>
<td valign="top" align="left">Male</td>
<td valign="top" align="left">9.7&#x00D7;7.3</td>
<td valign="top" align="left">Extratumor bleed</td>
<td valign="top" align="left">TACE</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Jain et al. (<xref ref-type="bibr" rid="B2">2</xref>)</td>
<td valign="top" align="left">Spontaneous rupture of a giant hepatic hemangioma&#x2013;sequential management with transcatheter arterial embolization and resection</td>
<td valign="top" align="left">Saudi J Gastroenterol</td>
<td valign="top" align="left">2010</td>
</tr>
<tr>
<td valign="top" align="left">25</td>
<td valign="top" align="left">Female</td>
<td valign="top" align="left"><xref ref-type="table-fn" rid="t1fns1">&#x002A;</xref></td>
<td valign="top" align="left">Extratumor bleed</td>
<td valign="top" align="left">Hepatectomy</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Santos Rodrigues (<xref ref-type="bibr" rid="B20">20</xref>)</td>
<td valign="top" align="left">Spontaneous rupture of giant hepatic hemangioma: a rare source of hemoperitoneum. Case report</td>
<td valign="top" align="left">G Chir</td>
<td valign="top" align="left">2010</td>
</tr>
<tr>
<td valign="top" align="left">54</td>
<td valign="top" align="left">Female</td>
<td valign="top" align="left">4.4&#x00D7;2.8</td>
<td valign="top" align="left">Intratumor bleed</td>
<td valign="top" align="left">Hepatectomy</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Kim (<xref ref-type="bibr" rid="B21">21</xref>)</td>
<td valign="top" align="left">Hemorrhagic hemangioma in the liver: A case report</td>
<td valign="top" align="left">World J Gastroenterol</td>
<td valign="top" align="left">2015</td>
</tr>
<tr>
<td valign="top" align="left">70</td>
<td valign="top" align="left">Male</td>
<td valign="top" align="left"><xref ref-type="table-fn" rid="t1fns1">&#x002A;</xref></td>
<td valign="top" align="left">Extratumor bleed</td>
<td valign="top" align="left">Peritoneal drainage</td>
<td valign="top" align="left">Survival</td>
<td valign="top" align="left">Goidescu (<xref ref-type="bibr" rid="B22">22</xref>)</td>
<td valign="top" align="left">Ruptured liver cavernous hemangioma-rare cause of hemoperitoneum</td>
<td valign="top" align="left">J Med Life</td>
<td valign="top" align="left">2015</td>
</tr>
</tbody>
</table>
<table-wrap-foot>
<fn id="t1fns1"><p><italic>&#x002A;Represent that there is no detailed description in the original text.</italic></p></fn>
</table-wrap-foot>
</table-wrap>
<p>Clinical manifestation of spontaneous rupture of HH consists of sudden abdominal pain, anemia that is secondary to a hemoperitoneum, and disseminated intravascular coagulopathy (DIC). Spontaneous rupture of HH is considered a life-threatening condition (<xref ref-type="bibr" rid="B9">9</xref>), as its clinical signs are not usually specific. Dynamic contrast-enhanced CT scanning, especially triple-phase CT with delayed imaging, is preferred. Conservative treatment may result in hypovolemic shock, and emergent hepatic resection should be applied, although high operative mortality (<xref ref-type="bibr" rid="B10">10</xref>). Thus, emergency surgery is needed for patients with clinical symptoms, and high-risk complications should be more actively treated.</p>
</sec>
<sec id="S4" sec-type="conclusion">
<title>Conclusion</title>
<p>At present, due to a poor understanding of the natural history of asymptomatic hemangiomas (<xref ref-type="bibr" rid="B11">11</xref>), patients might have died unexpectedly when hemangioma ruptured into the abdominal cavity without surgical treatment. An accurate diagnosis of a hemangioma as the cause of a hemoperitoneum would result in correct clinical decision-making and treatment. Thus, enhanced CT and emergency surgery are necessary for ruptured or bleeding HHs. We hope that our cases will attract more attention to this complication in clinical work. It requires us to reconsider indications for surgery.</p>
</sec>
<sec id="S5" sec-type="data-availability">
<title>Data Availability Statement</title>
<p>The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.</p>
</sec>
<sec id="S6">
<title>Ethics Statement</title>
<p>Written informed consent was obtained from the patient for the publication of this case report. Written informed consent was obtained from the patient for the publication of any potentially identifiable images or data included in this article.</p>
</sec>
<sec id="S7">
<title>Author Contributions</title>
<p>BP and S-CL contributed to the planning and organization, collected clinical data, supervised the findings of this work, analyzed the results, and prepared the manuscript. QH aided in the data collection and supervision. All authors contributed to the article and approved the submitted version.</p>
</sec>
<sec id="conf1" sec-type="COI-statement">
<title>Conflict of Interest</title>
<p>The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.</p>
</sec>
<sec id="pudiscl1" sec-type="disclaimer">
<title>Publisher&#x2019;s Note</title>
<p>All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.</p>
</sec>
</body>
<back>
<ref-list>
<title>References</title>
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