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\begin{document}

\title{Jejunal hemorrhage due to hemolymphangioma successfully detected and
treated by double-balloon enteroscopy}



\author[1]{Yuki Ito}%
\author[1]{Tatsuya Osuga}%
\author[1]{Kentaro Kawasaki}%
\author[1]{Y Ikura}%
\affil[1]{Aijinkai Takatsuki General Hospital}%


\vspace{-1em}



  
  \date{June 18, 2024}


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\selectlanguage{english}
\begin{abstract}
Lymphangiomas are a common benign neoplasm, which can develop in the
digestive tracts. Hemorrhagic jejunal tumors are relatively rare and
diagnostic challenge. We report herein a case of hemorrhagic jejunal
hemolymphangioma successfully diagnosed and treated by double-balloon
enteroscopy.%
\end{abstract}%



\sloppy




\emph{CLINICAL IMAGE}

\textbf{Jejunal hemorrhage due to hemolymphangioma successfully detected
and treated by double-balloon enteroscopy}

Yuki Ito\textsuperscript{1} \textbar{} Tatsuya
Osuga\textsuperscript{1}\textbar{} Kentaro Kawasaki\textsuperscript{2}
\textbar{} Yoshihiro Ikura\textsuperscript{3}

Departments of
\textsuperscript{1}Gastroenterology,\textsuperscript{2}Surgery and
\textsuperscript{3}Pathology, Takatsuki General Hospital,

1-3-13, Kosobecho, Takatsuki 569-1192, Japan

\textbf{Correspondence}

Yoshihiro Ikura, MD, Department of Pathology, Takatsuki General
Hospital, 1-3-13, Kosobecho, Takatsuki 569-1192, Japan.
E-mail:\emph{ikura@ajk.takatsuki-hp.or.jp}

\emph{Fax:} +81 72 682 3834 \emph{Tel:} +81 72 681 3801

\textbf{Running title}

Enteroscopic hemostasis of lymphangioma



\textbf{Abstract}

Lymphangiomas are a common benign neoplasm, which can develop in the
digestive tracts. Hemorrhagic jejunal tumors are relatively rare and
diagnostic challenge. We report herein a case of hemorrhagic jejunal
hemolymphangioma successfully diagnosed and treated by double-balloon
enteroscopy.

\textbf{KEYWORDS}

Intestinal hemorrhage; Hemolymphangioma; Enteroscopic hemostasis

\textbf{Key Clinical Message}

A small jejunal lymphangioma is diagnostic challenge and can evoke
life-threatening hemorrhage. Double-balloon enteroscopy is an useful
tool for its diagnosis and treatment.

\textbf{1 \textbar{} CASE PRESENTATION}

A 74-year-old woman receiving an anticoagulant for atrial fibrillation
was admitted to our hospital because of persistent abdominal pain and
melena. Laboratory test results indicated severe anemia (hemoglobin, 3.0
g/dL). Upper gastrointestinal endoscopy and colonoscopy were performed,
but no hemorrhagic lesion was identified. However, as tarry enteric
fluid flowing out from ileocecal valve was observed in the colonoscopic
examination, we speculated that the bleeding point existed at small
intestin. Double-balloon enteroscopy was performed, and as was expected,
a small (\selectlanguage{ngerman}ø6 mm) polypoid lesion with active bleeding was found at the
jejunum (Fig. 1a). Endoscopic clipping was immediately done for
hemostasis (Fig. 1b). Subsequently, partial jejunectomy was conducted to
prevent re-bleeding. The surgical specimen had a tiny hemorrhagic lesion
(Fig. 2a), which histologically consisted of aggregation of dilated
lympho-vascular channels in the subepithelial region (Fig. 2b). The
final pathologic diagnosis was jejunal hemolymphangioma. To date, no
recurrence of melena has been reported and her hematological data have
been improving (hemoglobin, 8.9 g/dL).

\textbf{2 \textbar{} DISCUSSION AND CONCLUSION}

Lymphangiomas are a common benign neoplasm, which can develop in the
digestive tracts. Bleeding is an important manifestation in usually \selectlanguage{english}[?]20
mm tumors.\textsuperscript{1,2} Our case suggests that lymphangiomas,
even if small, can cause life-threatening gastrointestinal hemorrhage.

\textbf{ACKNOWLEDGMENTS}

None.

\textbf{CONFLICT OF INTEREST}

None.

\textbf{AUTHOR CONTRIBUTIONS}

Y.Ito was responsible for writing the initial draft of the manuscript.
Y.Ikura was responsible for conception, design, drafting, image
modification, and finalizing the manuscript. KK was responsible for
surgery and image modification. TO was responsible for design and
finalizing. All authors read and approved the final manuscript.

\textbf{ETHICAL STATEMENT}

Informed consent for publication and related images has been obtained
from the patient.

\textbf{DATA AVAILABILITY STATEMENT}

No datasets were generated or analyzed during this case report.

\textbf{ORCID}

\emph{Yoshihiro Ikura} (ID) https://orcid.org/0000-0001-8599-2245

\textbf{REFERENCE}

1. Ikura Y, Hashimoto T, Takamine Y, \emph{et al.} Lymphangioma of the
duodenum: report of a case. \emph{Surg Today} 1994;24:160-163.

2. Xiao NJ, Ning SB, Li T, \emph{et al.} Small intestinal
hemolymphangioma treated with enteroscopic injection sclerotherapy: A
case report and review of literature. \emph{World J
Gastroenterol}2020;26:1540-1545.



\textbf{Figure legends}

\textbf{FIGURE 1} Representative images of double-balloon enteroscopy.
(\textbf{a} ) A small polypoid lesion with active bleeding is seen
(arrowheads). (\textbf{b} ) Endoscopic clipping successfully stopped
bleeding.

\textbf{FIGURE 2} Pathologic findings of the surgical specimen.
(\textbf{a} ) A small hemorrhagic polypoid lesion was confirmed in
macroscopic observation (arrowheads). In a cut surface, the hemorrhagic
change is located only at the apex of the lesion (inset). (\textbf{b} )
A microscopic image of the apex portion shows aggregation of dilated
vessels filled with lymphatic fluid and blood accompanied by
extravasation of erythrocytes. (Hematoxylin-eosin stain, x40
magnification)\selectlanguage{english}
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\includegraphics[width=0.70\columnwidth]{figures/Figure-1/Figure-1}

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\includegraphics[width=0.70\columnwidth]{figures/Figure-2/Figure-2}

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