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Case Reports
. 2023 Mar;84(2):504-511.
doi: 10.3348/jksr.2022.0043. Epub 2023 Mar 24.

Rare Case of Large Hemolymphangioma in the Small Bowel Mesentery: A Case Report

Case Reports

Rare Case of Large Hemolymphangioma in the Small Bowel Mesentery: A Case Report

Hyun-Jae Lim et al. J Korean Soc Radiol. 2023 Mar.

Abstract

Hemolymphangioma or hemangiolymphangioma is a rare venolymphatic vascular malformation composed of proliferations or networks of vascular spaces including the lymphatics, capillaries, veins, or arteries. The small bowel is a rare location for hemolymphangioma, and the small bowel mesentery is an even rarer site. Herein, we report a surgically confirmed large complex hemolymphangioma in the small bowel mesentery in a 55-year-old male.

혈관림프종은 혈관종과 림프종 성분으로 구성된 드문 종양으로, 증식된 림프관, 모세혈관, 정맥 또는 동맥과 둘러싸는 결합조직 기질로 구성되어 있다. 소장 기원의 혈관림프종은 매우 드물고 그중 소장 장간막 기원은 극히 드물다. 본 증례 보고에서 수술과 면역 화학 염색을 통하여 확인된 55세 남자 환자의 소장 장간막의 혈관림프종 증례를 보고하고자 한다.

Keywords: Hemangioma; Lymphangioma; Mesentery; Multidetector Computed Tomography; Ultrasonography; Vascular Malformation.

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Conflict of interest statement

Conflicts of Interest: The authors have no potential conflicts of interest to disclose.

Figures

Fig. 1
Fig. 1. A 55-year-old male patient with mesenteric hemolymphangioma, complaining abdominal discomfort.
A. Initial US image shows an approximately 10.0 cm × 8.5 cm sized, multiloculated heterogeneous cystic mass with or without inner hyperechogenic components located in the left mid-abdomen. No definite vascularity of the large mass is seen on color Doppler US. B. Axial and coronal images of contrast-enhanced CT show a 10.0 cm × 8.5 cm, large cystic mass in the mesentery of the ileal loops with loss of the fat plane between the mass and adjacent ileum. The ileum shows luminal narrowing due to the extrinsic compression of the mass, but there is no evidence of obstruction in the small bowel (arrow). Multiple thickened septa show gradual enhancement of the multiloculated main mass, without an enhancing solid mural nodule in delayed phase (right upper). In the coronal image, a visible feeding artery is arising from a branch of the superior mesenteric artery (yellow arrow). Diffusely distributed, multiple variably sized cystic nodules with hazy infiltration are in the small bowel mesentery. In the pre-contrast enhance phase, a locule of the cystic lesion shows an inner fluid-fluid level, suggesting debris, hemorrhage, or proteinaceous material (empty arrow). A tiny calcification, which could be a phlebolith, is in the mesentery (arrowhead). C. The main mass and adjacent small bowel in the operative field. The main mass is located at the border of the ileum and mesentery, and a few dark bloody lesions are also visible (arrow). D. Gross resected cystic mass specimen and adjacent small bowel loops. Dark bloody fluid is visible within the cystic lesion (arrow). Multiple variably sized cysts are visible when resected. Organizing thrombi and serous fluid were found within the cystic lesions. E. The mesenteric hemolymphangioma involves the mucosa and submucosa. The vascular channels are lined by endothelium and filled with RBCs and organizing thrombi (× 40; hematoxylin and eosin staining). The endothelium is positive for CD31 immunohistochemically. In addition, CD34 and D2-40 show partial positivity (× 100). US = ultrasonography

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