UCTN – Unusual cases and technical notes E217

Mixed cavernous - of the jejunum: detection by wireless capsule endoscopy

Fig. 1 Case 1. Double-balloon endoscopy showing a large hemi-circumferential , with whitish carpet-like villi and red spots.

A 26-year-old woman (case 1) was refer- red for evaluation of recurrent episodes of melena. Gastroscopy, colonoscopy with Fig. 2 Case 1. a The ileoscopy, and small-bowel computed to- lesion was easily identi- mography (CT) scan were normal. Capsule fied at laparoscopy be- endoscopy (Pill cam SB 2, Given Imaging, cause of its central blu- Yoqneam, Israel) disclosed a lesion with ish appearance, sur- rounded by whitish whitish carpet-like villi and superficial lymphatic tissue. red spots with spontaneous at b Internal aspect of the the proximal jejunum. At double-balloon surgical specimen. enteroscopy (Fujinon, Saitama, Japan) the lesion occupied two-thirds of the lumen (●" Fig. 1). The involved segment was resected by laparoscopy (●" Fig. 2). Microscopy showed a mixed lesion with a central core of dilated cavernous vascular Fig. 3 Case 1. The lesion involving the channels surrounded by dilated lymph mucosa and the sub- vessels (●" Fig. 3). mucosa is a cavernous The diagnosis of a mixed cavernous hemangioma (H) sur- hemangioma-lymphangioma was con- rounded by dilated firmed by immunostaining [1] (●" Fig. 4). cavernous lymphatic A 59-year-old man (case 2) was admitted channels (L). The over- for two episodes of melena. Gastroscopy lying intact mucosa is This document was downloaded for personal use only. Unauthorized distribution is strictly prohibited. thickened by numerous and colonoscopy were normal. Capsule en- lymphangiectasis teroscopy revealed a polypoid lesion cov- (arrowhead). The aster- ered by whitish and red spots at the proxi- isk indicates a focal mal jejunum (●" Fig. 5), which was con- hemorrhage in contact firmed on double-balloon enteroscopy. with the muscularis The patient underwent single-port lapa- propria (hematoxylin roscopy and the involved segment was and eosin staining; original magnification resected. The lesion, 3.5 cm × 7 cm in size, × 20). corresponded to a mixed cavernous hemangioma-lymphangioma. Gastrointestinal cavernous are congenital benign vascular that are usually located in the jejunum. Their endoscopic appearance at enteros-

Mavrogenis G et al. Mixed cavernous hemangioma-lymphangioma of the jejunum… Endoscopy 2011; 43: E217– E218 E218 UCTN – Unusual cases and technical notes

G. Mavrogenis1, D. Coumaros1, N. Lakh- Fig. 4 Case 1. Immu- rib1, C. Renard2, J. P. Bellocq2, J. Leroy3 nochemistry helped 1 to differentiate be- Department of Gastroenterology, tween hemangioma University Hospital, Strasbourg, France and lymphangioma. 2 Department of Histopathology, Factor VIII stained the University Hospital, Strasbourg, France endothelium of the 3 Department of Digestive Surgery, blood channels (left University Hospital, Strasbourg, France side; arrowheads), while D2–40 stained the endothelium of References lymphatic channels 1 Kahn HJ, Bailey D, Marks A. Monoclonal anti- – (right side; arrows) body D2 40, a new marker of lymphatic endothelium, reacts with Kaposi’s (original magnification and a subset of . Mod Pathol × 400). 2002; 15: 434– 440 2 Chen CH, Jones J. Profound iron deficiency anemia caused by a small-intestinal caver- nous hemangioma. Gastrointest Endosc 2009; 69: 1392– 1393 3 Quentin V, Lermite E, Lebigot J et al. Small copy or capsule endoscopy is usually of a bowel cavernous hemangioma: wireless sessile or polypoid, bluish or red lesion capsule diagnosis of a surgical case. Gastro- [2–4]. However, in our two cases, the sur- intest Endosc 2007; 65: 551– 552 4 Willert RP, Chong AK. Multiple cavernous face of the hemangioma was covered by hemangiomas with iron deficiency anemia white spots, suggesting a lymphatic com- successfully treated with double-balloon ponent. The mixed pattern of lymphatic- enteroscopy. Gastrointest Endosc 2008; 67: vascular tissue was confirmed on histo- 765– 766 logical examination. Mixed hemangioma- 5 Sylla P, Deutsch G, Luo J et al. Cavernous, ar- teriovenous, and mixed hemangioma-lym- lymphangioma has been previously de- phangioma of the rectosigmoid: rare causes scribed at the colon and the designation of rectal bleeding-case series and review of of hemangiolymphangioma has been pro- the literature. Int J Colorectal Dis 2008; 23: posed [5]. The images presented here are 653– 658 the first by means of capsule endoscopy and double-balloon enteroscopy. This his- Bibliography DOI 10.1055/s-0030-1256400 Fig. 5 Case 2. Capsule enteroscopy: vascular tological variation should be kept in mind Endoscopy 2011; 43: E217 –E218 lesion with whitish spots resembling lymphan- in the differential diagnosis of vascular © Georg Thieme Verlag KG Stuttgart · New York · giectasis. lesions with lymphangiectasias. ISSN 0013-726X

Endoscopy_UCTN_Code_CCL_1AC_2AB Corresponding author D. Coumaros Competing interests: None IRCAD/EITS University Hospital 1 Place de l’Hôpital 67091 Strasbourg France Fax: +333-887-51521 [email protected] This document was downloaded for personal use only. Unauthorized distribution is strictly prohibited.

Mavrogenis G et al. Mixed cavernous hemangioma-lymphangioma of the jejunum… Endoscopy 2011; 43: E217 –E218