Mesenteric lymphangiomas are rare congenital malformations of the lymphatic system, presenting as non-epithelial, neoplastic proliferations of dilated lymphatic channels [1]. The most common sites of origin are head and neck region or axilla, together contributing >95% of cases [9]. GI lesions account for <0.05% [10]. Definitive diagnosis relies on histologic features, using depth and size of abnormal lymphatics to recognize four tumor subtypes as follows: capillary, cavernous, cystic (hygroma), or hemolymphangioma (mixed vascular/lymphatic elements) [11]. Lesions in this patient corresponded with the latter. According to past reports, lymphangiomas resected for hemostasis have been conspicuous growths [4, 6, 12–14], harboring putative vascular constituents that promote blood loss.
Most mesenteric lymphangiomas (65%) are discovered at birth, and nearly all (90%) are reportedly diagnosed by age 2 [15]. Lymphangiomas of small intestine rarely occur, comprising 1-2% of benign small bowel tumors [16]. Without benefit of enteroscopy, they defy preoperative detection [17]. This case is fairly emblematic, failing to amass conclusive evidence despite longstanding anemia and repeated diagnostic testing. Enteroscopy is thus a worthwhile pursuit in any patient with obscure yet progressive anemia and hypoalbuminemia, bearing lymphangioma in mind.
While generally benign in nature, such tumors may pervade mesenteric roots and peripheries (at intestinal wall) or invade surrounding organs, with potentially life-threatening complications. Traumatic rupture (causing intra-abdominal or intracavitary bleeding and anemia), ischemic tissue necrosis, intestinal gangrene (due to axial torsion), and intermittent intestinal obstruction are possible [18, 19]. GI bleeding was episodic in our patient and difficult to control by medical means. The alternative was surgical resection. However, other published reports [4, 6, 8, 20–22] have warned of the elusiveness posed by bleeding sites amidst multiple lesions.
Ugwonali et al. have used intraoperative enteroscopy to identify and excise sources of hemorrhage in this setting, achieving good results [23]. Morris-Stiff et al. have also installed preliminary tattoos during small bowel endoscopy as flags for resection [7]. Our patient had numerous growths, often adjacent to intestinal wall, limiting the traceability of active bleeding by intraoperative palpation alone. Enteroscopy performed in advance of surgery to pinpoint and tattoo a suspected bleeding site helped expedite intraoperative resection of the culprit segment.
Complete resection of a localized lymphangioma is of course ideal [19, 24]. However, partial resection constitutes a valid approach to multiple lesions, offering substantial volume reduction and serving to improve patient quality of life [25]. Residual neoplastic involvement is a lingering concern that must be addressed through careful follow-up monitoring.
Herein, we describe a patient with multiple mesenteric and intestinal lymphangiomas who experienced chronic episodic bleeding. The situation was resolved through enteroscopic tattoo and laparoscopic-assisted surgical resection. In adults, mesenteric and GI lymphangiomas are rare occurrences to bear in mind as underlying causes of anemia and hypoalbuminemia. Although complete resection is best for symptom improvement, partial resection of identified bleeding points may likewise enhance patient quality of life. Preliminary enteroscopic tattooing is a means of facilitating minimally invasive surgery in these circumstances.