Benign tumors are a rare cause of gastrointestinal hemorrhage, being responsible for fewer than 1% of all cases [1
] and can present with conditions like volvulus, bleeding, obstruction and/or intussusceptions. Leiomyomas constitute a fraction of these benign lesions. They tend to occur mostly in the jejunum (44%) followed by ileum (37%) and duodenum (19%). Ezinger [2
] distinguished, clinically and pathologically, three main groups of leiomyomas, namely: superficial leiomyomas, vascular leiomyomas or angioleiomyomas and deep leiomyomas.
Vascular leiomyomas are well-defined benign smooth muscle tumors with prominent abnormal thick walled venous channels [2
]. The presence of vascular leiomyomas in the gastrointestinal tract is extremely rare but a few cases have been reported where they presented as volvulus [3
], peritonitis [4
] and perforation of the intestinal tract [5
]. To the best of our knowledge this is the first English language case report where angioleiomyoma has presented primarily with intestinal bleeding. As the investigation for GI bleeding normally involves OGD and/or colonoscopy, these lesions are difficult to localize and this was our experience. Cross sectional imaging with CT or MRI can be used to detect mass lesions and delineate the anatomical extent. Mesenteric angiography is thus the investigation of choice in cases where OGD and colonoscopy do not reveal the source of bleeding. However definitive diagnosis is histological.
Four histological subtypes of angioleiomyomas [6
] have been described namely:
• Capillary or solid angioleiomyomas having a rich smooth muscle cell stratification surrounding and holding a few thin vascular channels,
• Venous angioleiomyomas characterized by more numerous and thicker vascular channels than found in capillary angioleiomyomas,
• Cavernous angioleiomyomas having widened vascular channels surrounded by a thin layer of smooth muscle cells,
• Combined capillary and venous angioleiomyomas
Our patient had a mixed vascular angioleiomyomas with positive immunohistochemical staining with Desmin for smooth muscle cells. However, staining with Desmin is not necessary for such tumors as suggested by Hasegawa [8
], Lundgren [9
] and Matsuyama [10
]. This characteristic is attributable to the presence of aberrant smooth muscle cells. According to Matsuyama and colleagues, expression of desmin in vascular smooth muscle cells also varies according to the anatomical site, the layer in the vascular wall, the kind or the size of the blood vessel, and the cellular condition such as in contractile or synthetic states [11
One peculiar feature of the tumor found in our patient was its large size measuring 7 × 7 cm. Vascular leiomyomas are usually < 2 cm [14
] although in exceptional cases they can reach large sizes because they are painless and therefore only diagnosed at a late stage.
The operative management is usually surgical involving resection of the affected bowel segment with end-to-end anastomosis as was done in our patient. Embolization of the feeding vessel can be performed as suggested by Cho and colleagues [15
] to control exsanguination.