536
and retrograde enteroclysis confirmed the stenosis at the same lo-
cation (Fig. 1d).
tumor itself at the intussusception by enteroclysis in our
patient. To our knowledge, imaging findings of intesti-
nal angiosarcomatosis have not been reported previ-
ously. There are a few reports describing those of he-
mangiomatosis. Scafidi et al. reported imaging findings
of gastrointestinal hemangiomatosis. In their review
they described small bowel thickening with marked en-
hancement of the wall on abdominal CT and a diffuse
submucosal filling defect with conventional enteroclysis
[5]. Bank et al. also reported the same findings for in-
testinal hemangiomatosis [6]. Angiosarcomatosis is a
malignant form of hemangiomatosis; we therefore think
the imaging findings of angiosarcomatosis may resem-
ble those of hemangiomatosis.
Surgical treatment was advised for the relief of symptoms for
this patient. Intraoperative examination revealed a jejuno-jejunal
intussusception, 30 cm distal to Treizt's ligament. Furthermore, a
2.8 15-cm reddish flat tumor involving the serosal surface of the
jejuno-jejunal intussusception was identified (Fig. 1e). Pathologic
examination showed that the tumor was characterized by irregular
anastomosing vascular channels lined by abnormal endothelial
cells, and that the tumor had invaded to the muscularis propia
(Fig. 1f). Furthermore, immunohistologic staining was positive for
factor-VIII-related antigen. The final diagnosis was intestinal an-
giosarcomatosis. The other stenosis at the terminal portion of the
ileum revealed MR enteroclysis diagnosed as post-operative ad-
hesion.
Recently, the use of MR hydrography of the small
bowel with heavily T2-weighted images has been re-
ported in cases of bowel obstruction [7, 8]. We per-
Discussion
Angiosarcoma is a rare malignant tumor characterized formed MR enteroclysis with infusion of additional wa-
by proliferation of tumor cells with vascular endothelial ter through a nasojejunal catheter. The initial advantage
features, accounting for only 1±2% of all soft tissue of this method is that the distal bowel loop beyond the
sarcomas [3, 4]. Usually, angiosarcomas arise from the obstruction is well visualized compared with conven-
face and scalp in elderly patients, and intra-abdominal tional enteroclysis using a barium solution. In our pa-
angiosarcomas are extremely rare neoplasms which tient, a distal small bowel loop and another stenosis at
usually arise in the liver, spleen, and other organs. Al- the terminal ileum were clearly visualized. Secondly,
though the precise predisposing factors of this tumor MR enteroclysis requires no radiation exposure,
remain unclear, exposure to vinyl chloride, thorotrast, whereas in conventional enteroclysis there is radiation
arsenic chemotherapy, trauma, long-standing lymph- exposure. Thirdly, it may elucidate bowel imbrication,
oedema, and radiotherapy have been implicated in its since MR imaging can select various slice thicknesses.
pathogenesis [1, 3, 4]. Angiosarcomas usually form
In conclusion, we report a case of intestinal angio-
nodular tumors, but diffuse growth is rare. The diffuse sarcomatosis manifesting as intussusception. None of
infiltrative form has been named angiosarcomatosis. the imaging studies were useful in visualizing the tumor
The clinical presentation of patients with intestinal an- itself, but MR enteroclysis visualized not only a jejuno-
gioformative tumors has been diverse and nonspecific, jejunal intussusception, but also another stenosis at the
frequently characterized by altered bowel function, dis- terminal ileum. Some pathologic conditions, such as
tension, and hemotochezia [2, 3]. The clinical findings in Crohn's disease, tuberculosis, and metastatic tumors af-
this case revealed intussusception, but the tumor itself fecting the small bowel, represent multicentricity. Mag-
could not be identified preoperatively. Since intestinal netic resonance enteroclysis with an infusion of water
angiosarcomatosis arises from the serosal surface of the through a nasojejunal catheter may be useful in reveal-
intestine, the tumor in the early stage may not affect the ing disease distribution throughout the small bowel.
mucosal surface. Hence, it was difficult to visualize the
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