Two Cases of Neurofibromatosis Type 1 with Spontaneous Rupture of the Inferior Mesenteric Artery Treated with Transcatheter Arterial Embolization

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  • Iyama Ayumi
    Department of Radiology, National Hospital Organization Kumamoto Medical Center
  • Negishi Takanori
    Department of Radiology, National Hospital Organization Kumamoto Medical Center
  • Iwashita Koya
    Department of Radiology, National Hospital Organization Kumamoto Medical Center
  • Tanoue Shota
    Department of Radiology, National Hospital Organization Kumamoto Medical Center
  • Sugihara Hidetaka
    Department of Surgery, National Hospital Organization Kumamoto Medical Center
  • Karino Kohei
    Department of Emergency, National Hospital Organization Kumamoto Medical Center
  • Yoshimatsu Shunji
    Department of Radiology, National Hospital Organization Kumamoto Medical Center

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Other Title
  • 塞栓術が有用であった神経線維腫症1型に合併した下腸間膜動脈破綻の2例

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Abstract

Neurofibromatosis type 1 (NF-1) is infrequently associated with vascular lesions. We here report two patients with NF-1 who developed retroperitoneal bleeding caused by spontaneous rupture of the inferior mesenteric artery (IMA) and were successfully treated with transcatheter arterial embolization (TAE). Case 1 was a man in his 70s with NF-1 who presented with low back pain of sudden onset. Contrast-enhanced computed tomography (CT) and angiography showed a pseudoaneurysm and extravasation of contrast medium from the superior rectal artery. Embolization was performed using coils and n-butyl-2 cyanoacrylate (NBCA). Case 2 was a woman in her 60s with NF-1 who presented with low back pain and loss of consciousness. Contrast-enhanced CT and angiography showed a pseudoaneurysm at the bifurcation of the left colic branch. Embolization was performed using coils. In both cases, the hemorrhage was successfully terminated by TAE without complications. Non-traumatic vascular collapse caused by rupture of the IMA is extremely rare. It is important to keep in mind that underlying diseases such as NF-1 can be associated with fragile vessel walls. TAE may be the treatment of choice for arterial complications in patients with NF-1. An extremely careful catheter approach is necessary in patients with fragile vessel walls.

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