Korean J Gastroenterol.  2015 Sep;66(3):164-167. 10.4166/kjg.2015.66.3.164.

Life-threatening Duodenal Ulcer Bleeding from a Ruptured Gastroduodenal Artery Aneurysm in a Patient with Neurofibromatosis Type 1

• Im KS
• Kim S
• Lim JU
• Jeon JW
• Shin HP
• Cha JM
• Joo KR
• Lee JI
• Park JJ

• KMID: 2373333
• DOI: http://doi.org/10.4166/kjg.2015.66.3.164

Abstract

Vasculopathy is rarely reported in neurofibromatosis type 1, but when it occurs it primarily involves the aorta and its main branches. Among vasculopathies, aneurysmal dilatation is the most common form. Although several case reports concerning aneurysms or pseudoaneurysms of visceral arteries in neurofibromatosis type 1 patients have been reported, there are no reports describing gastroduodenal artery aneurysms associated with neurofibromatosis type 1. We experienced a case of life-threatening duodenal ulcer bleeding from a ruptured gastroduodenal artery aneurysm associated with neurofibromatosis type 1. We treated our patient by transarterial embolization after initial endoscopic hemostasis. To our knowledge, this is the first reported case of its type. High levels of suspicion and prompt diagnosis are required to select appropriate treatment options for patients with neurofibromatosis type 1 experiencing upper gastrointestinal bleeding. Embolization of the involved arteries should be considered an essential treatment over endoscopic hemostasis alone to achieve complete hemostasis and to prevent rebleeding.

Keyword

Neurofibromatoses; Duodenal ulcer; Hemorrhage; Aneurysm, ruptured; Embolization, therapeutic

MeSH Terms

Adult
Aneurysm/*diagnosis/etiology
Arteries
Embolization, Therapeutic
Gastroscopy
Head and Neck Neoplasms/complications/*diagnosis
Hepatic Artery/diagnostic imaging
Humans
Male
Neurofibromatosis 1/complications/*diagnosis
Peptic Ulcer Hemorrhage/*etiology
Radiography

Figure

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  Fig. 1. Photograph of the patient shows a number of cutaneous neurofibromas on the neck and trunk.

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  Fig. 2. Endoscopy upon admission reveals a small ulcer (3×7 mm) in the duodenal bulb with visible vessel.

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  Fig. 3. Selective angiography of the common hepatic artery shows an aneurysm (arrow) in the mid-portion of the gastroduodenal artery and contrast extravasation into the duodenal lumen before embolization.

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  Fig. 4. Postembolization angiography demonstrates successful embolization and cessation of blood flow into the gastroduodenal artery aneurysm with a metallic coil (arrow).

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  Fig. 5. (A) Upper endoscopy 5 days after transarterial embolization reveals an active duodenal ulcer with the metallic coil at the ulcer base. (B) Follow-up endoscopic view 11 days after transarterial embolization shows the ulcer with regenerating epithelium, and the coil material was not visible.

Reference

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