Ruptured visceral artery aneurysms in a patient of neurofibromatosis type 1 (NF-1) successfully treated by endovascular treatment

Naoko Fukushima,Hiroaki Aoki,Katsuhiko Yanaga,Masaichi Ogawa,Kazuhiko Morikawa,Shinsuke Takenaga

doi:10.1186/s40792-020-0791-6

Abstract

BackgroundNeurofibromatosis type 1 (NF-1) is an autosomal dominant disease and arteriovenous abnormalities are a well-recognized complication. There are several case reports of ruptured aneurysms; however, among them, reports of superior pancreaticoduodenal artery (PDA) and superior mesenteric artery (SMA) aneurysms are rare. We experienced the case of ruptured PDA and SMA aneurysms in a patient of neurofibromatosis type I successfully treated by endovascular treatment.Case presentationA 55-year-old woman with NF-1 came to our hospital with abdominal pain and vomiting. Enhanced abdominal computed tomography revealed a hematoma in the retroperitoneum and an aneurysm in the head of the pancreas. Angiography was performed, and a ruptured aneurysm was suspected the periphery of the PDA, and we embolized it using coils. However, on postoperative day 2, the hemoglobin level decreased, and a branch of the SMA was ruptured. She underwent embolization using coils again and discharged on postoperative day 27 without any further hemorrhage.ConclusionsTo our knowledge, this is the first successfully treated case of ruptured SMA and PDA aneurysms in a patient with NF-1.

Highlights

Neurofibromatosis type 1 (NF-1) is an autosomal dominant disease and arteriovenous abnormalities are a well-recognized complication
Neurofibromatosis type 1 (NF-1) is an autosomal dominant disorder affecting one in 3000 people which is characterized by café au lait spots and various features in the bones, eyes, and nervous system
NF-1 is associated with vascular abnormalities, which may be complicated by spontaneous rupture [5]

Summary

Background

Neurofibromatosis type 1 (NF-1) is an autosomal dominant disorder affecting one in 3000 people which is characterized by café au lait spots and various features in the bones, eyes, and nervous system. Vascular abnormalities such as arterial stenosis, aneurysms, arterial compression, and arteriovenous abnormalities have been known [1, 2], of which involvement of renal arteries aneurysms is the most common. Case presentation A 55-year-old woman was brought to our hospital by ambulance for acute abdominal pain and vomiting She had a past medical history of NF-I and schizophrenia.

Discussion

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Funding Not applicable