title: Rupture of hepatic aneurysm complicating hereditary hemorrhagic telangiectasia (Osler-Weber-Rendu disease) for which hepatic arterial coil embolization was effective.
abstract: A 74-year-old woman was transferred to our hospital for further examinations because of abdominal fullness and abnormal levels of serum liver/biliary enzyme persisting for 3 weeks. She had anemia and dilatation of many capillary vessels in her fingers, palms, and tongue in addition to reporting frequent incidences of nasal bleeding in herself and her family. Abdominal ultrasonography detected a cystic lesion in the right hepatic lobe, connected to a dilated tortuous hepatic artery. A low-echoic hepatic phyma was also detected in the back of the cystic lesion. Abdominal computed tomography and magnetic resonance imaging indicated that the cystic lesion was an aneurysm and the low-echoic phyma was a hematoma. Hepatic arteriography confirmed a hepatic aneurysm, tortuous dilatation of the hepatic artery, and the complication of an arteriovenous shunt in the liver. Taking all of these findings into consideration, this case was diagnosed as hereditary hemorrhagic telangiectasia (HTT) complicated by a hepatic aneurysm causing intrahepatic hematoma. To prevent re-rupture of the aneurysm, we performed a hepatic arterial coil embolization. After therapy, no blood flow to the aneurysm was detected by ultrasonic color Doppler method and the hematoma gradually diminished. There have been no reports of a case in which hepatic arterial embolization was effective for HHT-associated hepatic aneurysm causing intrahepatic hematoma. This very rare case provides important clinical information regarding abdominal vascular complications of HTT and a less invasive treatment for them.
authors.affiliation: Department of Gastroenterology, Gifu Prefectural Tajimi Hospital, Tajimi, Japan. kmiyabe@mint.ocn.ne.jp
authors.forename: Katsuyuki
authors.initials: K
authors.lastname: Miyabe
authors.forename: Shinji
authors.initials: S
authors.lastname: Akita
authors.forename: Yasuhiro
authors.initials: Y
authors.lastname: Kitajima
authors.forename: Masaaki
authors.initials: M
authors.lastname: Hirai
authors.forename: Itaru
authors.initials: I
authors.lastname: Naitoh
authors.forename: Kazuki
authors.initials: K
authors.lastname: Hayashi
authors.forename: Tetsu
authors.initials: T
authors.lastname: Okamoto
authors.forename: Yasutaka
authors.initials: Y
authors.lastname: Okayama
authors.forename: Kazuo
authors.initials: K
authors.lastname: Gotoh
authors.forename: Hirotaka
authors.initials: H
authors.lastname: Ohara
authors.forename: Takashi
authors.initials: T
authors.lastname: Joh
authors.forename: Yoshifumi
authors.initials: Y
authors.lastname: Yokoyama
authors.forename: Makoto
authors.initials: M
authors.lastname: Itoh
mesh_terms: D000368:Aged; D017542:Aneurysm, Ruptured / Q000150:complications* / Q000628:therapy*; D004621:Embolization, Therapeutic / Q000379:methods*; D005260:Female; D006499:Hepatic Artery / Q000473:pathology*; D006801:Humans; D018810:Magnetic Resonance Angiography; D013683:Telangiectasia, Hereditary Hemorrhagic / Q000150:complications* / Q000175:diagnosis / Q000628:therapy*; D014057:Tomography, X-Ray Computed