title: Successful lung transplantation in a case with diffuse pulmonary arteriovenous malformations and hereditary hemorrhagic telangiectasia.
abstract: Diffuse pulmonary arteriovenous malformations (AVMs) are associated with a poor prognosis and the therapeutic strategy remains controversial. We describe a pediatric patient with diffuse pulmonary AVMs associated with hereditary hemorrhagic telangiectasia (HHT), who presented with two cerebral AVMs in the parietal and occipital lobes as well. Of note, successful bilateral lung transplantation not only improved the hypoxemia but also resulted in size reduction of the cerebral AVMs. Although it is essential to consider involvements other than pulmonary AVMs, especially brain AVMs, to decide the indication, lung transplantation can be a viable therapeutic option for patients with diffuse pulmonary AVMs and HHT.
authors.affiliation: Department of Pediatrics, School of Medicine, Keio University, Tokyo, Japan.
authors.forename: H
authors.initials: H
authors.lastname: Fukushima
authors.forename: T
authors.initials: T
authors.lastname: Mitsuhashi
authors.forename: T
authors.initials: T
authors.lastname: Oto
authors.forename: Y
authors.initials: Y
authors.lastname: Sano
authors.forename: K F
authors.initials: KF
authors.lastname: Kusano
authors.forename: K
authors.initials: K
authors.lastname: Goto
authors.forename: M
authors.initials: M
authors.lastname: Okazaki
authors.forename: H
authors.initials: H
authors.lastname: Date
authors.forename: Y
authors.initials: Y
authors.lastname: Kojima
authors.forename: H
authors.initials: H
authors.lastname: Yamagishi
authors.forename: T
authors.initials: T
authors.lastname: Takahashi
mesh_terms: D000293:Adolescent; D001165:Arteriovenous Malformations / Q000150:complications* / Q000628:therapy; D005260:Female; D006801:Humans; D002538:Intracranial Arteriovenous Malformations / Q000150:complications; D002546:Ischemic Attack, Transient / Q000150:complications; D008171:Lung Diseases / Q000150:complications* / Q000628:therapy; D016040:Lung Transplantation; D013683:Telangiectasia, Hereditary Hemorrhagic / Q000150:complications / Q000628:therapy; D016896:Treatment Outcome
keywords: Cerebral arteriovenous malformation; hereditary hemorrhagic telangiectasia; lung transplantation; pulmonary arteriovenous malformation