title: An anomalous unilateral single pulmonary vein associated with a bone morphogenetic protein receptor II gene mutation.
abstract: Anomalous unilateral single pulmonary vein (AUSPV), a rare congenital anomaly, is associated with an aberrant course but normal drainage, and resembles arteriovenous malformation (AVM). We treated a 26-year-old man with AUSPV in the right lung and an anomalous segmental pulmonary vein in the left lung. CT revealed a tortuous vascular shadow with an enhancement pattern identical to that of the pulmonary vein, suggesting AUSPV. This was confirmed by pulmonary angiography. Although pulmonary AVMs were not detected on angiography, microvascular AVMs could not be excluded because delayed bubbles appeared on contrast echocardiography. A genetic examination revealed a missense mutation of BMPR2.
authors.affiliation: Division of Respiratory Medicine, Toho University School of Medicine, Japan.
authors.forename: Kazuya
authors.initials: K
authors.lastname: Koyama
authors.forename: Go
authors.initials: G
authors.lastname: Sano
authors.forename: Yoshinobu
authors.initials: Y
authors.lastname: Hata
authors.forename: Nobuyuki
authors.initials: N
authors.lastname: Shiraga
authors.forename: Hiroki
authors.initials: H
authors.lastname: Ota
authors.forename: Keishi
authors.initials: K
authors.lastname: Sugino
authors.forename: Kazutoshi
authors.initials: K
authors.lastname: Isobe
authors.forename: Susumu
authors.initials: S
authors.lastname: Sakamoto
authors.forename: Yujiro
authors.initials: Y
authors.lastname: Takai
authors.forename: Satoshi
authors.initials: S
authors.lastname: Koezuka
authors.forename: Takashi
authors.initials: T
authors.lastname: Makino
authors.forename: Hajime
authors.initials: H
authors.lastname: Otsuka
authors.forename: Fumitomo
authors.initials: F
authors.lastname: Sato
authors.forename: Shuichi
authors.initials: S
authors.lastname: Sasamoto
authors.forename: Akira
authors.initials: A
authors.lastname: Iyoda
authors.forename: Atsuko
authors.initials: A
authors.lastname: Kurosaki
authors.forename: Sakae
authors.initials: S
authors.lastname: Homma
mesh_terms: D000328:Adult; D000792:Angiography; D001165:Arteriovenous Malformations / Q000175:diagnosis / Q000235:genetics* / Q000378:metabolism; D052006:Bone Morphogenetic Protein Receptors, Type II / Q000235:genetics*; D004247:DNA / Q000235:genetics*; D004252:DNA Mutational Analysis; D003937:Diagnosis, Differential; D004452:Echocardiography; D006801:Humans; D008168:Lung / Q000000981:diagnostic imaging; D008297:Male; D009154:Mutation; D011667:Pulmonary Veins / Q000002:abnormalities*; D014057:Tomography, X-Ray Computed
chemical_list: D004247:DNA; C498218:BMPR2 protein, human; D052006:Bone Morphogenetic Protein Receptors, Type II