Ikeda Orito, Shimizu Kunihiko, Yamada Yoshitake, Sugiura Hiroaki, Suzuki Hideaki, Umetsu Syuichiro, Sato Kozo, Jinzaki Masahiro
Department of Radiology, Saiseikai Yokohama-shi Tobu Hospital, Shimosueyoshi 3-6-1, Tsurumi-ku, Yokohama-shi, Kanagawa 230-8765, Japan.
Department of Radiology, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan.
Radiol Case Rep. 2023 Jan 5;18(3):1033-1036. doi: 10.1016/j.radcr.2022.12.024. eCollection 2023 Mar.
Cystic fibrosis is an autosomal recessive genetic disorder that damages the exocrine function of the body, resulting in alterations of multiple organs. In the respiratory system, it is known to cause bronchiectasis, recurrent bronchitis, and pneumonia; however, to the best of our knowledge, there are no reported cases of pulmonary arteriovenous malformations associated with this disease. Herein, we report a case of cystic fibrosis with multiple pulmonary arteriovenous malformations. A 16-year-old girl, who has been monitored since childhood for pancreatitis of unknown cause, experienced respiratory symptoms and hypoxemia (PaO2 = 57 mmHg). At 13 years of age, chest computed tomography revealed bronchiectasis, bronchial wall thickening, and tree-in-bud sign. Genetic testing was performed, and the patient was diagnosed with cystic fibrosis. However, the computed tomography scan also showed incidental nodular lesions in the left superior and both the inferior pulmonary lobes, suggesting multiple arteriovenous malformations. Dynamic computed tomography was performed which, confirmed the presence of 3 pulmonary arteriovenous malformations. Coil embolization was performed on all lesions, and the hypoxemia was corrected. Marked hypoxemia in a patient with cystic fibrosis may not be explained only by the presence of bronchiectasis and/or bronchial wall thickening; in such cases, it may be necessary to examine possible additional findings on computed tomography images, such as arteriovenous malformations.
囊性纤维化是一种常染色体隐性遗传疾病,会损害身体的外分泌功能,导致多个器官发生改变。在呼吸系统中,已知它会引起支气管扩张、复发性支气管炎和肺炎;然而,据我们所知,尚无与该疾病相关的肺动静脉畸形的报道病例。在此,我们报告一例患有多发性肺动静脉畸形的囊性纤维化病例。一名16岁女孩,自幼因不明原因的胰腺炎接受监测,出现了呼吸道症状和低氧血症(动脉血氧分压=57mmHg)。13岁时,胸部计算机断层扫描显示支气管扩张、支气管壁增厚和树芽征。进行了基因检测,该患者被诊断为囊性纤维化。然而,计算机断层扫描还显示左上肺叶和两下肺叶有偶然发现的结节性病变,提示多发性动静脉畸形。进行了动态计算机断层扫描,证实存在3处肺动静脉畸形。对所有病变进行了弹簧圈栓塞,低氧血症得到纠正。囊性纤维化患者出现明显低氧血症可能不能仅用支气管扩张和/或支气管壁增厚来解释;在这种情况下,可能有必要在计算机断层扫描图像上检查是否存在其他可能的发现,如动静脉畸形。