A Congenital Intercostal Arteriovenous Fistula
A
n asymptomatic intercostal arteriovenous fistula was suspected because of a faint midthoracic intercostal bruit detected during auscultation in a 3-year-old girl with common cold. There was no history of trauma, iatrogenic therapeutic procedures, or congenital abnormality. Multislice contrast-enhanced computed tomography of the thorax revealed an engorged T8 intercostal artery arising from the thoracic aorta, engorged intercostal vein, and engorged intervertebral vein (Figures 1-3; Figures 1 and 3 available at www.jpeds.com). A three dimensional volume-rendered lateral oblique image confirmed the engorgement of the T8 intercostal artery arising from the thoracic aorta and the engorgement of the intercostal vein (Figure 4; available at www.jpeds.com). Based on the imaging and clinical findings, the diagnosis of congenital intercostal arteriovenous fistula was made. The patient underwent uneventful coil embolization. Intercostal arteriovenous fistulas and malformations are rare lesions, and only a few cases have been published.1 Most of them occur secondary to trauma or iatrogenic therapeutic procedures and in association with neurofibromatosis type 1, although a congenital origin is suggested in extremely rare cases.2 Clinical manifestations vary according
to the size and location of the vascular lesions and may include an accidentally detected intercostal bruit, ischemic symptoms, and mass effects from the involved artery or ruptured aneurysm. All symptomatic intercostal arteriovenous fistulas or malformations should be treated with surgery or embolization. The choice between surgical excision and transcatheter embolization depends on the accurate estimation of the number, size, and course of abnormal vessels. Multislice contrast-enhanced computed tomography can provide useful information about the arterial and venous angioarchitecture before and after surgery or embolization for patients with intercostal arteriovenous fistulas or malformations who cannot undergo conventional angiography for diagnosis and follow-up. n Yi-Lan Lin, MD Yu-Peng Liu, MD Department of Radiology Mackay Memorial Hospital Hsinchu Mackay Junior College of Medicine Nursing, and Management
Kun-Shan Cheng, MD Mackay Junior College of Medicine Nursing, and Management Taipei Department of Pediatrics Mackay Memorial Hospital
Yang-Kai Fan, MD Wen-Ko Su, MD Department of Radiology Mackay Memorial Hospital Hsinchu, Taiwan
References
Figure 2. Engorged intercostal vein (arrow).
1. Parashi HS, Bhosle KN, Thakare ND, Sharma A, Potwar SS. Giant congenital intercostal arteriovenous malformation with extensive involvement of chest wall and ribs: surgical experience. Ann Thorac Surg 2013;65:2157-9. 2. Fukuda W, Taniguchi S, Fukuda I. Endovascular treatment of ruptured intercostal arteriovenous fistulas associated with neurofibromatosis type 1. Ann Vasc Dis 2012;5:109-12.
J Pediatr 2015;167:203. 0022-3476/$ - see front matter. Copyright ª 2015 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpeds.2015.04.038
203
THE JOURNAL OF PEDIATRICS
www.jpeds.com
Figure 1. Engorged T8 intercostal artery arising from the thoracic aorta (arrow).
Vol. 167, No. 1
Figure 4. Three dimensional volume-rendered lateral oblique image confirmed the engorgement of the T8 intercostal artery arising from the thoracic aorta (arrows) and the engorgement of the intercostal vein (arrowheads).
Figure 3. Engorged intervertebral vein (arrows). 203.e1
Lin et al
A
n asymptomatic intercostal arteriovenous fistula was suspected because of a faint midthoracic intercostal bruit detected during auscultation in a 3-year-old girl with common cold. There was no history of trauma, iatrogenic therapeutic procedures, or congenital abnormality. Multislice contrast-enhanced computed tomography of the thorax revealed an engorged T8 intercostal artery arising from the thoracic aorta, engorged intercostal vein, and engorged intervertebral vein (Figures 1-3; Figures 1 and 3 available at www.jpeds.com). A three dimensional volume-rendered lateral oblique image confirmed the engorgement of the T8 intercostal artery arising from the thoracic aorta and the engorgement of the intercostal vein (Figure 4; available at www.jpeds.com). Based on the imaging and clinical findings, the diagnosis of congenital intercostal arteriovenous fistula was made. The patient underwent uneventful coil embolization. Intercostal arteriovenous fistulas and malformations are rare lesions, and only a few cases have been published.1 Most of them occur secondary to trauma or iatrogenic therapeutic procedures and in association with neurofibromatosis type 1, although a congenital origin is suggested in extremely rare cases.2 Clinical manifestations vary according
to the size and location of the vascular lesions and may include an accidentally detected intercostal bruit, ischemic symptoms, and mass effects from the involved artery or ruptured aneurysm. All symptomatic intercostal arteriovenous fistulas or malformations should be treated with surgery or embolization. The choice between surgical excision and transcatheter embolization depends on the accurate estimation of the number, size, and course of abnormal vessels. Multislice contrast-enhanced computed tomography can provide useful information about the arterial and venous angioarchitecture before and after surgery or embolization for patients with intercostal arteriovenous fistulas or malformations who cannot undergo conventional angiography for diagnosis and follow-up. n Yi-Lan Lin, MD Yu-Peng Liu, MD Department of Radiology Mackay Memorial Hospital Hsinchu Mackay Junior College of Medicine Nursing, and Management
Kun-Shan Cheng, MD Mackay Junior College of Medicine Nursing, and Management Taipei Department of Pediatrics Mackay Memorial Hospital
Yang-Kai Fan, MD Wen-Ko Su, MD Department of Radiology Mackay Memorial Hospital Hsinchu, Taiwan
References
Figure 2. Engorged intercostal vein (arrow).
1. Parashi HS, Bhosle KN, Thakare ND, Sharma A, Potwar SS. Giant congenital intercostal arteriovenous malformation with extensive involvement of chest wall and ribs: surgical experience. Ann Thorac Surg 2013;65:2157-9. 2. Fukuda W, Taniguchi S, Fukuda I. Endovascular treatment of ruptured intercostal arteriovenous fistulas associated with neurofibromatosis type 1. Ann Vasc Dis 2012;5:109-12.
J Pediatr 2015;167:203. 0022-3476/$ - see front matter. Copyright ª 2015 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpeds.2015.04.038
203
THE JOURNAL OF PEDIATRICS
www.jpeds.com
Figure 1. Engorged T8 intercostal artery arising from the thoracic aorta (arrow).
Vol. 167, No. 1
Figure 4. Three dimensional volume-rendered lateral oblique image confirmed the engorgement of the T8 intercostal artery arising from the thoracic aorta (arrows) and the engorgement of the intercostal vein (arrowheads).
Figure 3. Engorged intervertebral vein (arrows). 203.e1
Lin et al
