AJR 2003; 181:867-878
© American Roentgen Ray Society
Extracranial Aneurysms in Children: Practical Classification and Correlative Imaging
Ricardo Restrepo1,
Marilyn Ranson,
Peter G. Chait,
Bairbre L. Connolly,
Michael J. Temple,
Joao Amaral and
Phillip John
1 All authors: Department of Diagnostic Imaging, The Hospital for Sick Children,
555 University Ave., Toronto, ON, Canada M5G 1X8.
Received July 9, 2002;
accepted after revision January 24, 2003.
Address correspondence to M. Ranson.
Presented at the annual meetings of the Society for Pediatric Radiology,
Philadelphia, April 2002, and the European Society of Pediatric Radiology,
Bergen, Norway, June 2002.
Introduction
Although pediatric aneurysms are rare, they are important to recognize
because of the potentially fatal complications. In children, aneurysms are
often related to intrinsic factors rather than a degenerative cause
(atherosclerosis) as in adults. An aneurysm is a localized abnormal dilatation
of any vessel and may be true or false. A true aneurysm is thinning and
stretching of the vessel wall due to weakening of the structural integrity. A
false aneurysm (pseudoaneurysm) is an extravascular hematoma that communicates
with a vessel and is confined by a fibrous capsule
[1]
(Fig. 1).
Imaging of Aneurysms
Evaluation of aneurysms can be performed by noninvasive methods including
sonography, CT, and MR angiography. Color Doppler sonography is helpful for
differentiating and assessing vascular masses. True aneurysms show fusiform or
saccular dilatation of a vessel; sonography is superior to angiography for
showing mural thrombus. Sonographic findings of pseudoaneurysm include flow
within a mass adjacent to a vessel with a small communication between the mass
and the vessel. Characteristic features include swirling color flow with a
"yin-yang" sign and to-and-fro Doppler signal at the neck of the
pseudoaneurysm as the flow enters during systole and exits during diastole
[2].
CT and MR angiography are helpful for revealing the characteristic features
of aneurysms. The advantages of CT and MR angiography over conventional
angiography include a noninvasive technique and the capability to reveal
luminal and mural abnormalities
[3]. Both single-detector
helical CT and multidetector CT (MDCT) can show high levels of uniform
gadolinium enhancement, allowing angiographic evaluation of the major vessels.
MDCT results in superior image quality because of its ability to obtain
thinner sections in a shorter time with greater range of coverage. As a
result, multiplanar two- and three-dimensional images can be reconstructed
with higher resolution and fewer artifacts
[4,
5].
Aneurysms are well delineated on MR angiography, and multiple techniques
may be used. These include black blood, time-of-flight, phase-contrast, and
three-dimensional gadolinium-enhanced imaging. Volume acquisition minimizes
pulsatility artifacts and eliminates slice misregistration. Pseudoaneurysms
may not be evident on time-of-flight or phase-contrast MR angiography as a
result of slow flow; three-dimensional MR angiography performed using
gadolinium is the modality of choice. In addition, gadolinium-enhanced MR
angiography of the entire body can be performed to detect multiple aneurysms
in infants and small children
[6].
Conventional angiography is reserved for specific patients in whom further
information is required about the vascular anatomy and the origin of the
aneurysm or when percutaneous intervention is contemplated. The sensitivity of
digital subtraction angiography is greater than the sensitivity of other
methods, but it is an invasive procedure.
Pseudoaneurysms (False Aneurysms)
Common causes of pseudoaneurysms are trauma, iatrogenesis, or infection.
Traumatic pseudoaneurysms in solid organs are usually diagnosed more than 48
hr after the trauma. Therefore, additional delayed imaging is important when
there is significant injury (Figs.
2A,
2B,
2C and
3A,
3B). In the liver, the
development of pseudoaneurysms is related to extravasated bile, which retards
liver healing and causes clot lysis, perpetuating bleeding (Figs.
4A, and
4B).
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Fig. 2A. —Renal pseudoaneurysm in 14-year-old boy who was injured in
fall. Contrast-enhanced CT scan shows fragment of enhancing kidney and large
perirenal hematoma. Extravasation of hyperdense contrast material from renal
hilum consistent with tear of collecting system is revealed.
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Fig. 2B. —Renal pseudoaneurysm in 14-year-old boy who was injured in
fall. Repeated CT scan obtained 6 days after A shows small, round
enhancing focus in region of renal hilum that was suspected to be
pseudoaneurysm (arrow).
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Fig. 2C. —Renal pseudoaneurysm in 14-year-old boy who was injured in
fall. Anteroposterior selective left renal arteriogram depicts large area of
contained contrast material extravasation that confirms pseudoaneurysm.
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Fig. 3A. —Splenic pseudoaneurysm in 12-year-old boy who was involved in
motor vehicle crash 1 week earlier. Contrast-enhanced CT scan of abdomen shows
two areas of pooling of contrast material (arrows) in spleen, finding
that is consistent with pseudoaneurysms. Note surrounding contusion and
hematoma.
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Fig. 3B. —Splenic pseudoaneurysm in 12-year-old boy who was involved in
motor vehicle crash 1 week earlier. Selective splenic arteriogram shows two
pseudoaneuryms (arrows) in mid pole of spleen.
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Fig. 4A. —Hepatic pseudoaneurysm in 5-year-old boy with history of
congenital immunodeficiency who presented with hypovolemic shock 4 days after
liver biopsy. Transverse sonogram of left lobe of liver shows superficial
lesion with fluid level.
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Fig. 4B. —Hepatic pseudoaneurysm in 5-year-old boy with history of
congenital immunodeficiency who presented with hypovolemic shock 4 days after
liver biopsy. Selective hepatic arteriogram shows focal area of contrast
material accumulation in left lobe of liver, finding that is consistent with
pseudoaneurysm.
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The therapeutic approach for visceral pseudoaneurysms varies according to
the organ involved and the hemodynamic state of the patient. Splenic
pseudoaneurysms are usually managed conservatively because they often resolve
spontaneously. Preservation of splenic tissue is important in children because
of the high risk of postsplenectomy sepsis
[7]. Transcatheter embolization
with coils is an option in hemodynamically unstable patients. Renal and
hepatic aneurysms and pseudoaneurysms are also currently treated with
transcatheter coil embolization, which is a relatively safe and minimally
invasive technique resulting in parenchymal sparing. Embolization should be as
selective as possible to decrease ischemia
[8,
9].
Fractures and exophytic bone lesions such as osteochondromas may result in
the formation of a pseudoaneurysm. The popliteal artery is the most common
location for a pseudoaneurysm resulting from an osteochondroma. This is
because of the high incidence of osteochondroma in this location and the
relative fixation of the popliteal artery by the adductor canal
[10] (Figs.
5A,
5B,
5C, and
5D). Aneurysms caused by
extrinsic compression, such as an osteochondroma, should be treated
surgically. Resection of the osteochondroma is the first step, followed by
resection of the pseudoaneurysm and primary anastomosis or interposition of
the lesion with a graft
[11].
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Fig. 5A. —Popliteal artery pseudoaneurysm resulting from osteochondroma
in 8-year-old boy with multiple hereditary exostoses who presented with
pulsatile mass in popliteal fossa. Lateral radiograph of right knee shows
large, pedunculated osteochondroma of distal femur.
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Fig. 5B. —Popliteal artery pseudoaneurysm resulting from osteochondroma
in 8-year-old boy with multiple hereditary exostoses who presented with
pulsatile mass in popliteal fossa. Sagittal time-of-flight unenhanced MR
angiogram shows mass effect on popliteal artery. No significant flow was shown
in mass.
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Fig. 5C. —Popliteal artery pseudoaneurysm resulting from osteochondroma
in 8-year-old boy with multiple hereditary exostoses who presented with
pulsatile mass in popliteal fossa. Sagittal T1-weighted image with fat
saturation after gadolinium administration depicts small area of enhancement
along posterior aspect of mass (arrow), suggesting
pseudoaneurysm.
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Fig. 5D. —Popliteal artery pseudoaneurysm resulting from osteochondroma
in 8-year-old boy with multiple hereditary exostoses who presented with
pulsatile mass in popliteal fossa. Sagittal color flow Doppler sonogram shows
communication between mass and popliteal artery. Note swirling flow on color
Doppler sonography that is characteristic of pseudoaneurysm (arrow)
and surrounding hematoma (arrowheads).
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Arterial catheterization can produce vascular trauma, particularly when
large intravascular devices are used. The most common complications are
hemorrhage and pseudoaneurysm of the common femoral artery
(Fig. 6). Peripheral
pseudoaneurysms caused by arterial catheterization may be treated with
sonographically guided compression. This technique is noninvasive and is
successful in up to 75% of patients. However, this procedure may be painful
and often requires more than half an hour to achieve thrombosis.
Sonographically guided thrombin injection into the pseudoaneurysm has also
been used to achieve faster thrombosis with less discomfort. There is a risk
of distal thrombin embolization, and therefore this technique should be used
only if the pseudoaneurysm has a narrow neck
[12,
13]. Percutaneous or
transcatheter coil embolization has also been described in the literature
[14].
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Fig. 6. —Common femoral artery pseudoaneurysm after cardiac
catheterization in 1-year-old boy who presented with pulsatile mass in right
groin after cardiac catheterization. Color Doppler sonogram shows vascular
mass adjacent to common femoral artery with associated thrombus
(arrowheads). Swirling arterial flow with color Doppler
characteristic of pseudoaneurysm is present.
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Infectious Aneurysms
Infectious or mycotic aneurysms can be true or false aneurysms, and there
is a high mortality rate as a result of spontaneous rupture. The pathogenesis
in children is similar to that in adults: involving septic emboli cause
endarteritis or hematogenous seeding during episodes of bacteremia
[15]. Pseudoaneurysm after
umbilical artery catheterization is due to a combination of catheter tip
trauma and bacteremia [16]
(Figs. 7A,
7B, and
7C). Direct extension from a
contiguous area of infection and infected sutures may also result in a
pseudoaneurysm (Fig. 8). Other
predisposing conditions include underlying cardiac malformations (Figs.
9A, and
9B) and immunologic compromise
[15].
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Fig. 7A. —Left common iliac artery pseudoaneurysm resulting from
umbilical artery catheter in 8-day-old male neonate with pelvic mass and low
hematocrit level. Abdominal radiograph shows mal-positioned umbilical arterial
catheter with tip in region of left common iliac artery.
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Fig. 7B. —Left common iliac artery pseudoaneurysm resulting from
umbilical artery catheter in 8-day-old male neonate with pelvic mass and low
hematocrit level. Color-flow Doppler sonogram and spectral analysis confirm
vascular nature of mass and show communication with common iliac artery.
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Fig. 7C. —Left common iliac artery pseudoaneurysm resulting from
umbilical artery catheter in 8-day-old male neonate with pelvic mass and low
hematocrit level. Pelvic arteriogram shows contained accumulation of contrast
material (arrows) in midline mass overlying spine, which represents
pseudoaneurysm.
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Fig. 8. —Mycotic pseudoaneurysm in 8-month-old girl who developed
fever 10 days after coarctation repair. Blood cultures were positive for
Staphylococcus aureus bacterium. CT scan of chest shows enhancing
mass anterior to and communicating with dilated ascending aorta (Ao),
consistent with scan pseudoaneurysm (PA). Hypodense rim surrounding
pseudoaneurysm (arrows) represents thrombus.
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Fig. 9A. —Postcoarctation repair of aortic pseudoaneurysm in
4-month-old female infant 1 week after repair. Ao = aortic arch. Coronal
aortic MR angiogram with gadolinium enhancement shows lobulated enhancing mass
arising from aortic arch consistent with pseudoaneurysm (arrows).
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Fig. 9B. —Postcoarctation repair of aortic pseudoaneurysm in
4-month-old female infant 1 week after repair. Ao = aortic arch. Posterior
three-dimensional reconstruction of MR angiogram confirms presence of
pseudoaneurysm (arrows) arising from aortic arch.
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Mycotic aneurysms should be treated with antibiotic therapy and surgery,
but there is some controversy about the timing of surgery. Surgery carried out
immediately lowers the risk of rupture; however, the incidence of graft
infection is high because of the contaminated field. Alternatively, delayed
surgery with aneurysmectomy may be performed after adequate antibiotic
treatment [15].
Inflammatory Aneurysms (Arteritis)
Aneurysms are seen in children with inflammatory arterial disorders such as
Kawasaki disease, polyarteritis nodosa, and Takayasu's and giant cell
arteritis [17]. Other
inflammatory diseases such as Churg-Strauss syndrome, Wegener's
granulomatosis, and collagen vascular diseases may also be associated with
vasculitis and occasionally with aneurysm formation.
Kawasaki disease is a panarteritis usually involving children younger than
5 years old. Coronary artery aneurysms occur in 20-30% of patients but can
also involve the iliac arteries and the abdominal aorta. Follow-up for 2 years
is required because delayed aneurysm formation is possible
[18]. Occasionally, on chest
radiography, a tubular calcification associated with a calcified coronary
artery aneurysm is seen along the left border of the heart (Figs.
10A, and
10B). Two-dimensional
echocardiography represents the standard screening test to detect coronary
aneurysms; however, visualization of the distal coronary arteries is often
limited, requiring ultrafast CT angiography.
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Fig. 10A. —Kawasaki disease in 2-year-old girl with fever, adenopathy,
and rash. Magnified frontal radiograph of pulmonary hila shows tubular
calcification along left heart border consistent with calcified aneurysm
(arrow) of left coronary artery.
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Fig. 10B. —Kawasaki disease in 2-year-old girl with fever, adenopathy,
and rash. Left anterior oblique coronary arteriogram shows giant fusiform
aneurysm (arrow) of proximal segment of left coronary artery.
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Polyarteritis nodosa has a broad age at presentation of 10-80 years, with
most cases occurring in adults. Aneurysm formation is caused by transmural
necrosis, and the number of aneurysms is significantly greater in
polyarteritis nodosa when compared with other types of vasculitis. Findings on
angiography include multiple aneurysms throughout the vascular system but most
commonly in the kidney, liver, and spleen
[19] (Figs.
11A, and
11B). Microaneurysms can
occur, and arteriovenous fistula formation and hemorrhage after renal biopsy
in these patients have been reported
[20].
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Fig. 11A. —Polyarteritis nodosa in 9-year-old boy with hematuria and
elevated creatinine level. Superior mesenteric arteriogram shows small
aneurysm (arrow) arising from branch of ileocolic artery.
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Fig. 11B. —Polyarteritis nodosa in 9-year-old boy with hematuria and
elevated creatinine level. Selective right renal arteriogram shows diffuse
involvement of kidney with multiple microaneurysms of intraparenchymal
branches.
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Takayasu's arteritis can result in arterial stenosis and occlusion and
aneurysm formation. The aneurysms can be multiple and saccular or fusiform in
shape. The disease can affect any aortic segment but most frequently involves
the ascending aorta. Takayasu's arteritis may also involve the major aortic
branches and pulmonary arteries.
In systemic disorders, the initial treatment should be medical to prevent
aneurysm formation. In Kawasaki disease, antiinflammatory therapy, mainly with
acetylsalicylic acid, is indicated in the acute phase. If patients develop
aneurysms, they require antithrombotic treatment to decrease the rate of
complications [21].
Spontaneous rupture may occur, requiring surgical ligation or resection of the
aneurysm.
Connective Tissue Disorders
Connective tissue disorders such as Ehlers-Danlos syndrome and Marfan
syndrome result in increased vessel fragility. Ehlers-Danlos syndrome is rare
but is the most common group of hereditary disorders of connective tissue.
Aneurysms most commonly occur in Ehlers-Danlos syndrome type IV, and the
underlying defect is absent or decreased type III collagen
[17]. Aneurysms are often
multiple and may be fusiform or saccular in shape (Figs.
12A,
12B,
12C and
13A,
13B,
13C). In adults, the aorta and
multiple peripheral branches are usually involved, but in children usually
only the aorta is involved. Noninvasive diagnostic modalities should be used
whenever possible because of vessel fragility. Arteriography may result in
severe complications such as rupture or dissection and is relatively
contraindicated. Surgery is difficult, with a high mortality rate, so
treatment is directed toward prevention of injury
[22,
23].
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Fig. 12A. —16-year-old boy with type IV Ehlers-Danlos syndrome.
Contrast-enhanced CT scan of abdomen shows dilatation of entire abdominal
aorta. Hypodense rim consistent with mural thrombus and calcification of
vessel wall is present.
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Fig. 12B. —16-year-old boy with type IV Ehlers-Danlos syndrome.
Contrast-enhanced aortic MR angiogram shows giant fusiform aneurysm involving
abdominal aorta. In addition, small aneurysm of proximal right common iliac
artery is present.
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Fig. 13B. —Ehlers-Danlos syndrome in 12-year-old boy with sudden onset
of chest pain. Axial CT scan shows large lobulated aneurysm involving long
segment of right subclavian artery (arrows) and associated
mediastinal hematoma (arrowheads).
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Marfan syndrome is an autosomal dominant genetic disease of connective
tissue that can present with aneurysms. An alteration near the fibrillin-1
gene results in cystic medial necrosis with irregular and disorganized elastic
fibers. Classic findings include annuloaortic ectasia, and in children, the
mitral valve may be involved
[17] (Figs.
14A, and
14B). Treatment includes
medical therapy with ß-blockers. If progressive aortic root dilatation is
present, surgery is advocated. Aortic root complications include aortic
insufficiency or dissection. They usually occur during adolescence and
adulthood and are the primary cause of death
[24].
Phakomatoses
Aneurysms may be associated with inherited diseases such as tuberous
sclerosis and neurofibromatosis. Tuberous sclerosis may involve arteries in
the intracranial, thoracic, and abdominal circulation. In tuberous sclerosis,
vascular hamartomatous changes may result in obliteration of the vasa vasorum
causing medial ischemia and subsequent aneurysmal degeneration. Aneurysms,
ectasia, and stenoses are seen on angiography. An aneurysm involving the
entire abdominal aorta as a result of widespread vascular dysplasia has been
reported (Figs. 15A, and
15B). Most renal artery
aneurysms in children are caused by fibromuscular dysplasia or
neurofibromatosis and are often associated with stenoses. Patients usually
present with hypertension [25]
(Fig. 16).
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Fig. 16. —Fibromuscular dysplasia in 14-year-old girl with recently
diagnosed hypertension. Previous renal sonogram (not shown) showed turbulence
in main renal artery. Arteriogram of left renal artery shows tight stenosis
(arrow) associated with distal post-stenotic aneurysm
(arrowhead).
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Metabolic Diseases
Aneurysms related to metabolic diseases are exceedingly rare and have been
reported to be associated with cystinosis and Menkes' syndrome.
Idiopathic or Congenital Aneurysms
Idiopathic or congenital aneurysms usually occur in young children and most
commonly involve the aortoiliac, extremity, and renal arteries. Idiopathic
aneurysms may be a variant of Ehlers-Danlos syndrome that lacks the clinical
skin and joint abnormalities. On the other hand, these aneurysms might be
considered part of a true idiopathic childhood aneurysm
[26].
Acknowledgments
The causes of pediatric aneurysms are varied and may be classified as
traumatic, infectious, inflammatory, hereditary, or metabolic. Cross-sectional
imaging plays an important role in the detection and evaluation of aneurysms,
and conventional angiography is reserved for inconclusive cases or when
interventional therapy is required.
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Introduction
Imaging of Aneurysms
