AJR 2005; 184:663-670
© American Roentgen Ray Society
CT of Hemangiomas of the Upper Airways in Children
Benjamin Z. Koplewitz1,
Chaim Springer2,
Benjamin S. Slasky1,
Avraham Avital2,
Kammal Uwyyed2,
Elie Piccard3 and
Jacob Bar-Ziv1
1 Department of Radiology, Hadassah-Hebrew University Medical Center, PO Box
12000, Jerusalem 91120, Israel.
2 Institute of Pulmonology, Hadassah-Hebrew University Medical Center, Jerusalem
91120, Israel.
3 Division of Pediatric Pulmonology, Shaare-Zedek Medical Center, Jerusalem,
Israel.
Received February 7, 2004;
accepted after revision June 1, 2004.
Presented in part in 35th congress of the European Society for Pediatric
Radiology (ESPR), Lugano, Switzerland, May 1997, and in the 104th annual
meeting of the American Roentgen Ray Society (ARRS) in Miami Beach, Florida,
May 2004.
Address correspondence to B. Z. Koplewitz.
Abstract
OBJECTIVE. Hemangiomas of the airways in infants are commonly
diagnosed at bronchoscopy performed for the investigation of stridor or other
respiratory symptoms. Occasionally on bronchoscopy the appearances are
atypical or the entire extent of the suspected hemangioma cannot be
appreciated. We report on the clinical usefulness of dynamic contrast-enhanced
CT in the evaluation of suspected hemangiomas of the airways in infants. CT
findings of 11 infants who underwent investigation for a suspected airway
hemangioma were correlated with bronchoscopic findings.
CONCLUSION. Dynamic contrast-enhanced CT is a valuable noninvasive
method for the evaluation of airway hemangiomas. Although it can be used to
confirm the diagnosis in patients with equivocal findings on bronchoscopy, we
believe that CT findings are specific enough that CT can be recommended as the
primary method of establishing the diagnosis. Multiplanar reconstructions
illustrate the location, extent, and degree of luminal narrowing and any
involvement of adjacent tissues.
Introduction
Hemangioma is the most common benign tumor of the head and neck in infants,
constituting about 1.6% of all congenital laryngeal anomalies
[1]. Hemangiomas are
characterized by a proliferative phase of rapid growth during the first
6–18 months of life, followed by a more gradual involutional phase that
can last up to the age of 10 years. Although present at birth, hemangiomas of
the airway usually become symptomatic between 1 and 6 months of age, when
their rapid growth causes increasing obstruction of the airway
[1–6].
Currently, hemangiomas of the airway in infants are diagnosed on bronchoscopy
as part of the investigation of stridor or other respiratory symptoms.
Hemangiomas usually appear as a purple, blue, or pinkish soft-tissue mass
protruding into the tracheal or laryngeal lumen, most commonly in the
subglottic region
[2–4,6].
Occasionally, however, the appearance at bronchoscopy is atypical, or the
entire extent of the hemangioma cannot be established on bronchoscopy. In such
cases, cross-sectional imaging methods such as CT
[3,
7] and MRI
[8–10]
can yield additional useful information in a relatively noninvasive manner.
The purpose of this article is to present the clinical usefulness and
contribution of dynamic contrast-enhanced CT in the evaluation of suspected
airway hemangiomas in infants.
Materials and Methods
Between January 1, 1996, and December 31, 2002, 11 patients underwent CT as
part of an examination for a suspected upper airway hemangioma. Three patients
were referred for CT because of uncertain bronchoscopic findings, and eight
patients underwent CT to assess the full extent of the lesion and to better
define its characteristics. In three of the latter eight patients the
bronchoscopic findings were highly suggestive of a hemangioma, and in the
other five the bronchoscopic findings were equivocal and the full extent of
the lesion was difficult to determine. Of the 11 patients, four were boys and
seven were girls. All patients presented with inspiratory or biphasic stridor,
and their age at initial presentation ranged between 1 and 6 months.
Bronchoscopy was performed with the patient under general anesthesia (IV
propofol, 2 mg/kg of body weight) or topical anesthesia (1% lidocaine spray)
by one of four experienced pediatric pulmonologists using a pediatric flexible
bronchoscope with an outer diameter of 3.5 mm (Olympus BF 3C20, Olympus BF
XP40, or Pentax FB 10X). Findings were described according to location
(arytenoids, vocal cords, subglottic, tracheal), side (right, left, posterior,
circumferential), and severity of airway obstruction (mild, moderate,
severe).
All CT studies were performed within 2 weeks after the initial bronchoscopy
and were performed with the patient under sedation (oral chloral hydrate, 75
mg/kg; or IV propofol, 2 mg/kg). In six patients, scans were initially
obtained without IV contrast material for better localization and
characterization of the bronchoscopic finding. Two mL/kg of contrast media
(Iopamidol, [iohexol, 300 mg/mL I equivalent], Braaco) was then manually
injected as fast as possible, at an approximate rate of 1.0–1.5 mL/sec,
through a 23- or 25-gauge needle. Scans were obtained using the smallest
possible field of view and 120 kV. In the earlier studies, tube current was
200 mA; this was reduced to as low as 30 mA in the later studies. Different
slice thickness and increments were used as a result of the various CT
scanners used over time. In four patients, 1.2-mm collimation with 1.2-mm
increments was used; in two patients, 2.7-mm collimation with 2.5-mm
increments; and in five patients, 3.2-mm collimation with 1.6-mm increments.
Axial images were acquired directly, and coronal and sagittal reconstructions
were obtained from the raw data. The CT findings were reported by a senior
pediatric radiologist who was aware of the suspicion that a hemangioma was
present in the airway (in 8/11 patients) or unaware of such suspicion (in
3/11). All CT and bronchoscopic studies were then reviewed together by two
pediatric pulmonologists and two pediatric radiologists, findings in both
studies of each patient were correlated, and diagnosis was reached by
consensus.
Results
Between January 1, 1996, and December 31, 2002, 382 children (neonates to 7
years old) underwent 407 bronchoscopies at the Institute of Pulmonology for
stridor or noisy breathing. Six of these children had a suspected hemangioma
of the airways. Three other children were found to have an intraluminal lesion
of uncertain nature and were referred for further evaluation with CT. Two
additional children who had a suspected airway hemangioma at bronchoscopy, and
in whom the diagnosis was also subsequently confirmed on CT, were referred
from another hospital. Table 1 details the sex, age at diagnosis, presenting symptoms, and bronchoscopic and
CT findings of the 11 infants.
Seven patients had isolated subglottic hemangiomas. Four of these were
unilateral. In one patient, the hemangioma was bilateral and larger on the
left side, as seen on bronchoscopy. CT showed a posterolateral,
semicircumferential lesion with moderate narrowing of the lumen, which
correlated closely with the findings at bronchoscopy (Fig.
1A,
1B,
1C). In another patient, the
hemangioma was centrally located on the posterior tracheal wall. In the last
patient, the hemangioma was circumferential, causing severe concentric
narrowing (Fig. 2A,
2B,
2C).
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Fig. 1B. —Right posterolateral subglottic hemangioma in 1-month-old boy
with stridor. CT scan immediately after contrast administration shows intense
enhancement of hemangioma (arrow) and greater degree of luminal
narrowing after contrast administration. Contrast-enhanced study more
optimally shows full extent of lesion, which is semicircumferential, and
correlates more closely with findings at bronchoscopy.
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Fig. 1C. —Right posterolateral subglottic hemangioma in 1-month-old boy
with stridor. Bronchoscopic image shows bilateral nature of hemangioma
(arrows), which is larger on right side, and narrowed trachea
(asterisk). V = vocal cords.
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Fig. 2A. —Symmetric subglottic stenosis (arrows) caused by
circumferential hemangioma in 2-month-old girl with noisy breathing.
Anteroposterior chest radiograph with patient in supine position shows
symmetric bilateral narrowing of the trachea (arrows).
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Fig. 2B. —Symmetric subglottic stenosis (arrows) caused by
circumferential hemangioma in 2-month-old girl with noisy breathing.
Contrast-enhanced axial CT scan shows hemangioma (arrows) with
characteristic intense immediate enhancement after IV contrast injection.
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Fig. 2C. —Symmetric subglottic stenosis (arrows) caused by
circumferential hemangioma in 2-month-old girl with noisy breathing. Image
from bronchoscopy shows bronchoscopic appearance of hemangioma (H). V = vocal
cords, T = narrowed tracheal lumen.
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Three patients had a supraglottic hemangioma. In one of these, the
hemangioma involved the aryepiglottic fold (Fig.
3A,
3B,
3C,
3D). In the second patient,
the supraglottic part of the hemangioma was bilateral and posterior, and the
subglottic part was circumferential, causing severe subglottic stenosis. In
the third patient, the hemangioma extended from the epiglottis inferiorly to
involve the subglottic region (bilaterally) and the proximal trachea (where it
was circumferential) and caused marked tracheal narrowing (Fig.
4A,
4B,
4C,
4D,
4E). This patient was referred
for evaluation because she had only a mild response to low-dose oral steroid
therapy. On CT, the hemangioma was shown to extend obliquely and
semicircumferentially along 12 mm of the mid trachea, resulting in significant
narrowing of the lumen. The patient's symptoms improved after continuous
treatment with high-dose oral steroids, with gradual tapering of the dose.
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Fig. 3B. —24-month-old girl with recurrent episodes of acute stridor.
Contrast-enhanced axial image (B) and coronal (C) and sagittal
(D) reconstructions show soft-tissue mass in right aryepiglottic fold
(arrows) with marked enhancement after contrast administration.
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Fig. 3C. —24-month-old girl with recurrent episodes of acute stridor.
Contrast-enhanced axial image (B) and coronal (C) and sagittal
(D) reconstructions show soft-tissue mass in right aryepiglottic fold
(arrows) with marked enhancement after contrast administration.
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Fig. 3D. —24-month-old girl with recurrent episodes of acute stridor.
Contrast-enhanced axial image (B) and coronal (C) and sagittal
(D) reconstructions show soft-tissue mass in right aryepiglottic fold
(arrows) with marked enhancement after contrast administration.
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Fig. 4A. —Supraglottic hemangioma with subglottic and tracheal
extension in 14-month-old girl with persistent stridor. Bronchoscopic image at
supraglottic level shows marked engorgement of aryepiglottic folds (H). Arrows
= vocal cords, T = tracheal lumen.
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Fig. 4B. —Supraglottic hemangioma with subglottic and tracheal
extension in 14-month-old girl with persistent stridor. Contrast-enhanced
axial CT image at same level as A shows intensely enhancing lesion
(arrows).
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Fig. 4C. —Supraglottic hemangioma with subglottic and tracheal
extension in 14-month-old girl with persistent stridor. Bronchoscopic image at
subglottic level shows asymmetric bilateral subglottic stenosis caused by
tracheal extension of hemangioma (arrows). T = tracheal lumen, V =
vocal cords.
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Fig. 4D. —Supraglottic hemangioma with subglottic and tracheal
extension in 14-month-old girl with persistent stridor. Coronal (D) and
sagittal (E) reconstructed CT images show entire length of lesion and
degree of tracheal narrowing. These images show lesion extending in spiral
manner from subglottic region (arrows) and curving around posterior
and lateral wall of trachea (arrows).
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Fig. 4E. —Supraglottic hemangioma with subglottic and tracheal
extension in 14-month-old girl with persistent stridor. Coronal (D) and
sagittal (E) reconstructed CT images show entire length of lesion and
degree of tracheal narrowing. These images show lesion extending in spiral
manner from subglottic region (arrows) and curving around posterior
and lateral wall of trachea (arrows).
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One patient had a hemangioma at the origin of the left main bronchus that
caused marked bronchial narrowing (mimicking a foreign body) and obstructive
emphysema of the left lung (Fig.
5A,
5B,
5C,
5D).
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Fig. 5A. —Hemangioma in orifice of left main bronchus in 4-month-old
girl that caused one-way valve obstructive effect and simulated clinical and
imaging features of foreign body. Anteroposterior chest radiograph shows
marked hyperinflation of left lung and contralateral mediastinal shift.
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Fig. 5B. —Hemangioma in orifice of left main bronchus in 4-month-old
girl that caused one-way valve obstructive effect and simulated clinical and
imaging features of foreign body. Axial CT scan using soft-tissue window
settings at level of carina shows enhancing soft-tissue mass (arrows)
in orifice of left main bronchus.
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Fig. 5C. —Hemangioma in orifice of left main bronchus in 4-month-old
girl that caused one-way valve obstructive effect and simulated clinical and
imaging features of foreign body. CT scan using lung window settings at same
level as B shows obstructive emphysema of entire left lung secondary to
narrowing of orifice of left main bronchus by hemangioma
(arrows).
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Fig. 5D. —Hemangioma in orifice of left main bronchus in 4-month-old
girl that caused one-way valve obstructive effect and simulated clinical and
imaging features of foreign body. Bronchoscopic image reveals soft-tissue
hemangioma (H) with capillary engorgement bulging into orifice of left main
bronchus.
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When correlated with bronchoscopic findings, CT findings clearly showed in
all patients the presence, nature, and full extent of the lesion. The
contrast-enhanced study showed immediate and intense enhancement, a finding
that was seen in all patients. These characteristic findings established the
diagnosis and proved to be definitive, particularly when the bronchoscopic
findings were uncertain or equivocal. This intense enhancement was shown to
diminish rapidly (within 1–2 min) after the injection of contrast
material in those patients in whom repeated scanning was performed. Another
important feature noted on CT in some patients was that the size of the lesion
was seen to enlarge after the IV administration of contrast material, and the
degree of narrowing was seen to increase compared with that on the unenhanced
CT images (Fig. 1A,
1B,
1C). In three patients, the
diagnosis was not suspected on bronchoscopy and was made solely on the basis
of the findings seen on CT. Coronal and sagittal reconstructions further
improved delineation of the entire extent of the hemangioma and displayed more
clearly the extent and degree of luminal narrowing (Figs.
3A,
3B,
3C,
3D and
4A,
4B,
4C,
4D,
4E).
Discussion
Hemangioma is the most common benign tumor of the head and neck in infants
[1]. The biologic natural
history of the hemangioma consists of an initial proliferative phase, with
rapid growth in size during the first 18 months of life. This is followed by a
more gradual involutional phase that can last until the age of 10 years.
Although present at birth, hemangiomas of the airway characteristically become
symptomatic between the ages of 1 and 6 months because of their rapid growth
at that stage
[1–6].
Clinically, hemangiomas present with sudden onset of acute respiratory
difficulty that varies in severity. Usually the respiratory difficulty
consists of attacks of stridor that typically are episodic and random in
onset. The degree of respiratory distress can fluctuate from day to day or
even from hour to hour. The intermittent nature of the symptoms is directly
related to the degree of distention of the vascular spaces that make up this
lesion. When the hemangioma is located in the infant's airway passages, which
initially are relatively small, the sudden swelling of the hemangioma can
cause varying degrees of airway narrowing up to and including life-threatening
asphyxia. Exercise and excitement frequently exacerbate and intensify the
symptoms. Generally, these lesions are self-limiting; with the growth of the
airway the attacks of respiratory distress tend to diminish and may disappear.
However, the behavior of the individual lesion is unpredictable and the
symptoms are variable in severity.
As shown in our group of patients, the location of the hemangioma along the
airway is variable. Hemangiomas can be present in the supraglottic area (Fig.
3A,
3B,
3C,
3D), in the subglottic region
(Figs. 1A,
1B,
1C,
2A,
2B,
2C, and
4A,
4B,
4C,
4D,
4E), in the trachea (Fig.
4A,
4B,
4C,
4D,
4E), and in the mainstem
bronchus (Fig. 5A,
5B,
5C,
5D). In each of these
locations, the hemangioma has the potential complication of causing
significant, sometimes life-threatening airway obstruction. Consequently, when
a hemangioma is suspected, the diagnosis needs to be established as soon as
possible without waiting for symptoms to recur. The workup includes
radiographic studies, cross-sectional imaging, and direct laryngoscopy or
bronchoscopy. Although the latter will generally establish or confirm the
diagnosis, the endoscopic findings may be inconclusive, especially when the
vessels in the hemangioma are underfilled or collapsed.
In the past, the radiologic diagnosis of a hemangioma was made by
inspiratory anteroposterior and lateral radiography of the neck
[1,
11,
12], using special filters for
better visualization of the upper airways
[12]. It was generally
accepted that an asymmetric narrowing or a soft-tissue shadow bulging into the
tracheal lumen was suggestive of a subglottic hemangioma, whereas symmetric
narrowing is more typical of a congenital subglottic stenosis
[12,
13]. However, symmetric
narrowing of the trachea can be caused by a hemangioma that is located central
to the plane of projection or by a circumferential hemangioma. The latter
diagnosis is more difficult to establish, as noted by Sherrington et al.
[4], who reported 31 infants
with subglottic hemangioma. Those authors found circumferential lesions in
seven of 23 infants for whom such data were available, and in four of six
children bronchoscopic diagnosis was found to be incorrect
[4]. In our series, in two of
the eight patients with subglottic hemangioma, the lesion was located on the
posterior wall of the trachea and did not cause any asymmetry on the
anteroposterior projection of the neck. In two other patients in our series,
the hemangioma was completely circumferential and showed concentric narrowing
of the trachea (Figs. 2A,
2B,
2C and
4A,
4B,
4C,
4D,
4E). In the other four
patients in our series with subglottic hemangioma, the hemangioma was either
lateral or partially circumferential, and it caused asymmetric narrowing in
the anteroposterior projection (Fig.
1A,
1B,
1C). These findings are in
keeping with those of Cooper et al.
[14], who also showed that
symmetric narrowing of the trachea can be a result not only of a congenital
subglottic stenosis but also of a subglottic hemangioma. Thus, the cases
presented in our study clearly illustrate how a hemangioma can cause symmetric
narrowing, asymmetric narrowing, or no narrowing at all in the anteroposterior
plane, according to its location in relation to the airway.
The differential diagnosis of an airway hemangioma in infants is similar to
that of other structural lesions that can cause stridor due to luminal
narrowing. These include laryngomalacia and tracheomalacia, laryngeal
papilloma, granuloma, mucocele, cysts, and lymphangioma
[15–17].
Acquired subglottic stenosis can be sequelae of a viral or a bacterial
infection, of an intubation, or of a foreign body, as initially seemed to be
the case in one of our patients (Fig.
5A,
5B,
5C,
5D). None of these entities,
however, would show as intense enhancement as a hemangioma after IV contrast
injection. This intense enhancement appears to be the result of the rapid
filling of the vessels that make up the hemangioma, indicating a high-flow
lesion. This engorgement can explain the increase in volume of the lesion
after contrast administration in some of our patients and the resulting
increase in the pressure effect and narrowing of the airway (Figs.
1A,
1B,
1C and
2A,
2B,
2C). The high-flow
characteristics also explain the rapid emptying of contrast material in those
patients in whom delayed scanning was performed. These two features do not
appear in any of the other lesions or anomalies causing stridor, and we
consider them to be characteristic for, or at least highly suggestive of, a
hemangioma.
Several technical factors must be stressed when planning the CT examination
of infants with a suspected airway hemangioma. The infant must be sedated and
an IV indwelling catheter (as large as possible) must be placed. If the
location of the lesion is not exactly specified by bronchoscopic findings or
is not well seen in the prescanning review, unenhanced scanning can be
performed initially to define the region of interest; otherwise, this should
not be performed in order to reduce radiation dose. When the study is
performed specifically to investigate a lesion suspected to be a hemangioma,
the area to be scanned can in some cases be limited. We use the smallest
possible field of view with 90–120 kV. In keeping with the
as-low-as-reasonably-achievable (ALARA) principles, the tube current can be
reduced to levels as low as 30 mA and the kilovoltage to as low as 80. These
parameters enable minimizing radiation dose, without significant degradation
of the images obtained following contrast injection. When the suspected lesion
is small, it may be best visualized using very thin slices (i.e., 1 mm);
however, with modern scanners, a slice thickness of 3.2 mm with an increment
of 1.6 or 2 mm gives excellent results and enables good coronal and sagittal
reconstructions (Figs. 3A,
3B,
3C,
3D and
4A,
4B,
4C,
4D,
4E). Once the examination is
planned, contrast material is injected and scanning is immediately begun.
Airway hemangiomas show immediate intense enhancement. However, as we have
already mentioned, this enhancement fades rapidly, and within 1–2 min
after injection, the enhancement may be difficult to discern. Therefore, it is
important to time the scanning to start immediately after the contrast
injection. Although a power injector can be used, in our experience with
infants and small children we have achieved satisfactory results with hand
injection, even through a 23- or 25-gauge needle.
MRI also has been used for the visualization of airway hemangiomas in
infants
[8–10]
and in the differentiation between hemangiomas and vascular malformations
[8,
10]. MRI has the advantages of
avoiding ionizing irradiation and providing excellent soft-tissue resolution
and multiplanar imaging. However, general anesthesia is usually necessary in
this age group, and facilities providing that might be available only in
tertiary pediatric centers. The thin slices often necessary for the small
airway lesions suffer from a low signal-to-noise ratio, and motion artifacts
can further degrade images. In addition, MRI is not as widely available as CT
and it is still more costly than CT; consequently, MRI presently is not as
widely used in the workup of stridor or respiratory problems.
Airway hemangiomas, particularly the subglottic type, have been shown to be
associated with superficial hemangiomas of the head and neck and with other
cardiovascular anomalies (e.g., coarctation of a cervical right aortic arch)
[3,
7,
13,
18–21].
Orlow et al. [20] reported
that 10 of 16 patients with a facial hemangioma in a "beard"
distribution also had a clinically significant airway hemangioma. Therefore,
those authors recommend that any respiratory symptoms in an infant with a
superficial hemangioma in a "beard" distribution should prompt
investigation of the possibility of an associated airway hemangioma
[20,
21].
Despite the expected natural history of the hemangioma, the individual
lesion in the airway of the infant has a variable and unpredictable behavior.
The therapeutic approach to such a patient is controversial and not within the
scope of this paper. Therapy may vary according to the location and extent of
the hemangioma, the experience of the attending physicians, and the facilities
available. Because no single clinical parameter can reliably determine which
lesions can be treated expectantly and which may need more aggressive therapy,
the article by Seikaly and Cuyler is interesting
[22]. Those authors proposed
an objective guide to determine the optimal therapy of the patient with
subglottic hemangioma according to the estimated percentage of narrowing of
the tracheal lumen determined at bronchoscopy. In these terms, CT, with its
multiplanar imaging, would appear to be the optimal tool for applying such a
protocol to assess the degree of luminal narrowing and to stage the lesion in
preparation for therapy.
Until now the definitive study for the diagnosis of an airway hemangioma
has been endoscopy. The findings of our series, however, suggest that dynamic,
contrast-enhanced CT is a valuable, relatively noninvasive method that can
directly image such lesions and establish the diagnosis in infants. CT has
three major advantages over endoscopy. It is a noninvasive (other than IV
contrast injection) diagnostic method that permits visualization of the
maximal size of the lesion by using IV contrast material that appears to
distend the vessels of the hemangioma. With multiplanar reconstructions, CT
can illustrate the location and entire extent of the lesion in the axial,
sagittal, and coronal planes, and it can indicate the degree of luminal
narrowing and involvement of adjacent tissues. CT can serve not only as the
primary technique in the workup of suspected hemangioma of the airways but
also as a guide to therapy based on the protocol of Seikaly and Cuyler
[22]. Initial experience with
virtual bronchoscopy has also shown promising results
[23,
24]. We therefore conclude
that not only can CT confirm the diagnosis in equivocal cases, it can also
reduce or even obviate the more invasive bronchoscopic study.
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Abstract
Introduction
