DOI:10.2214/AJR.07.2079
AJR 2007; 189:393-399
© American Roentgen Ray Society
Airway Leiomyoma: Imaging Findings and Histopathologic Comparisons in 13 Patients
Yoon Kyung Kim1,
Hojoong Kim2,
Kyung Soo Lee1,
Joungho Han3,
Chin A Yi1,
Jhingook Kim4 and
Myung Jin Chung1
1 Department of Radiology and Center for Imaging Science, Samsung Medical
Center, Sungkyunkwan University School of Medicine, 50, Ilwon-Dong,
Kangnam-Ku, Seoul 135-710, Korea.
2 Division of Pulmonary and Critical Care Medicine, Department of Medicine,
Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul
Korea.
3 Department of Pathology, Samsung Medical Center, Sungkyunkwan University
School of Medicine, Seoul, Korea.
4 Department of Thoracic Surgery, Samsung Medical Center, Sungkyunkwan
University School of Medicine, Seoul, Korea.
Received January 2, 2007;
accepted after revision March 26, 2007.
Address correspondence to K. S. Lee
(kyungs.lee{at}samsung.com).
Abstract
OBJECTIVE. The aim of our study was to review retrospectively the
imaging findings on tracheobronchial leiomyoma and to compare them with the
pathologic findings.
CONCLUSION. Leiomyoma of the respiratory tract is located in the
bronchi in two thirds of patients and in the trachea in one third. The tumor
most commonly manifests on CT scans as a homogeneously enhancing airway tumor
with intraluminal growth. In approximately 15% of patients, the tumor has an
iceberg appearance.
Keywords: bronchial neoplasms • CT • leiomyoma • lung neoplasms • trachea • tracheal neoplasms
Introduction
Benign tracheobronchopulmonary neoplasms account for approximately
1% of respiratory tract tumors and 5-10% of resected tumors
[1]. Most of these neoplasms
are asymptomatic parenchymal lesions divisible by origin into mesenchymal,
submucosal, and epithelial tumors
[2]. Primary parenchymal
leiomyoma of the airway and lung is of mesenchymal origin. This tumor is rare,
approximately 2% of surgically resected benign lung tumors
[3]. In incidence, these tumors
follow carcinoids, hamartoma, lipoma, and chondroma
[1,
4]. Approximately 45% of
leiomyomas are endobronchial; the others occur in the lung parenchyma and
trachea [2].
Tracheobronchial leiomyoma occurs most commonly in the fourth decade of
life, although one third of patients are younger than 20 years. More than 90%
of lung parenchymal leiomyomas are incidental findings on chest radiographs.
Bronchial lesions are important causes of obstructive pneumonia and
atelectasis, and tracheal lesions can manifest as bronchial asthma
[3]. The histopathologic and
immunohistochemical staining properties of airway and parenchymal leiomyoma
are well known [5]. Because of
the rarity of these tumors, however, the radiologic findings have been
described only anecdotally in case reports
[3,
6-10].
The purpose of our study was to review retrospectively the imaging findings of
airway leiomyoma and to correlate the observations with the pathologic
findings.
Materials and Methods
Our institutional review board approved this retrospective study and waived
the requirement for informed consent. Written informed consent for CT had been
routinely obtained from all patients.
Patient Enrollment
Between November 1994 and October 2006, we reviewed all surgical biopsy
files and selected 16 patients with a pathologic diagnosis of
tracheobronchopulmonary leiomyoma. Two patients with small leiomyomas (< 2
mm in maximal diameter) were excluded. The tumors in these cases were
confirmed as incidental findings at bronchoscopic biopsy (bronchoscopy
performed for reasons other than airway tumor evaluation, i.e., evaluation of
interstitial lung disease and tuberculosis), but volume (helical) CT data were
not available; only unenhanced thin-section CT scans were obtained at 10-to
20-mm intervals. Also excluded was one patient with intrapulmonary leiomyoma.
Thus we included a total of 13 patients with histopathologically proven airway
leiomyoma.
Demographic Evaluation
Cigarette smoking history and the presence of symptoms and signs were
assessed. We also reviewed how the leiomyomas were detected and treatments
administered in each case. Follow-up details on patients who underwent
surgical or bronchoscopic tumor removal were recorded..
Image Acquisition
Initial chest radiographs and CT scans were available for all patients and
were obtained within 30 days of each other (mean, 6 days; median, 1 day;
range, 0-30 days). Chest radiographs were obtained in standard posteroanterior
projection with high-voltage technique (125 kVp, 5 mAs, 10-12:1 grid
ratio).
CT scans were obtained with helical technique: single-detector CT (HiSpeed
Advantage, GE Healthcare) for five patients, 4-MDCT (LightSpeed QX/i, GE
Healthcare) for one patient, 8-MDCT (LightSpeed Ultra, GE Healthcare) for two
patients, and 16-MDCT (LightSpeed 16, GE Healthcare) for five patients.
Scanning was performed from the level of the thoracic inlet to the midlevel of
the kidney. For two patients, only unenhanced scans were obtained; for one
patient, only enhanced scans; and for 10 patients, both enhanced and
unenhanced scans. The 10 patients included the two with leiomyoma < 2 mm in
diameter, neither of which was identified. For enhancement studies, 100 mL of
contrast medium (iomeprol, Iomeron 300, Bracco) was administered IV. Imaging
was started 20 seconds after completion of the contrast injection. For all
patients, the scanning parameters used were 120 kVp and 170-200 mA. For
single-detector helical CT, 7-mm collimation, pitch of 1, and a reconstruction
thickness of 7 mm were used for scanning and image reconstruction. For 4- to
16-MDCT, a beam width of 10 mm, beam pitch of 1.375-1.5, and reconstruction
thickness of 2.5-5.0 mm were used. All image data were reconstructed with a
bone algorithm. Data were entered directly into a PACS (PathSpeed or
Centricity 2.0, GE Healthcare), which displayed all image data on monitors
(four monitors, 1,536 x 2,048 image matrices, 8-bit viewable gray scale,
60-foot-lambert luminescence). These monitors were used to view both
mediastinal (width, 400 H; level, 20 H) and lung (width, 1,500 H; level, -700
H) window images.
Image Interpretation
The findings of chest radiographic and CT studies were analyzed
retrospectively and jointly by two chest radiologists, who had 2 and 16 years
of experience interpreting chest CT scans. Decisions on findings were reached
by consensus. Analysis of chest radiographs included locating the main tumors
and identifying the presence of postobstructive pneumonia or atelectasis. When
these conditions were absent, lesions were regarded as solitary nodules if the
diameter was less than 3 cm and as masses if the diameter was more than 3
cm.
CT analysis included determination of the location of the tumor in the
airway and of the shape, size, homogeneity, and attenuation coefficients of
the tumor. Tumors were subcategorized by location as tracheal or as being
located in a main, lobar, segmental, or subsegmental bronchus. The tumors also
were subcategorized as round, oval, or lobulated. Short- and long-axis tumor
diameters were measured. Tumors were subcategorized according to internal
content as homogeneous or heterogeneous on unenhanced and enhanced scans. When
both unenhanced and enhanced scans were available, enhancement attenuation
values of tumor nodules were calculated. The presence of postobstructive
pneumonia, bronchial dilatation, mucoid impaction, and atelectasis was
recorded, as were the presence and type of intratumoral calcification.
CT-Pathologic Comparisons
A lung pathologist with 14 years of experience reviewed all pathologic
specimens. To diagnose leiomyoma, immunostaining, that is, smooth muscle actin
and desmin staining, was conducted to differentiate this tumor from other
spindle cell tumors, such as fibroma, neurofibroma, and schwannoma
[2]. In the five patients who
underwent surgical resection, macroscopic tumor appearance and relation
between tumor and airways were described. During histopathologic examinations,
histologic tumor grade (low or high) and the presence of intratumoral
calcification, tumor necrosis, and extraluminal tumor components were
evaluated.
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Fig. 1B —63-year-old man (patient 4) with tracheal leiomyoma. Enhanced
transverse (B) and coronal reformatted (C) CT scans show
endotracheal nodule (arrow) with lobulated contour in intrathoracic
trachea.
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Fig. 1C —63-year-old man (patient 4) with tracheal leiomyoma. Enhanced
transverse (B) and coronal reformatted (C) CT scans show
endotracheal nodule (arrow) with lobulated contour in intrathoracic
trachea.
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Fig. 1D —63-year-old man (patient 4) with tracheal leiomyoma.
Low-magnification photomicrograph of pathologic specimen obtained at segmental
resection of trachea shows endotracheal tumor arising from membranous tracheal
wall (arrow). (H and E, x10)
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Fig. 1F —63-year-old man (patient 4) with tracheal leiomyoma. Photomicrograph
of transverse section of resected trachea lesion shows strongly positive
results. Both intratracheal and transmural (arrow) tumor growth are
evident. (Immunostain for smooth-muscle actin, x10)
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Results
Demographic and Clinical Features
Demographic data and imaging findings are summarized in
Table 1. The patients were
eight men and five women with an age range of 17-70 years (mean age, 48 years;
median, 51 years). At bronchoscopy the tumors were located in the trachea
(n = 4, 31%) (Figs.
1A,
1B,
1C,
1D,
1E,
1F and
2A,
2B,
2C,
2D) and in a main (n
= 4, 31%) (Figs. 3A,
3B,
3C,
3D and
4A,
4B,
4C,
4D), lobar (n = 3,
23%, including two lesions in the bronchus intermedius), or segmental
(n = 2, 15%) bronchus. Lesions detected with bronchoscopy and CT had
the greatest diameters, ranging from 2 to 40 mm (mean ± SD, 15 ±
9.3 mm; median, 22 mm).
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Fig. 2B —55-year-old woman (patient 2) with tracheal leiomyoma of iceberg
type. Enhanced transverse (B) and coronal-reformatted (C) CT
scans show tracheal tumor of irregular contour with both intraluminal
(arrowhead) and extraluminal (white arrows) components.
Stippled calcifications (black arrow) are present within mass.
Calcification was confirmed at microscopic examination.
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Fig. 2C —55-year-old woman (patient 2) with tracheal leiomyoma of iceberg
type. Enhanced transverse (B) and coronal-reformatted (C) CT
scans show tracheal tumor of irregular contour with both intraluminal
(arrowhead) and extraluminal (white arrows) components.
Stippled calcifications (black arrow) are present within mass.
Calcification was confirmed at microscopic examination.
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Fig. 2D —55-year-old woman (patient 2) with tracheal leiomyoma of iceberg
type. Photograph of gross pathologic specimen obtained at segmental resection
of trachea shows well-encapsulated mass with lobulated contour.
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Fig. 3A —21-year-old man (patient 7) with recurrent bronchial leiomyoma and
history of bronchial stenting for bronchial wall defect during bronchoscopic
removal of leiomyoma in left main bronchus. Posteroanterior chest radiograph
shows intrabronchial (arrowhead) and extrabronchial (arrows)
components of mass in left main bronchus. Lateral displacement of
azygoesophageal recess interface is evident in subcarinal area.
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Fig. 3B —21-year-old man (patient 7) with recurrent bronchial leiomyoma and
history of bronchial stenting for bronchial wall defect during bronchoscopic
removal of leiomyoma in left main bronchus. Lung (B) and mediastinal
(C) window transverse CT scans show lobulated mass in left main
bronchus. Intraluminal (arrowhead, B) and extraluminal
(arrow) components of tumor are evident.
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Fig. 3C —21-year-old man (patient 7) with recurrent bronchial leiomyoma and
history of bronchial stenting for bronchial wall defect during bronchoscopic
removal of leiomyoma in left main bronchus. Lung (B) and mediastinal
(C) window transverse CT scans show lobulated mass in left main
bronchus. Intraluminal (arrowhead, B) and extraluminal
(arrow) components of tumor are evident.
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Fig. 3D —21-year-old man (patient 7) with recurrent bronchial leiomyoma and
history of bronchial stenting for bronchial wall defect during bronchoscopic
removal of leiomyoma in left main bronchus. Photograph of gross specimen of
segmentally resected distal trachea and main bronchus (sleeve resection) shows
well-circumscribed grayish-white solid mass measuring 2.5 x 2 x
1.5 cm. Intraluminal (arrowhead) and extraluminal (arrows)
tumor components are evident.
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Fig. 4A —17-year-old boy (patient 6) with bronchial leiomyoma.
Posteroanterior chest radiograph shows endobronchial nodule (arrow)
in right main bronchus leading to atelectasis of right middle and lower
lobes.
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Fig. 4B —17-year-old boy (patient 6) with bronchial leiomyoma. Enhanced
transverse (B) and coronal reformatted (C) CT scans show oval
tumor nodule (arrow) occupying right main bronchus and attendant
atelectasis (arrowhead, C) of right middle and lower
lobes.
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Fig. 4C —17-year-old boy (patient 6) with bronchial leiomyoma. Enhanced
transverse (B) and coronal reformatted (C) CT scans show oval
tumor nodule (arrow) occupying right main bronchus and attendant
atelectasis (arrowhead, C) of right middle and lower
lobes.
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Fig. 4D —17-year-old boy (patient 6) with bronchial leiomyoma.
Low-magnification photomicrograph of pathologic specimen obtained with
bronchoscopic snaring shows nodule composed of subepithelial spindle cells. (H
and E, x12)
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In two (15%) of the patients, leiomyomas were detected incidentally during
bronchoscopic evaluation of the airways for other conditions (tuberculosis in
one patient, lung cancer in the other). Two other patients presented with
radiographic abnormalities without specific symptoms or signs. The other nine
patients had symptoms including cough (n = 7), dyspnea (n =
5), sputum (n = 4), fever (n = 2), wheezing (n =
1), and chest discomfort (n =1). Six men were smokers (mean, 36
pack-years; range, 3-80 pack-years); two men and five women were
nonsmokers.
Treatments administered for leiomyoma were bronchoscopic tumor resection in
eight cases, tracheal segmental resection in three cases, lobectomy in one
case, and segmental resection of the trachea and main bronchi with carinal
reconstruction in one case. During follow-up lasting a mean of 844 days
(range, 23-2,739 days; median, 379 days), CT (n = 7) and bronchoscopy
(n =1) showed no evidence of recurrence. One patient (patient 7) who
underwent surgical tumor removal had undergone bronchoscopic removal of an
endobronchial leiomyoma approximately 18 months previously.
Radiographic Findings
Chest radiographic findings were normal in three patients with a small
tumor nodule. In the other 10 patients, the tumor was identifiable and
appeared as an endobronchial nodule with postobstructive pneumonia or
atelectasis (n = 5) (Fig.
4A,
4B,
4C,
4D), an endotracheal nodule
(n = 3) (Fig. 1A,
1B,
1C,
1D,
1E,
1F), or an intratracheal and
extratracheal (n = 1) (Fig.
2A,
2B,
2C,
2D) or intrabronchial and
extrabronchial (n = 1) (Fig.
3A,
3B,
3C,
3D) mass.
CT Findings
Lesions were identified in 11 patients. In two other patients (patients 8
and 12), lesions were small and were found incidentally during bronchoscopy
performed to evaluate other lung lesions. CT images reconstructed with a 7-mm
section thickness did not depict these lesions. The lesions manifested as an
airway intraluminal nodule in nine cases (Figs.
1A,
1B,
1C,
1D,
1E,
1F and
4A,
4B,
4C,
4D) and as an iceberg tumor
(small intraluminal component and large extraluminal component) in two cases
(Figs. 2A,
2B,
2C,
2D and
3A,
3B,
3C,
3D). Lesions had an oval
contour in seven cases, a lobulated contour in three cases, and a round
contour in one case. Obstructive pneumonia, atelectasis, or mucus plugging was
found in five patients in whom a tumor nodule was located in a main
(n = 2), lobar (n =2), or segmental (n = 1)
bronchus. The internal contents of tumors were homogeneous in nine of the 10
patients for whom unenhanced CT scans were available. In the tenth patient,
the tumor was heterogeneous with a greatest diameter of 22 mm. On the enhanced
scans available for nine patients, five tumors (greatest diameter, 25 mm;
mean, 20 ± 6.9 mm; median, 23 mm) had homogeneous enhancement, and four
had heterogeneous enhancement (greatest nodule diameter, 40 mm; mean, 26
± 17.3 mm; median, 20 mm). In eight patients for whom both unenhanced
and enhanced scans were available, one lesion had a heterogeneous appearance
on unenhanced scans, and three lesions, including the lesions with a
heterogeneous appearance on unenhanced scans, had a heterogeneous appearance
on enhanced scans (Table 1).
Leiomyomas had an attenuation of 25-46 H on unenhanced CT and 46-85 H on
enhanced CT (mean attenuation, 32 ± 14.7 H; range, 15-60 H; median, 30
H). Stippled calcifications were found in a patient (patient 2) with a large
tumor that had both intraluminal and extraluminal components.
CT-Pathologic Comparison
In the five patients who underwent surgical resection, CT precisely
depicted tumor location and extent. The macroscopic appearances of these
tumors were whitish-tan and oval or lobulated margin. The tumors were polypoid
intraluminal masses in three patients and intraluminal and extraluminal masses
in two patients. Microscopic examination showed all tumors were composed of
proliferating spindle cells. Immunostaining for smooth muscle actin and desmin
was performed in four cases, and all results were strongly positive. No
cellular pleomorphism was found. Mitotic figures were found in one patient but
were not prominent (1/20 high-power fields). No tumor necrosis was found.
Intratumoral dystrophic calcification was found in one patient. Histologic
results revealed reactive hyperplasia in the regional lymph nodes of two
patients.
Discussion
Approximately 45% of primary airway and parenchymal leiomyomas have an
endobronchial location; others occur in the lung parenchyma and the trachea
[2]. In our study, most (nine
of 13) of the airway leiomyomas were in a bronchus, and the others were in the
trachea.
An iceberg tumor growth pattern (a small intraluminal component and a large
extraluminal component) is a known finding among airway tumors such as
carcinoid and mucoepidermoid carcinomas. Because of the large extraluminal
component, bronchoscopic resection of these tumors is not recommended
[11,
12]. The iceberg growth
pattern of respiratory tract leiomyoma has not been previously reported, to
our knowledge. In our study, two leiomyomas (one tracheal and the other main
bronchial) manifested as iceberg tumors. In the case in which the iceberg
tumor was located in a main bronchus, the patient had been treated previously
with bronchoscopy, but the tumor recurred 18 months later. After curative
resection of the distal tracheal and left main bronchial iceberg tumors, no
evidence of recurrence was found over a 3-year follow-up period.
In our study, most leiomyomas had identifiable enhancement (15 H or more)
after IV contrast injection. This identifiable enhancement of leiomyomas has
been observed in locations other than the lung. The attenuation of uterine
leiomyoma is similar to that of normal myometrium and thus high levels of
attenuation (
78 H on unenhanced CT scans and 127 H on enhanced CT scans)
[13]. However, esophageal
leiomyoma has been found to exhibit relatively little attenuation ( 21-39
H on unenhanced CT scans and 25-51 H on enhanced CT scans) compared with
leiomyoma of other organs. These differences in degrees of attenuation may be
due to organ-related differences in vascular supply
[14].
Stippled calcification was noticed in only one tumor in our study.
Calcification in respiratory tract leiomyomas is rare; only two cases, which
occurred in parenchymal leiomyomas, have been reported in the literature
[3], although in that study,
image analysis was based on findings on radiographs in most cases.
Calcification is also uncommon in leiomyoma of other organs, for example, 8%
of cases of esophageal leiomyoma and 3-10% of cases of uterine leiomyoma
[13,
14].
The prognosis of leiomyoma, excluding low-grade leiomyosarcoma, is
excellent after complete resection. In the presence of increased mitotic
activity (> 3/10 high-power fields), cytologic atypia, and necrosis,
leiomyosarcoma should be seriously considered
[2]. In our study, there was no
pathologic evidence of cytologic atypia or necrosis. Only one tumor had low
mitotic activity (1/20 high-power fields). In addition, over a mean follow-up
period of 844 days, no tumor recurred after bronchoscopic tumor removal or
surgical resection.
One noteworthy fact is that small nodules (2 mm or less in diameter) within
the airway, which are identifiable with bronchoscopy, may be missed on imaging
studies when a thick (7 mm or greater) reconstruction interval is used. Had we
routinely used thin-section volumetric data sets, we might not have missed
these small nodules.
Our study was intrinsically limited by its retrospective design. In
addition, selection bias might have been introduced because we included
patients with biopsy-proven tumors, thus the observed tumor distribution in
the trachea and bronchi may not be representative. We also reconstructed
mediastinal and lung window images by using a bone algorithm, which might have
adversely affected accurate characterization of internal architecture,
detection of calcifications, and assessment of the attenuation of leiomyoma
nodules. Moreover, we did not attempt to standardize the volume of contrast
material administered according to patient weight or to deliver contrast
medium uniformly by using a saline chaser and a double-barreled injector.
In summary, leiomyoma of the airway is rare and most commonly manifests as
an airway tumor with intraluminal tumor growth, although our findings suggest
that these tumors have an iceberg appearance in approximately 15% of cases.
Most of these tumors appear as a homogeneous nodule on unenhanced CT scans and
become enhanced after IV injection of contrast medium. The prognosis is
uniformly good, and recurrence is rare after complete resection.
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Abstract
Introduction
