AJR 2003; 181:1544-1546
© American Roentgen Ray Society
Pulmonary Arteriovenous Fistulas Developed After Chemotherapy of Metastatic Choriocarcinoma
Seung Hong Choi1,
Jin Mo Goo,
Hyo-Cheol Kim and
Jung-Gi Im
1 All authors: Department of Radiology, Seoul National University College of
Medicine, the Institute of Radiation Medicine, SNUMRC, and Clinical Research
Institute, 28 Yongon-dong, Chongno-Gu, Seoul 110-744, Korea.
Received January 3, 2003;
accepted after revision May 14, 2003.
Address correspondence to J. M. Goo
(jmgoo{at}plaza.snu.ac.kr).
Partially supported by the 2001 BK21 Project for Medicine, Dentistry, and
Pharmacy.
Introduction
Agestational choriocarcinoma is a highly malignant neoplasm arising from
the trophoblast of a human pregnancy and has a high metastatic potential. The
most common site of metastasis is the lung, with an incidence of 45–87%
[1]. Angiographic studies
characteristically have depicted prominent arteriovenous shunts in the uterus,
often with persistence of the shunts after the successful eradication of the
tumor by chemotherapy [2]. A
variety of pseudonyms including arteriovenous fistula, arteriovenous
malformation, benign cavernous hemangioma, and arteriovenous angiomatosis are
used for arteriovenous shunts. Pulmonary metastatic lesions resemble the
primary tumor both on gross specimens and at histology. The arteriovenous
fistulas of pulmonary metastatic tumors can be visualized angiographically
before treatment [3]. We
recently encountered the development of arteriovenous fistulas associated with
pulmonary metastases of a choriocarcinoma after successful chemotherapy. We
report a case of pulmonary arteriovenous fistulas developed in the regions of
metastatic disease after treatment.
Case Report
A 30-year-old woman underwent an evacuation of a uterine choriocarcinoma in
another hospital in May 2000. She had delivered a healthy baby 5 years before.
The serum human chorionic gonadotropin (HCG) level was 24,323 mIU/mL. She was
transferred to our hospital for further evaluation and treatment in July 2000.
On admission, the serum HCG level was 4,250 mIU/mL. Findings of pelvic
sonography and MRI revealed a residual hypervascular tumor that was compatible
with choriocarcinoma in the uterine fundus. Dilatation and curettage were
performed, and there was no evidence of residual choriocarcinoma on the
pathologic report. At that time, chest radiography and chest CT revealed
multiple metastatic nodules in both lungs (Figs.
1A and
1B). No other metastasis was
found in the brain, abdomen, or pelvic cavity on abdominal CT and brain MRI. A
biopsy of the lung lesions was not performed because of the small size of the
nodules and the risk of bleeding. She received eight courses of combination
chemotherapy with methotrexate and leucovorin for 2 years and attained normal
serum HCG levels. Findings of serial chest radiography showed that the
multiple metastatic nodules in both lungs had decreased in size, but had not
completely disappeared.
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Fig. 1A. —30-year-old woman with choriocarcinoma and multiple pulmonary
metastases. Unenhanced chest CT scans obtained at admission show multiple
variable-sized nodules (arrows) through both lungs, indicating
pulmonary metastases of choriocarcinoma.
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Fig. 1B. —30-year-old woman with choriocarcinoma and multiple pulmonary
metastases. Unenhanced chest CT scans obtained at admission show multiple
variable-sized nodules (arrows) through both lungs, indicating
pulmonary metastases of choriocarcinoma.
|
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The patient was regularly followed up and was healthy until April 2002 when
she presented with intermittent hemoptysis. Findings of chest radiography
revealed subtle ground-glass opacity in the right mid lung zone. On unenhanced
chest CT, many metastatic nodules had disappeared compared with the previous
CT scan, and two nodules remained in the left upper lobe. However, there were
curvilinear structures that were connected to the residual nodules suggesting
arteriovenous fistulas (Figs.
1C and
1D). Unenhanced MDCT with 3D
reconstruction was performed to confirm this diagnosis, which identified many
arteriovenous fistulas (Figs.
1E,
1F,
1G). Because there was no
recurrent hemoptysis and no evidence of tumor recurrence (ß-HCG level,
< 3 mIU/mL), she was given no specific treatment.
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Fig. 1C. —30-year-old woman with choriocarcinoma and multiple pulmonary
metastases. Unenhanced chest CT scans obtained after eight courses of
methotrexate and leucovorin therapy for 2 years show two nodules
(arrows) in left lung. These residual nodules communicate with
curvilinear structures (arrowheads), suggesting arteriovenous
fistulas.
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Fig. 1D. —30-year-old woman with choriocarcinoma and multiple pulmonary
metastases. Unenhanced chest CT scans obtained after eight courses of
methotrexate and leucovorin therapy for 2 years show two nodules
(arrows) in left lung. These residual nodules communicate with
curvilinear structures (arrowheads), suggesting arteriovenous
fistulas.
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Fig. 1E. —30-year-old woman with choriocarcinoma and multiple pulmonary
metastases. Unenhanced transverse thin-slab maximum-intensity-projection MDCT
scan with lung window setting shows two nodules (arrows) associated
with arteriovenous fistulas (arrowheads) in left upper lobe.
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Fig. 1F. —30-year-old woman with choriocarcinoma and multiple pulmonary
metastases. Curved reformatted MDCT image shows nodule (black arrow)
associated with arteriovenous fistula in lingular segment of left upper lobe.
Note pulmonary artery (arrowheads) and pulmonary vein (white
arrows).
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Fig. 1G. —30-year-old woman with choriocarcinoma and multiple pulmonary
metastases. Volume-rendered MDCT image shows nodule (large arrow)
associated with arteriovenous fistula in lingular segment of left upper lobe.
Note pulmonary artery (arrowheads) and pulmonary vein (small
arrows).
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Discussion
Pulmonary arteriovenous fistula, a direct communication between the
pulmonary artery and vein without an intervening capillary bed, is a rare
vascular anomaly [4]. Although
most (80%) are congenital, approximately 20% are acquired as a result of
trauma, infection, metastatic carcinoma, mitral stenosis, or chronic hepatic
cirrhosis [4].
It has been reported that pulmonary metastatic tumors of a choriocarcinoma
are supplied by the pulmonary artery
[3]. The classic triad of
exertional dyspnea, cyanosis, and clubbing is found in 30% of patients, and
the large arteriovenous fistulas undoubtedly contribute significantly to the
hypoxemia that is refractory to supplemental oxygen
[3]. In this patient, a few
small arteriovenous fistulas were detected, and there was no clinical
manifestation such as dyspnea, cyanosis, or central nervous system symptoms
other than episodic hemoptysis. Although most patients are asymptomatic, it is
well known that pulmonary arteriovenous fistulas can cause dyspnea from a
right-to-left shunt, bleeding, or result in hemoptysis and hemothorax,
creating the need for angiographic treatment or surgery
[4]. Because of paradoxical
cerebral emboli, various central nervous system complications have been
described, including stroke and brain abscess
[4]. In our patient, the
hemoptysis improved with conservative treatment. Because she declined
embolization therapy, no specific treatment was performed for the
arteriovenous fistulas. We think that the intratumoral fistulas between the
pulmonary arteries and the pulmonary veins were developed by a pulmonary
metastatic choriocarcinoma and only arteriovenous fistulas remained at the
metastatic site, even though the pulmonary metastatic tumors have been
controlled by successful chemotherapy. Persistent arteriovenous fistulas
associated with a uterine choriocarcinoma remained after eradication by
chemotherapy [2]. Casson et al.
[5] hypothesized that
hematogenous metastases of choriocarcinoma in the lung might develop a local
arteriovenous shunt, which persists after the successful completion of
chemotherapy.
Angiography is important for detecting the site and size of pulmonary
arteriovenous fistulas when embolization technique or surgical therapy is
considered. CT can reveal more abnormal regions when compared with
angiography. The superiority of CT was based on the transverse view of the
lung, which showed lesions of any size without superimposition
[6]. Remy et al.
[7] reported that conventional
CT enabled identification of 98% of pulmonary arteriovenous fistulas, although
angiography did so for 60% of pulmonary arteriovenous fistulas. As illustrated
in our series, unenhanced MDCT with various 3D reconstruction images allows
the imaging of the vessel along any axis. In our patient, the feeding
(pulmonary) arteries and the draining (pulmonary) veins of the arteriovenous
fistulas were directly visible by volume-rendered imaging. In addition,
unenhanced MDCT with various 3D reconstruction techniques excellently showed
the site and size of the pulmonary arteriovenous fistulas as well as the lung
parenchyma. Tsunezuka et al.
[8] reported two nidi with
feeding arteries and draining veins on enhanced 3D CT angiography, indicating
pulmonary arteriovenous fistulas. We could thoroughly evaluate the pulmonary
arteriovenous fistulas using unenhanced MDCT with various 3D reconstruction
techniques.
In conclusion, the outcomes of pulmonary metastatic tumors of a uterine
choriocarcinoma have markedly improved since the advent of several
chemotherapy regimens based on chest radiography, chest CT, and ß-HCG
levels. The arteriovenous fistulas associated with the pulmonary metastatic
lesions can be found using unenhanced MDCT with various 3D reconstruction
techniques.
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Introduction
Case Report
