Case Report pISSN 1738-2637 / eISSN 2288-2928 J Korean Soc Radiol 2018;78(4):284-288 https://doi.org/10.3348/jksr.2018.78.4.284
Pulmonary Metastasis Originated from Uterine Sarcoma, Presenting as Multiple Nodules with Tortuous, Serpentine, Aneurysmal, Dilated Intratumoral Vessels: A Case Report 종양 내 구불구불한 동맥류성 혈관 확장을 보이는 자궁 육종의 폐전이: 증례 보고 Hyeon Ji Jang, MD1, Song Soo Kim, MD1*, Hee Sun Park, MD2, Jeong Eun Lee, MD2, Jin Hwan Kim, MD1 Department of Radiology, Chungnam National University Hospital, Chungnam National University School of Medicine, Daejeon, Korea Division of Pulmonology, Department of Internal Medicine, Chungnam National University Hospital, Chungnam National University School of Medicine, Daejeon, Korea 1 2
Pulmonary metastases present a wide spectrum of radiological findings, some of which have been known to be useful for analogizing the possible origin or site of primary tumors. In the present report, we describe a unique case of pulmonary metastasis manifesting on chest computed tomography as multiple nodules with tortuous, serpentine, aneurysmal, dilated, inner intratumoral vessels. The metastasis originated from uterine sarcoma. Index terms Sarcoma Lung Neoplasms Neoplasm Metastasis Tomography, X-Ray Computed
INTRODUCTION
Received June 29, 2017 Revised September 20, 2017 Accepted November 11, 2017 *Corresponding author: Song Soo Kim, MD Department of Radiology, Chungnam National University Hospital, Chungnam National University School of Medicine, 282 Munhwa-ro, Jung-gu, Daejeon 35015, Korea. Tel. 82-42-280-7333 Fax. 82-42-253-0061 E-mail:
[email protected] This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
air bronchogram,” “cavity,” and “perinodular” or “peritumoral ground-glass opacity” (2). These radiological features are help-
Pulmonary metastases are common and can be classified ac-
ful in determining the primary site of malignancy. However, to
cording to the growth site, number of tumors, and pattern of
the best of our knowledge, only a few reports have documented
radiological manifestations. With respect to the seeding mecha-
chest computed tomography (CT) showing a dilated vascular
nism, pulmonary metastases usually develop when tumor cells
structure in metastatic nodules or masses. Herein, we present a
lodge in a small distal arteriole or in the interstitium. However,
unique case of pulmonary metastasis manifesting as multiple
sometimes the cancerous cells are deposited as an endobron-
nodules with tortuous, serpentine, aneurysmal, dilated, intratu-
chial lesion (1). Pulmonary metastases most commonly present
moral vessels, which originated from uterine sarcoma.
as multiple nodules or masses; however, in some cases of colorectal cancer or skeletal sarcoma, solitary metastases are predominant. Furthermore, pulmonary metastases can manifest in various patterns, including “military pattern,” “consolidation with
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Case Report A 60-year-old woman presented with generalized weakness, Copyrights © 2018 The Korean Society of Radiology
Hyeon Ji Jang, et al
dyspnea, and weight loss that had persisted for 5 months. Her
lar pseudoaneurysms associated with pulmonary vasculitis or
medical history revealed hypertension diagnosed two years
septic pneumonia. However, the vascular structures did not
earlier. After admission, she underwent all available routine
have any direct connection with the pulmonary artery or its
laboratory tests, which displayed the following abnormal labo-
branches. They were confined within the masses and nodules.
ratory findings: increased white blood cell count (17400/µL) and
Some nodules had a low attenuation portion, suggesting inter-
high level of C-reactive protein (23.3 mg/dL), suggesting active
nal necrosis. Collectively, these findings suggested pulmonary
inflammation throughout the body. Moreover, her hemoglobin
metastasis. Laboratory examination revealed that the patient’s
level was 5.3 g/dL, mean corpuscular volume was 70.9 fL, se-
serum cancer antigen-125 level was markedly elevated (245.2
rum iron level was 22 µg/dL, and ferritin level was 1478 ng/mL,
U/mL; normal: < 35 U/mL). Subsequently, for further evalua-
suggesting microcytic anemia with iron deficiency.
tion, she underwent pelvic ultrasonography, which revealed a
She underwent radiological testing, and a chest radiograph
6.6-cm, heterogeneous, irregular, putative primary mass in the
showed diffuse, variably sized, nodular opacities in both lungs
uterus. She then underwent abdominal CT to rule out other
(Fig. 1A). They were well-defined round masses. For further
malignancies, such as ovarian cancer. The abdominal CT showed
evaluation, she underwent chest CT. Pre-contrast axial chest CT
a large, cystic, uterine mass with a partially enhancing solid
showed multiple, variably sized, well-defined, round-to-ovoid
portion in the right uterine wall; the mass was regarded as uter-
soft tissue nodules and masses without any inner calcific foci in
ine malignancy, and a biopsy was recommended, but the pa-
both lungs (Fig. 1B). Contrast-enhanced CT revealed multiple
tient refused the procedure. Therefore, a percutaneous core-nee-
diffuse, heterogeneous, enhancing nodules and masses with
dle biopsy of a nodule in the right lower lobe of the lung was
tortuous, serpentine, or engorged inner vascular structures (Fig.
performed. Microscopic examination revealed multiple cystic
1C-E). We first considered the possibility of pulmonary vascu-
spaces with a flat, endothelial-like cell lining, indicating vascular spaces, and the tumors proved to be lung metastases derived from sarcomatous carcinoma (Fig. 1F). The patient underwent one course of palliative chemotherapy, but her general condition worsened. After discussions with her family, she decided not to undergo any more aggressive treatments, such as chemotherapy and radiotherapy. She was then transferred to another hospital in her hometown for conservative treatment.
Discussion In this report, we have presented a unique case of multiple, variably sized, pulmonary metastatic masses and nodules showing engorged, aneurysmal, and dilated tumor vessels, and these metastatic masses and nodules originated from uterine sarcoma. Pulmonary metastases are common and show a wide range of radiological features. On one hand, these features help in determining the original tumor site; on the other hand, as in the A Fig. 1. A 60-year-old woman with pulmonary metastasis from uterine sarcoma, presenting as multiple nodules with tortous, serpentine, aneurysmal dilated intraumor vessels. A. Initial chest radiograph shows multiple, round, variably sized nodules and masses in both lungs, as well as a small amount of pleural effusion.
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J Korean Soc Radiol 2018;78(4):284-288
present study, the unusual radiological features of some metastases make it difficult to distinguish them from non-malignant pulmonary diseases (2). The various known features of lung metastases are cavitation, calcification, hemorrhage around the metastatic nodules, pneumothorax, air-space pattern, tumor
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B
C
D
E
F Fig. 1. A 60-year-old woman with pulmonary metastasis from uterine sarcoma, presenting as multiple nodules with tortous, serpentine, aneurysmal dilated intraumor vessels. B. Pre-enhanced image shows well-defined homogeneous, variably sized, soft-tissue nodules with a density similar to that of the back muscles and no inner calcific foci. C. Axial contrast-enhanced CT image shows aneurysmal dilatated intratumoral vessel (thick arrow). It has no connection with adjacent branch of the pulmonary artery (thin arrow). D. Coronal contrast-enhanced CT image also show aneurysmal dilatated intratumoral vessels (thick arrows). Branches of the pulmonary artery (thin arrows) pass through the nodules without any connection. E. Coronal contrast-enhanced CT image show multiple variable sized nodues and masses with tortous, dilatated intratumoral or intranodal vessels with an inner cystic or necrotic portion. F. Photomicrographs show tumor cell infiltration with circumscribed cystic spaces lined with flat, endothelial-like cells (arrow), suggesting vascular structures (hematoxylin and eosin, × 100). Cystic spaces show CD-31 positivity (CD-31, × 100) and tumor cells react strongly with mesenchymal antigen (Vimentin, × 400). CD-31 = cluster of differentiation-31, CT = computed tomography
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Hyeon Ji Jang, et al
embolism, endobronchial metastasis, solitary mass, and dilated
sion and aneurysmal dilatation of the pulmonary artery itself (9,
vessels within a mass (2). Squamous cell carcinoma is the most
10). However, in the present case, the vascular structures did
common cancer type that yields a cavitating metastasis with a
not show any direct connection with the pulmonary artery or
thick and irregular wall. In contrast, thin-walled cavities can be
its branches. Infection with tuberculosis bacteria, pyogenic bac-
observed in metastases from sarcomas and adenocarcinomas.
teria, and fungi can also cause pseudoaneurysms (9, 10). How-
Furthermore, calcification can occur in metastatic sarcomas or
ever, in such cases, the main findings are likely to be ground-glass
adenocarcinomas, making it difficult to differentiate these dis-
opacity or consolidation, reflecting infection or inflammation
eases from benign granulomas or hamartomas. Peritumoral
of the lung, rather than multiple metastatic nodules.
hemorrhage results in areas of nodular attenuation surrounded
In summary, we have presented a unique case of pulmonary
by a halo of ground-glass opacity. Pneumothorax commonly
metastasis manifesting as multiple nodules with well-devel-
occurs as a result of metastases from an osteosarcoma. Air-
oped, tortuous, serpentine, aneurysmal, dilated inner intratu-
space consolidation is often seen in metastases from gastroin-
moral vessels, and the metastasis may have originated from
testinal tract malignancies, whereas a common radiological
uterine sarcoma.
manifestation of endobronchial metastasis is atelectasis (2). Therefore, these findings are helpful in the differential diagnosis
References
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J Korean Soc Radiol 2018;78(4):284-288
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종양 내 구불구불한 동맥류성 혈관 확장을 보이는 자궁 육종의 폐전이: 증례 보고 장현지1 · 김성수1* · 박희선2 · 이정은2 · 김진환1 악성 종양으로부터의 폐전이는 다양한 범위의 영상학적 소견을 보인다. 폐전이의 몇 가지 특징들에서는 종양의 원발 위치 를 유추할 수 있는 것으로 알려져 있다. 저자들은 흉부 전산화단층촬영에서 다수의 전이성 폐 결절 내부에 구불구불하게 늘어난 혈관들을 보이는, 독특한 양상의 자궁 육종 폐전이 1예를 경험하였기에 이를 보고하고자 한다. 충남대학교 의과대학 충남대학교병원 1영상의학과, 2호흡기내과
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