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CASE REPORT
Metastatic tumor to the orbital cavity from a primary carcinomaof the uterine cervix : a case report
Hiroshi Kagusa1), Yoshifumi Mizobuchi1), Kohei Nakajima1), Toshitaka Fujihara1), Yoshimi Bando2), and Yasushi Takagi1)
Department of Neurosurgery, Institute of Health Biosciences, the University of Tokushima, Japan, 2)Department of Pathology, Tokushima
University hospital, Japan1)

Abstract : Metastatic tumors to the orbit of the eye, especially from primary carcinomas of the uterine cervix arevery rare. A 64-year-old woman with a history of carcinoma of the uterine cervix presented with right eye pain
and blepharoptosis for 2 weeks. Magnetic resonance imaging revealed a mass at the right orbital apex. Surgicalextirpation was performed due to severe pain. Postoperative pathology demonstrated a poorly differentiatedsquamous cell carcinoma. The origin was ultimately considered to be the carcinoma of the uterine cervix. In conclusion, this report describes a rare case of a metastatic tumor at the orbital apex derived from the cervix of theuterus. J. Med. Invest. 66 : 355-357, August, 2019
Keywords : orbital metastasis, carcinoma of the uterine cervix, ophthalmalgia

INTRODUCTION
Metastasis to the orbital cavity is a rare manifestation of systemic cancer (1). Intraorbital tumors account for approximately
1% of ophthalmic diseases, and intraorbital metastatic tumors
have been reported to account for only 3-5% of all intraorbital
tumors (2). On the other hand, there are seven case reports ofuterine malignant tumors as primary lesion (3, 4).
We here report the characteristic symptoms and findingsof orbital metastasis from a primary carcinoma of the uterinecervix.

CASE REPORTA 64-year-old woman with a history of carcinoma of the uterine cervix (Figure 1), which had been treated by radiotherapy
and chemotherapy 7 months earlier, presented with a two-weekhistory of ophthalmalgia, blepharoptosis, and proptosis of theright eye. Ocular and neurological examination showed limitation of movement of the right eye and right mydriasis. Cranial
MRI revealed an intraconal mass at the right orbital apex (Figure 2A). Eight days after the first examination, follow-up MRI
showed that the orbital mass had doubled in size, with a well-circumscribed enhancing mass observed upon contrast-enhanced
imaging (Figure 2B, C).
To remove the severe ophthalmalgia and diagnose the mass,surgical extirpation was performed. The postoperative pathology
demonstrated a poorly differentiated squamous cell carcinoma(Figure 3) that was same as pathological information of uterine
cervical carcinoma (Figure 4). So, it was considered to be derived
from the primary carcinoma at the uterine cervix.
After the operation, the ophthalmalgia was ameliorated. MRI
performed five days postoperatively showed that the mass had
been totally removed (Figure 5).
Received for publication March 7, 2018 ; accepted July 21, 2019.
Address correspondence and reprint requests to Hiroshi Kagusa, Department of Neurosurgery, Institute of Health Biosciences, The Universityof Tokushima Graduate School, Tokushima, 3-18-15, Kuramoto-cho,
Tokushima 770-8503, Japan and Fax : +81-88-632-9464.

In addition to the operation, radiation therapy (60 Gy/30fractions) was performed at the right orbital cavity. However,whole-body computed tomography revealed metastases to theleft hypochondrial area and hypodermis of the back, and chemotherapy was hence initiated. Two months postoperatively, thepatient experienced peritonitis by perforation and died.

DISCUSSION
Intraorbital tumors are rare, accounting for only about 1% of
ophthalmic diseases, with intraorbital metastatic tumors being
highly rare, accounting for only 3-5% of intraorbital tumors. The
peak age for these tumors has been reported as 44 years, withlung cancer being the most common primary tumor, accountingfor 17% of cases, followed by breast cancer at 14% (2). On theother hand, there are very few reports of uterine malignanttumors as the primary lesion ; to the best of our knowledge, only
seven such cases have been previously reported (3, 4). In thesecases, the histology was variable, including 4 squamous cell
carcinomas, one adenocarcinoma, one sarcoma, and one undifferentiated carcinoma.
The blood flow in the eye area accounts for 2-5% of the total
circulation (5). Thus, compared to other organs, the blood flow isvery low. In addition, the intraorbital space is a site with a lot ofmotion, including due to the extraocular muscles ; thus, tumor
cells cannot easily survive in this space. For these reasons, metastasis to the orbit is rare.
The symptoms include ophthalmalgia, bulging eyes, eyemovement disorders, and vision disorders, among others. The
most common symptom of intraorbital tumors is exophthalmos,and ophthalmalgia emerges from an early stage in intraorbital
metastatic tumors (6).
In the present case, intolerable eye pain was the main symptom ; the tumor diameter doubled in 8 days, and the pain wasimproved by tumor removal. We consider that the primary tumor
was highly malignant and that the progress of the metastaticlesions was also fast, as evidenced by not only the growth of theorbital tumor but also by the fact that trunk metastasis also
occurred after the surgery. Especially, in the orbit space, thespace is narrow, and symptoms accompanying tumor growth(ocular pain and protrusion of the eyes) are likely to appear. If

The Journal of Medical Investigation Vol. 66 2019

H. Kagusa, et al. Metastatic tumor to the orbital cavity

356
AB

DC

Figure 1.

A : Axial T2-weighted magnetic resonance image showing a mass at the pelvic cavity.
B : Axial diffuse-weighted magnetic resonance image showing a mass at the pelvic cavity. The mass shows high intensity.
C : Contrast-enhanced T1-weighted image (Axial).
D : Contrast-enhanced T1-weighted image (Sagittal).

A

B

C

Figure 2.

A : Axial T1-weighted magnetic resonance image showing a right orbital apex mass at the first examination.
B : Repeat magnetic resonance image taken eight days after the first examination.
C : Contrast-enhanced T1-weighted image taken eight days after the first examination.

AB

100 μmFigure 3.

C100 μm

A : Hematoxylin and eosin stain. Dyskaryosis and nuclear fission are observed. Arrowheads indicate atypical cells.
B : AE1/AE3 stain. This marker stains epithelial cells. Arrowheads indicate stained cells.
C : p63 stain. This marker stains squamous cell carcinoma cells. Arrowheads indicate stained cells.

100 μm357

The Journal of Medical Investigation Vol. 66 August 2019AB

100 μm

100 μmFigure 4.

A : Hematoxylin and eosin stain. Dyskaryosis and nuclear fission are observed.
B : p63 stain.

Figure 5. Contrast-enhanced T1-weighted magnetic resonance image after the operation.

the primary tumor is solid, the metastatic tumor may strongly
push against the trigeminal nerve, resulting in symptoms suchas ocular pain manifesting earlier.
Although intraorbital metastatic tumors are very rare, especially when symptoms such as ocular pain are examined in advance, rapidly progressing intraorbital tumors are occasionallyfound ; in 19% of such cases, the tumor is a metastatic lesion (7).
When ocular symptoms such as eyeball protrusion and ocular
pain appear and exacerbate rapidly, metastatic tumors should
be suspected, both in cases with and without a cancer history.
In these cases, whole-body imaging studies may be necessary.

2.

3.

4.

5.
6.

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