Downloaded from www.ajronline.org by 164.77.172.146 on 11/12/15 from IP address 164.77.172.146. Copyright ARRS. For personal use only; all rights reserved
1163
Case .
.;
Report
.
Intracranial Metastatic Adenoid Cystic Carcinoma: Presumed Hematogenous Spread from a Primary Tumor in the Parotid Gland Nicholas
D. Gelber,1
Ronald
L. Ragland,1
John
R. Knorr,
Intracranial extension of adenoid cystic carcinomas of the head and neck is well recognized, and typically such spread is by way of contiguous perineural invasion [1 -3]. Hematogenous intracranial extension, although reported, is much less common. We report a case of hematogenous metastasis of parotid adenocystic carcinoma to the inner table of the calvaria, epidural compartment, and brain parenchyma.
man presented
with ataxia of several days’ duration,
slurred speech, and some dizziness. Six to eight months earlier, he had undergone resection of a right parotid mass. Subsequent pathologic examination showed an adenocystic carcinoma of the parotid gland, and the patient underwent radiotherapy. No tumor spread was identified at this time. On admission, the man was alert and his vital signs were normal. The patient’s affect was blunted and his mood was depressed. His
gait was slightly
ataxic.
No other focal neurologic
signs were de-
tected. A
CT scan showed
an irregular
in the Ieftfrontal
region.
ial mass
with multiple
Underlying
lesion,
Thomas
W. Smith,2
parenchyma lesion
was
This
was
area of focal calvarial destruction associated
peripheral
with
areas
a lobulated
extraax-
of ring enhancement.
this area, a focal area of vasogenic
type
of white matter
edema was seen in the left frontal lobe with mass effect and compression of the ipsilateral frontal horn (Figs. 1 A-i C). At surgery, an infiltrating lobulated irregular soft-tissue mass was
found involving the left frontal bone, the adjacent
Bernard
of the superficial incompletely
Pathologic
examination
(Fig.
tumor
This
portion
showed
pseudoglandular
1 D).
B. Stone3
of the left frontal
lobe. The
excised.
like structures,
and
the
tumor
tissue
spaces, original
consisting
pseudocysts, pathologic
parotid tumor had virtually identical appearances. No evidence of lung, bone, or liver metastases time.
of lumen-
and necrosis
specimen
of
the
was found at this
dura, and the brain
Adenoid cystic carcinoma (cylindroma, adenocystic carcinoma) is a reasonably common malignant tumor of the large and small salivary glands, constituting approximately 8% of all salivary gland tumors and approximately 22% of malignant salivary gland tumors [4]. While most salivary gland lesions are commonly found in the parotid and submandibular glands, these tumors are relatively more common in the sublingual and accessory salivary glands [4]. The overall frequency of intracranial invasion of adenoid cystic carcinoma has been reported to be between 4% and 22%. However, the vast majority of these tumors spread either directly or via perineural spread [1 -3]. True hematogenous metastases to the CNS are rare, even though hematogenous metastases to liver, lung, and bone are reasonably common late in the course of this disease [3]. Extraaxial intracranial metastatic adenocystic carcinoma mimicking meningioma has been reported [2, 3], and cylin-
Received October 23, 1 991 ; accepted after revision December 4, 1991. 1 Department of Radiology, University of Massachusetts Medical Center, 55 Lake Ave. N., Worcester, 2 Department of Neuropathology, University of Massachusetts Medical Center, Worcester, MA 01655. 3 Department of Neurosurgery, University of Massachusetts Medical Center, Worcester, MA 01655. AJR 158:1163-1164,
and
Discussion
Case Report A 69-year-old
1
May 1992 0361-803x/92/1
585-1 163 © American Roentgen Ray Society
MA 01 655. Address
reprint
requests
to N. D. Gelber.
Downloaded from www.ajronline.org by 164.77.172.146 on 11/12/15 from IP address 164.77.172.146. Copyright ARRS. For personal use only; all rights reserved
1164
GELBER
Fig. 1.-69-year-old
ET AL.
man with ataxia, slurred
AJR:158,
speech,
May 1992
and dizziness.
A, CT scan (bone windows) shows localized left frontal calvarial destruction. B, Unenhanced CT scan shows left frontal vasogenic edema with mass effect on left frontal horn. C, Enhanced CT scan shows lobulated tumor in left and, to a lesser extent, right frontal region, contiguous with area of bone destruction (solid arrow), which has marked peripheral enhancement (open arrow). 0, Photomicrograph of metastatic adenoid cystic carcinoma. Section shows interface of tumor with adjacent brain. (H and E, original magnification x30)
droma tulated
in the gasserian ganglion region has even been posto arise from auditory system mucosal cells. These
reported
lesions
consistent
with
were, the
however,
perineural
basal and therefore invasion
so typical
other organs such as bronchial mucus glands, the prostate, lacrimal glands, Bartholin’s glands, and the uterine cervix [5],
the metastasis found by Hara et al. [6] may not have originated in a salivary gland. Reports of spread of adenoid cystic to the ocular
choroid
plexus
and
brain,
a particularly
were
of these
tumors. The lesion in our case, while partially extraaxial, is in an anatomic position inconsistent with retrograde perineural spread from the primary site in the parotid gland. Furthermore, there was no evidence of contiguous spread of tumor at craniotomy. Koller et al. [5] reported a hematogenous brain metastasis from an adenoid cystic carcinoma of the breast, and Hara et al. [6] reported a hematogenous frontal lobe metastasis of adenoid cystic carcinoma, but no primary tumor was identified. Since adenoid cystic carcinoma can arise in
carcinoma
cystic carcinoma to the meninges unusual manifestation of this tumor.
[7] and to the spinal
cord [8] also have appeared. In summary, our case demonstrates a histologically confirmed hematogenous metastasis of salivary gland adenoid
REFERENCES 1. Parker GD, Hamsberger HR. Clinical and radiologic issues in perineural tumor spread of malignant diseae of the extracranial head and neck. RadioGraphics 1991;1 1:383-399 2. Piepmeier JM, Virapongse C, Kier EL, Kim J, Greenberg A. Intracranial adenocystic carcinoma presenting as a primary brain tumor. Neurosurgery 1983;i2:348-352 3. Lee Y, Castillo M, Nauert C. Intracranial perineural metastasis of adenoid cystic carcinoma of head and neck. J Comput Tomogr 1985:9:219-223 4. Fitzpatrick PJ, Theriault C. Malignant salivary gland tumors. Int J Radiat Oncol Biol Phys i986;12: 1743-1747 5. KOIIer M, Ram Z, Findler G, Lipshitz M. Brain metastasis: a rare manifestation of adenoid cystic carcinoma of the breast. Surg Neurol 1986;26: 470-472 6. Hara H, Tanaka Y, Tsuji T, Momose G, Kobayashi S. Intracranial adenoid cystic carcinoma. A case report. Acta Neurochir (Wien) i983;69:291 -295 7. Gutmann SM, Weiss JS, Albert DM. Choroidal metastasis ofadenoid cystic carcinoma of the salivary gland. Br J Ophthalmol 1986;70: 100-103 8. Riela AR, Meyer D, MCCOOI JA, Pikula L Jr. Metastatic adenoid cystic carcinoma of the major salivary glands presenting as a spinal cord tumor. Surg Neurol 1983;i9:365-368
1163
Case .
.;
Report
.
Intracranial Metastatic Adenoid Cystic Carcinoma: Presumed Hematogenous Spread from a Primary Tumor in the Parotid Gland Nicholas
D. Gelber,1
Ronald
L. Ragland,1
John
R. Knorr,
Intracranial extension of adenoid cystic carcinomas of the head and neck is well recognized, and typically such spread is by way of contiguous perineural invasion [1 -3]. Hematogenous intracranial extension, although reported, is much less common. We report a case of hematogenous metastasis of parotid adenocystic carcinoma to the inner table of the calvaria, epidural compartment, and brain parenchyma.
man presented
with ataxia of several days’ duration,
slurred speech, and some dizziness. Six to eight months earlier, he had undergone resection of a right parotid mass. Subsequent pathologic examination showed an adenocystic carcinoma of the parotid gland, and the patient underwent radiotherapy. No tumor spread was identified at this time. On admission, the man was alert and his vital signs were normal. The patient’s affect was blunted and his mood was depressed. His
gait was slightly
ataxic.
No other focal neurologic
signs were de-
tected. A
CT scan showed
an irregular
in the Ieftfrontal
region.
ial mass
with multiple
Underlying
lesion,
Thomas
W. Smith,2
parenchyma lesion
was
This
was
area of focal calvarial destruction associated
peripheral
with
areas
a lobulated
extraax-
of ring enhancement.
this area, a focal area of vasogenic
type
of white matter
edema was seen in the left frontal lobe with mass effect and compression of the ipsilateral frontal horn (Figs. 1 A-i C). At surgery, an infiltrating lobulated irregular soft-tissue mass was
found involving the left frontal bone, the adjacent
Bernard
of the superficial incompletely
Pathologic
examination
(Fig.
tumor
This
portion
showed
pseudoglandular
1 D).
B. Stone3
of the left frontal
lobe. The
excised.
like structures,
and
the
tumor
tissue
spaces, original
consisting
pseudocysts, pathologic
parotid tumor had virtually identical appearances. No evidence of lung, bone, or liver metastases time.
of lumen-
and necrosis
specimen
of
the
was found at this
dura, and the brain
Adenoid cystic carcinoma (cylindroma, adenocystic carcinoma) is a reasonably common malignant tumor of the large and small salivary glands, constituting approximately 8% of all salivary gland tumors and approximately 22% of malignant salivary gland tumors [4]. While most salivary gland lesions are commonly found in the parotid and submandibular glands, these tumors are relatively more common in the sublingual and accessory salivary glands [4]. The overall frequency of intracranial invasion of adenoid cystic carcinoma has been reported to be between 4% and 22%. However, the vast majority of these tumors spread either directly or via perineural spread [1 -3]. True hematogenous metastases to the CNS are rare, even though hematogenous metastases to liver, lung, and bone are reasonably common late in the course of this disease [3]. Extraaxial intracranial metastatic adenocystic carcinoma mimicking meningioma has been reported [2, 3], and cylin-
Received October 23, 1 991 ; accepted after revision December 4, 1991. 1 Department of Radiology, University of Massachusetts Medical Center, 55 Lake Ave. N., Worcester, 2 Department of Neuropathology, University of Massachusetts Medical Center, Worcester, MA 01655. 3 Department of Neurosurgery, University of Massachusetts Medical Center, Worcester, MA 01655. AJR 158:1163-1164,
and
Discussion
Case Report A 69-year-old
1
May 1992 0361-803x/92/1
585-1 163 © American Roentgen Ray Society
MA 01 655. Address
reprint
requests
to N. D. Gelber.
Downloaded from www.ajronline.org by 164.77.172.146 on 11/12/15 from IP address 164.77.172.146. Copyright ARRS. For personal use only; all rights reserved
1164
GELBER
Fig. 1.-69-year-old
ET AL.
man with ataxia, slurred
AJR:158,
speech,
May 1992
and dizziness.
A, CT scan (bone windows) shows localized left frontal calvarial destruction. B, Unenhanced CT scan shows left frontal vasogenic edema with mass effect on left frontal horn. C, Enhanced CT scan shows lobulated tumor in left and, to a lesser extent, right frontal region, contiguous with area of bone destruction (solid arrow), which has marked peripheral enhancement (open arrow). 0, Photomicrograph of metastatic adenoid cystic carcinoma. Section shows interface of tumor with adjacent brain. (H and E, original magnification x30)
droma tulated
in the gasserian ganglion region has even been posto arise from auditory system mucosal cells. These
reported
lesions
consistent
with
were, the
however,
perineural
basal and therefore invasion
so typical
other organs such as bronchial mucus glands, the prostate, lacrimal glands, Bartholin’s glands, and the uterine cervix [5],
the metastasis found by Hara et al. [6] may not have originated in a salivary gland. Reports of spread of adenoid cystic to the ocular
choroid
plexus
and
brain,
a particularly
were
of these
tumors. The lesion in our case, while partially extraaxial, is in an anatomic position inconsistent with retrograde perineural spread from the primary site in the parotid gland. Furthermore, there was no evidence of contiguous spread of tumor at craniotomy. Koller et al. [5] reported a hematogenous brain metastasis from an adenoid cystic carcinoma of the breast, and Hara et al. [6] reported a hematogenous frontal lobe metastasis of adenoid cystic carcinoma, but no primary tumor was identified. Since adenoid cystic carcinoma can arise in
carcinoma
cystic carcinoma to the meninges unusual manifestation of this tumor.
[7] and to the spinal
cord [8] also have appeared. In summary, our case demonstrates a histologically confirmed hematogenous metastasis of salivary gland adenoid
REFERENCES 1. Parker GD, Hamsberger HR. Clinical and radiologic issues in perineural tumor spread of malignant diseae of the extracranial head and neck. RadioGraphics 1991;1 1:383-399 2. Piepmeier JM, Virapongse C, Kier EL, Kim J, Greenberg A. Intracranial adenocystic carcinoma presenting as a primary brain tumor. Neurosurgery 1983;i2:348-352 3. Lee Y, Castillo M, Nauert C. Intracranial perineural metastasis of adenoid cystic carcinoma of head and neck. J Comput Tomogr 1985:9:219-223 4. Fitzpatrick PJ, Theriault C. Malignant salivary gland tumors. Int J Radiat Oncol Biol Phys i986;12: 1743-1747 5. KOIIer M, Ram Z, Findler G, Lipshitz M. Brain metastasis: a rare manifestation of adenoid cystic carcinoma of the breast. Surg Neurol 1986;26: 470-472 6. Hara H, Tanaka Y, Tsuji T, Momose G, Kobayashi S. Intracranial adenoid cystic carcinoma. A case report. Acta Neurochir (Wien) i983;69:291 -295 7. Gutmann SM, Weiss JS, Albert DM. Choroidal metastasis ofadenoid cystic carcinoma of the salivary gland. Br J Ophthalmol 1986;70: 100-103 8. Riela AR, Meyer D, MCCOOI JA, Pikula L Jr. Metastatic adenoid cystic carcinoma of the major salivary glands presenting as a spinal cord tumor. Surg Neurol 1983;i9:365-368
