A long survival of a patient with brain metastasis of unknown site of the primary tumor
Eighty percent of brain metastases (BM) are diagnosed in patients with known primary site of cancer. BM of unknown primary represents a difficult diagnosis. In up to 15% of patients with BM, the site of the primary tumor will not be detected despite investigations. The prognosis of this entity is ve...
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| Опубліковано в: : | Experimental Oncology |
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Інститут експериментальної патології, онкології і радіобіології ім. Р.Є. Кавецького НАН України
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| Цитувати: | A long survival of a patient with brain metastasis of unknown site of the primary tumor / W. Ben Kridis, S. Sghaier, N. Toumi, Z. Boudawara, A. Khanfir, J. Daoud, M. Frikha // Experimental Oncology. — 2018 — Т. 40, № 1. — С. 85–87. — Бібліогр.: 29 назв. — англ. |
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Ben Kridis, W. Sghaier, S. Toumi, N. Boudawara, Z. Khanfir, A. Daoud, J. Frikha, M. 2018-06-19T21:13:01Z 2018-06-19T21:13:01Z 2018 A long survival of a patient with brain metastasis of unknown site of the primary tumor / W. Ben Kridis, S. Sghaier, N. Toumi, Z. Boudawara, A. Khanfir, J. Daoud, M. Frikha // Experimental Oncology. — 2018 — Т. 40, № 1. — С. 85–87. — Бібліогр.: 29 назв. — англ. 1812-9269 https://nasplib.isofts.kiev.ua/handle/123456789/139253 Eighty percent of brain metastases (BM) are diagnosed in patients with known primary site of cancer. BM of unknown primary represents a difficult diagnosis. In up to 15% of patients with BM, the site of the primary tumor will not be detected despite investigations. The prognosis of this entity is very poor. We report here a case of a long survival of a patient with brain metastasis of unknown primary. The conclusion that can be drawn is that within BM of unknown primary exist patients with a very good prognosis that must be collected and published in order to base recommendations. en Інститут експериментальної патології, онкології і радіобіології ім. Р.Є. Кавецького НАН України Experimental Oncology Case report A long survival of a patient with brain metastasis of unknown site of the primary tumor Article published earlier |
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Digital Library of Periodicals of National Academy of Sciences of Ukraine |
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| title |
A long survival of a patient with brain metastasis of unknown site of the primary tumor |
| spellingShingle |
A long survival of a patient with brain metastasis of unknown site of the primary tumor Ben Kridis, W. Sghaier, S. Toumi, N. Boudawara, Z. Khanfir, A. Daoud, J. Frikha, M. Case report |
| title_short |
A long survival of a patient with brain metastasis of unknown site of the primary tumor |
| title_full |
A long survival of a patient with brain metastasis of unknown site of the primary tumor |
| title_fullStr |
A long survival of a patient with brain metastasis of unknown site of the primary tumor |
| title_full_unstemmed |
A long survival of a patient with brain metastasis of unknown site of the primary tumor |
| title_sort |
long survival of a patient with brain metastasis of unknown site of the primary tumor |
| author |
Ben Kridis, W. Sghaier, S. Toumi, N. Boudawara, Z. Khanfir, A. Daoud, J. Frikha, M. |
| author_facet |
Ben Kridis, W. Sghaier, S. Toumi, N. Boudawara, Z. Khanfir, A. Daoud, J. Frikha, M. |
| topic |
Case report |
| topic_facet |
Case report |
| publishDate |
2018 |
| language |
English |
| container_title |
Experimental Oncology |
| publisher |
Інститут експериментальної патології, онкології і радіобіології ім. Р.Є. Кавецького НАН України |
| format |
Article |
| description |
Eighty percent of brain metastases (BM) are diagnosed in patients with known primary site of cancer. BM of unknown primary represents a difficult diagnosis. In up to 15% of patients with BM, the site of the primary tumor will not be detected despite investigations. The prognosis of this entity is very poor. We report here a case of a long survival of a patient with brain metastasis of unknown primary. The conclusion that can be drawn is that within BM of unknown primary exist patients with a very good prognosis that must be collected and published in order to base recommendations.
|
| issn |
1812-9269 |
| url |
https://nasplib.isofts.kiev.ua/handle/123456789/139253 |
| citation_txt |
A long survival of a patient with brain metastasis of unknown site of the primary tumor / W. Ben Kridis, S. Sghaier, N. Toumi, Z. Boudawara, A. Khanfir, J. Daoud, M. Frikha // Experimental Oncology. — 2018 — Т. 40, № 1. — С. 85–87. — Бібліогр.: 29 назв. — англ. |
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2025-11-25T20:35:54Z |
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Experimental Oncology 40, 85–87, 2018 (March) 85
A LONG SURVIVAL OF A PATIENT WITH BRAIN METASTASIS
OF UNKNOWN SITE OF THE PRIMARY TUMOR
W. Ben Kridis1, *, S. Sghaier1, N. Toumi1, Z. Boudawara2, A. Khanfir1, J. Daoud3, M. Frikha1
1Department of Oncology, Habib Bourguiba Hospital, Sfax 3029, Tunisia
2Department of Neurosurgery, Habib Bourguiba Hospital, Sfax 3029, Tunisia
3Department of Radiotherapy, Habib Bourguiba Hospital, Sfax 3029, Tunisia
Eighty percent of brain metastases (BM) are diagnosed in patients with known primary site of cancer. BM of unknown primary
represents a difficult diagnosis. In up to 15% of patients with BM, the site of the primary tumor will not be detected despite in-
vestigations. The prognosis of this entity is very poor. We report here a case of a long survival of a patient with brain metastasis
of unknown primary. The conclusion that can be drawn is that within BM of unknown primary exist patients with a very good
prognosis that must be collected and published in order to base recommendations.
Key Words: brain metastases, unknown primary, prognosis.
Eighty percent of brain metastases (BM) are diag-
nosed in patients with known primary site of cancer.
BM of unknown primary (BMUP) represents a difficult
diagnosis [1, 2]. In up to 15% of patients with BM, the site
of the primary tumor will not be detected despite investi-
gations [3]. Even though the primary remains undefined
during the follow up, some of these patients found to have
a favorable prognosis. In fact, cancer of unknown primary
(CUP) has been divided into favorable group (20%) and
unfavorable group (80%) based on histopathology and
clinical manifestations [2]. We report here a case of an ex-
ceptional long survival of a patient with BMUP.
CASE REPORT
A 50-year-old man was referred to our hospital in Oc-
tober 2002 because of increased headaches, vomiting
and dizziness evolving for two weeks. Glasgow score was
quoted at 14/15. Neurological examination revealed ki-
netic cerebellar syndrome. Fundus examination revealed
a peripapillary hemorrhage in the right eye. Brain mag-
netic resonance imaging (MRI) showed a 4 × 2 cm lesion
of the right cerebellum with a T1 weighted hyposignal,
T2 weighted hypersignal and a heterogeneous enhance-
ment. This lesion compressed the brainstem and the
fourth ventricle causing a triventricle expansion with signs
of active hydrocephalus (Fig. 1). The patient underwent
a complete resection of the tumor. The pathologic study
showed polyhedral tumor cells arranged in solid sheets
with a clear abundant cytoplasm. The tumor stroma had
a vascular component of endocrine type without necro-
sis. The antibody panel consisted of reagents directed
to antigens: CK7, CK20, EMA, NSE, chromogranin,
synaptophysin, glial fibrillary acidic protein (GFAP), TTF1,
ACE and vimentin. The immunohistochemical staining
was positive for vimentin, NSE, TTF1, CEA but negative
for GFAP. These results were compatible with cerebellar
metastasis from a clear cell carcinoma. After surgery, the
patient maintained a minimal static cerebellar syndrome.
Brain imaging showed no residual tumor. No whole brain
radiation was delivered. In order to detect the primary
cancer, lung auscultation, thyroid examination as well
as organs of abdomen examination and digital rectal
exam were normal. Full blood counts were normal. Total
body computed tomography (CT) was normal. Bron-
chofibroscopy revealed an inflammatory aspect of the
left bronchi and its biopsy showed chronic inflamma-
tion. Positron emission tomography (PET) scan was not
available in 2002. Tumor markers levels including PSA,
CEA, CA19-9, and NSE were also normal. The patient
was discharged with physical examination and CT every
4 months in the first year, then every six months during
two years and then annually. In June 2014, CT scan re-
vealed a nonspecific 5 mm node in the right lung. Brain
CT showed no recurrent disease (Fig. 2). Actually, our
patient remains asymptomatic with no recurrent disease
14 years after the diagnosis of BM.
Fig. 1. Cerebral MRI: cerebellar metastasis
Fig. 2. Cerebral CT: cerebellar porencephalic cavity after surgery
with no recurrence
Submitted: December 23, 2017.
*Correspondence: E-mail: walabenkridis@yahoo.fr
Tel.: +21694492526
Abbreviations used: BM — brain metastases; BMUP — BM of unknown
primary; CT — computed tomography; CUP — cancer of unknown
primary; GFAP — glial fibrillary acidic protein; KPS — Karnofsky per-
formance status; MRI — magnetic resonance imaging; PET — positron
emission tomography; WBRT — whole brain radiation therapy.
Exp Oncol 2018
40, 1, 85–87
86 Experimental Oncology 40, 85–87, 2018 (March)
DISCUSSION
The definition of CUP includes patients with histo-
logical confirmed metastatic cancer in whom a detailed
medical history, thorough physical examination and
complete laboratory investigations fail to identify the
primary site [2]. This is due to the fact that the pre-
sumed primary tumor is able to metastasize before
becoming large enough to be identified [7]. BMUP
represents 15% of all BM [4–6]. In about 50% of BMUP
cases, primary lung cancer is found to be the most
frequent primary site (51%) [8]. Although the breast
is the second most primary site in BM (10–17%). In the
reported cases of BMUP, melanoma represented 8%,
the pancreatic cancer was found in 8% and gastroin-
testinal cancers in 19% cases [9–12]. In our patient
histopathological study with immunohistochemistry
was compatible with cerebellar metastasis from a clear
cell carcinoma. However, exploration did not find the
primitive.
About 85% of BMUP are found in the cerebral
hemispheres, 10–15% found in the cerebellum and
3% in the brainstem. In our case, the BM was located
in the right cerebellum. Certain primary tumors such
as those originating from kidney and colon are more
likely to metastasize to the cerebellum.
As for the clinical presentation, the mean age
of BMUP’s patients ranges from 50 to 60 years with
a male predilection. The predominant presenting
symptom in BM is headaches as it is the case of our
patient. Fifty percent of patients with BM have motor
or language deficits and 10–15% of them present
seizures [13]. Approximately, 10% of BM are asymp-
tomatic and the diseases’ spread is identified through
routine imaging [14, 15].
The preferred modality for detecting brain lesions
is MRI. Despite the accuracy of MRI, CT imaging is of-
ten used when MRI is either not available or not suit-
able for particular patients [16]. Despite the sensitivity
of MRI, 11% of patients with a brain lesion are given
a false-positive diagnosis of either a BM or a primary
brain cancer [17–20].
The dilemma of distinguishing between a BM and
a primary brain tumor can only be solved by biopsy
or tumor resection with histopathological study [21,
22]. The choice between biopsy and surgical resection
depends on many factors: number of brain lesions, its
location, patient’s general condition (Karnofsky per-
formance status — KPS, age, comorbidity) and pres-
ence or not of accessible extra-cranial metastases.
In our case, cerebral MRI showed a single lesion of the
right cerebellum, so that he underwent a complete
resection of the tumor.
In order to detect the primary cancer, physical
examination was normal. Full blood counts were nor-
mal. Total body CT was normal. Bronchofibroscopy
revealed an inflammatory aspect of the left bronchi
and its biopsy showed chronic inflammation. PET scan
was not available in 2002. Nowadays, the combination
of PET/CT plays an important role to search the primi-
tive in case of CUP [23, 24].
In our case, the immunohistochemical staining was
positive for vimentin, NSE, TTF1, CEA but negative for
GFAP. These results concluded to cerebellar metasta-
sis from a clear cell carcinoma.
In the study of Drlicek et al. [25], 40 cases
of BM from known primary tumors were blinded and re-
evaluated based on the immunohistochemical staining
pattern. An established panel of antibodies was used
including: CK AE1/AE3, CK7, CK20, CK10/13, CK18,
vimentin, S100, TTF-1, surfactant, PSA, CA15-3,
CA125, and CA19-9. This panel of 13 different an-
tibodies was able to identify the primary in 29 out
of 40 BM correctly (72.5%). In this series, 1/11 of lung
cancer BM had a CK7 negative-CK20 negative profile.
The treatment of BM is an oncologic emergency. The
recommendation of the subcommittee of the American
Academy of Neurology (AAN) is that in patients with newly
diagnosed brain tumor prophylactic anticonvulsants
should not be used, it has to be initiated from the time
of the first seizure [26]. For patients who undergo surgery,
AAN recommends to stop them in 1 to 4 weeks postop-
eratively. All patients should have corticotherapy. Patients
with single BM, good KPS and no disseminated disease,
should have surgery or radiosurgery (if lesion less than
3 cm) to achieve local tumor control. Radiosurgery is less
invasive and gives the same result as with surgery but
it does not provide tissue diagnosis. Postoperative whole
brain radiation therapy (WBRT) helps to sterilize the other
areas and improves survival [18]. In case of multiple BM,
WBRT is given usually in doses of 30 Gy in 10 fractions
or 20 Gy in 5 fractions. Chemotherapy has a relatively
small role in case of BMUP. In fact, only few molecules
could cross the blood-brain barrier.
Our patient was aged less than 65 years, he had
a KPS > 70, his BM was single and completely resected
and finally, he had no other metastases. All these fac-
tors explain the good evolution and the long survival
of this patient [27, 28].
In a retrospective study, the median overall survival
for patients with a solitary BMUP was 7.3 vs 3.9 months
for patients with multiple metastases (p = 0.05). Pa-
tients who underwent resection of BM before WBRT
had a significant better overall survival than those who
underwent WBRT alone (9.5 vs 3.6 months median sur-
vival, respectively) [15]. The majority of these patients
die from extracranial disease and only a minority (15%)
will live more than five years [29]. To our knowledge,
our patient represents the first case described in the
literature with an overall survival exceeding 10 years.
CONCLUSIONS
BMUP is an ambiguous disease. It poses a problem
for the oncological patient and his doctor. The progno-
sis of this entity is very poor. This case is exceptional;
firstly given its rarity and secondly due to the long sur-
vival that exceeded 10 years. The conclusion that can
be drawn is that within BMUP exist patients with a very
good prognosis that must be collected and published
in order to base recommendations.
Conflict of interest. None.
Experimental Oncology 40, 85–87, 2018 (March) 87
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