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146
Y. Gecrts et al
Neuropediatrics 27 (1996)
of hemisyndromes (11%). Neurological examination revealed
in about 3 3 % of the patients symptoms of hemisyndromes,
cranial nerve palsy (25 %), ataxia and gait disturbances (17 %).
Papilledema was the most common clinical finding (35%).
Choroid plexus carcinomas have no specific
radiological characteristics. On computed tomography, relation
to ventricles is a radiological clue. Choroid plexus carcinomas
usually have irregular margins surrounded by hypodensities
due to edema because of infiltration in adjacent neural tissue. A
marked contrast enhancement is noted (16, 37, 49). Hydro­
cephalus, usually of the obstructive type, is generally less severe
than found in the case of benign choroid plexus papillomas,
where hypersecretion of cerebrospinal fluid occurs (10, 16, 37,
42). Tumor calcification is uncommon (13, 14, 16, 22, 37, 47,
61). Magnetic resonance imaging does not provide more in­
formation about the nature of the tumor than computed
tomography, but facilitates three-dimensional visualization be­
fore surgical treatment, postoperative evaluation of residual
and recurrent tumor, am detection of leptomeningeal metastases (16, 37, 61).
We found 46 cases in the literature with in­
vestigation for dissemination at the moment of diagnosis,
recurrence of the tumor or autopsy (Cases 1 and 2 and ref. 1 ,
4,10, 14,15,17,18, 22, 24,28-30,33, 34, 36, 38-39,41,43,
45,46,52, 54, 55,59-63). Investigation was done with analysis
of the cerebrospinal fluid for malignant cells and/or lumbar
myelography and, more recently, with magnetic resonance
imaging. Twenty-two patients (48%) did not show cerebro­
spinal dissemination. Sub arachnoid seeding was found in 20
patients (43%). Occasionally metastases outside the central
nervous system are described: pleura and lung (63), ascitic
fluid (46), peritoneum (46) and bone (17, 33)* We report one
case with several metastatic tumors in the omentum, bladder,
uterus and rectum. We agree with Griffith that the real
incidence of cerebrospinal dissemination is not known (30).
Staging studies evaluating the entire neuraxis are particularly
important after local tumor recurrence is proven, but a negative
result does not exclude the development of subsequent metas­
tases (38).
The diagnosis of choroid plexus carcinoma is
based on histological examination; in typical cases, the tumor
shows architectural disarray with poorly formed papillae, high
cellularity, severe cytonuclear atypia and a high mitotic index
(12). These carcinomas usually show immunohistochemical
staining with epithelial markers (keratin, Cam 5.2, EMA), but
compared to plexus papillomas staining with markers for glial
differentiation (SI00, GFAP) is diminished or absent (2,19,21,
51, 58). Like in plexus papillomas, in plexus carcinomas
ultrastructural features of normal choroid plexus epithelium
(basal lamina, interdigitation of cell membranes, apical micro­
villi, desmosomes, scattered cilia) may be present (25, 47). The
distinction between plexus carcinomas and plexus papillomas is
sometimes difficult, atypical plexus papillomas may show sig­
nificant cytonuclear atypia and scattered mitotic figures (12). In
our two cases the severe cytonuclear atypia and high mitotic
index qualified these tumors as plexus carcinomas. Histopathologically, other malignant tumors like malignant ependymoma
and metastatic carcinoma may resemble a choroid plexus
carcinoma. In contrast to plexus carcinomas, malignant epen­
dymomas generally show clear-cut glial differentiation and
GFAP-staining (12, 48).
Table 3
up).
Treatment and outcome: disease-free survival period H follow-
Surge ty
Adjuvant therapy
Complete resection
n
t()
None
Radiotherapy
Chemotherapy
Radio- i- chemotherapy
10
10
5
1
60 (5-1181
64 (4-240)
54 (8-156)
12
Total
26
58
Partial resection
n
t()
—
4
6
—
10
—
32 (12-48)
34 (5-72)
33
n: number of patients; ( j; range of disease-free survival period (In months);
t: median disease-free survival period
Reviewing the literature it is not possible to
establish the optimal therapy for choroid plexus carcinomas.
Therapy was described for 121 of the 231 patients. Four
children just underwent inspection of the brain; follow-up data
were not available for 2 patients. A group of 25 (21 %) patients
died preop era tively or within two months after surgery; they are
considered as perioperative deaths and are excluded from
Table 3. The treatment varied from surgical resection alone to
more aggressive therapy with surgery followed by radiotherapy
and/or chemotherapy. The follow-up period ranged from
3 months to 20 years. Most authors described gross total,
subtotal or partial resection of the tumor. However, in many
cases postsurgical evaluation of the tumor is not available.
Information about the exact field and dosage of radiotherapy
is often lacking and the chemotherapeutic scheme varies widely.
A group of 28 patients who died within the period of follow-up,
had a median survival of 13 months after diagnosis. Surgery
without adjuvant therapy can be curative as shown in Table 3
(Case 1 and ref. 1 , 4-6, 10, 1 1 , 14, 17, 18, 20, 22-25, 28, 32,
39, 50-52, 56, 60, 64). Table 3 indicates that, whenever pos­
sible, complete resection of the choroid plexus carcinoma has to
be pursued. We did not find a difference between surgery alone
and surgery followed by radiotherapy when complete resection
was performed. This was also noted by Packer et al and Pierga
et al (52, 56). The shorter median relapse-free period for
patients who underwent complete resection followed by che­
motherapy may be due to the more recent use of chemother­
apeutic treatment as adjuvant therapy Adjuvant therapy fol­
lowing surgery of choroid plexus carcinoma is useful when only
partial resection of the tumor was obtained. There is no
difference in disease-free interval between the groups receiving
either chemotherapy or radiotherapy after partial resection:
median period of 34 and 32 months respectively (Table 3).
No difference could be found between children and adults
concerning the outcome of adjuvant therapy as there were only
a few adults amongst the 16 reported cases with a detailed
follow-up history after diagnosis. Knowing the possible long­
term sequelae of irradiation with intellectual deterioration,
leukoencephalopathy, endocrinopathy, especially in the very
young children, delay of radiotherapy is sought until the age
of at least four years is reached. This delay of radiotherapy is
important because 44% of the patients with choroid plexus
carcinoma are younger than 2 years. Several schemes of drugs
have been used but the value of each of them can not be
evaluated due to the small number of patients in each scheme.
Because of her age, our youngest patient received chemother­
apy after gross total resection of the tumor. Interestingly, the
metastasis which was present at the moment of diagnosis
disappeared under chemotherapy whereas ten months after
Choroid Plexus Carcinoma
surgery several intracerebral, spinal and cranial leptomeningeal
metastases had occurred. Reviewing the literature, leptomenin­
geal seeding of the tumor can disappear after craniospinal
radiotherapy (54, 56, 59), whereas chemotherapy is less favor­
able (1, 10, 24, 52).
Conclusion
Reviewing the literature, initial staging studies
evaluating the entire neuraxis should be performed at the
moment of diagnosis of a choroid plexus carcinoma, as sub­
arachnoid seeding occurs in almost half of the patients. Even
under chemotherapeutic treatment, peritoneal metastases can
occur by ventriculo-peritoneal shunting. No difference could be
found between radiotherapy and chemotherapy following par­
tial resection of the tumor, but radiotherapy should be delayed
as much as possible for the youngest patients because of
possible long-term sequelae. In the past, several chemothera­
peutic schemes have been used and therefore more cases or
protocols are needed to decide for the best therapy.
Neuropediatrics 27 (1996)
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17
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IK
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27
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30
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2-1
But this review suggests that whenever possible
complete resection of the tumor has to he pursued, and no
further therapy is necessary.
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R , Lippens, M. D.
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