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Arq Neuropsiquiatr 2001;59(2-B):424-430
INFRASELLAR CRANIOPHARYNGIOMA
Case report
Asdrubal Falavigna1, Jorge Luiz Kraemer2
ABSTRACT - We report a case of infrasellar craniopharyngioma in a 34 year-old woman who presented withprogressive headache and diplopia. Computed tomographic and magnetic resonance images showed aheterogeneous tumor originating from the sphenoid bone with ethmoid sinus and sella turcica extension. Asublabial rhinoseptal transsphenoidal surgery was performed. Craniopharyngiomas with infrasellar developmentare very rare. Infrasellar craniopharyngioma is uncommon, thirty-five cases has been reported in literature.The embryology, clinical features and radiographic investigation of these tumors are discussed.
KEY WORDS: craniopharyngioma, sphenoid sinus, skull base tumor.
Craniofaringioma infra-selar: relato de caso
RESUMO – Relatamos um caso de craniofaringioma infra-selar em uma paciente de 34 anos com sintomas decefaléia e diplopia. A investigação radiológica com tomografia computadorizada e ressonância magnética deencéfalo demonstrou um tumor heterogêneo localizado no osso esfenoidal e com extensão para o seio etmoidale sela turcica. Realizada cirurgia pela via transesfenoidal. A ocorrência de craniofaringioma com topografiainfra-selar é incomum, havendo relato na literatura de 35 casos. A literatura é revisada, sendo discutidas aembriologia, a apresentação clínica e características radiológicas do tumor.
PALAVRAS-CHAVE: craniofaringioma, seio esfenoidal, tumor de base de crânio.
1Professor Assistente da Disciplina de Neurologia da Faculdade de Medicina da Universidade de Caxias do Sul, Pós-Graduando emNeurocirurgia da Universidade Federal de São Paulo - Escola Paulista de Medicina, São Paulo SP, Brasil (UNIFESP-EPM); 2Professor docurso de Pós-Graduação em Cirurgia da Universidade Federal do Rio Grande do Sul (UFRGS) e da Fundação Faculdade de CiênciasMédicas de Porto Alegre (FFCMPA-ISCMPA), Mestre pela UFRGS e Doutor em Neurocirurgia pela UNIFESP- EPM.
Received 14 November 2000, received in final form 18 January 2001. Accepted 29 January 2001.
Dr. Asdrubal Falavigna - Rua Dr. Moreira César 271/sala.1 - 95034-000 Caxias do Sul RS - Brasil. E-mail: asdrubal@doctor.com
Craniopharyngioma is a rare tumor1. It arises wi-thin the sella turcica and expands mainly into thesuprasellar region2. Occasionally, the tumor can oc-cur without sellar involvement. These tumors rarelyextend below the sellar floor into the sphenoid si-nus or invade the pharynx and the nasal cavities3-34.The infrasellar craniopharyngioma may then origi-nate anywhere along the tract of the obliteratedcraniopharyngeal duct, which would include thesphenoid bone, vomer, and nasopharynx35,36.
In this study we report a case of infrasellar cranio-pharyngioma with the epicenter situated in the sphe-noid sinus and review the 35 other reported cases inthe literature of this unusual localization since 1924.
CASEA 34 years-old woman presented with frontal head-
ache with progression of 7 months and diplopia that ap-peared in the last month. Clinical evaluation revealed leftabducens nerve palsy. No signs and symptoms of pitu-
itary dysfunction were evident. The laboratory studies,including hypophyseal function were normal.
Coronal computed tomographic (CT) scans after intra-venous contrast showed a large sphenoid sinus heteroge-neous mass with destruction of the ethmoid sinus andsella turcica (Fig 1). T1-weighted (plain and gadolinium-enhanced 600/11/2 [repetion-time/echo time/excitations])and T2-weighted (3000/108/1) magnetic resonance sho-wed isointense image (Fig 2). Post-contrast the brightly en-hancing, heterogeneous mass was clearly visible in the sphe-noid bone (Fig 3). There was extension of the tumor toethmoid sinus and to the sella turcica. Carotid and verte-bral angiography revealed that the tumor was avascular.
Sublabial rhinoseptal transsphenoidal surgery was per-formed. After removal of the anterior wall of the sphe-noid sinus, a solid firm mass was filled the sphenous si-nus. The circumferential wall of the tumor was firmly atta-ched to the cavernous sinus, with profuse bleeding dur-ing the maneuver. A subtotal removal was performed. Atthe end of the tumor resection, posterior pharyngeal pack-ing was left in place. Her postoperative course was good
Arq Neuropsiquiatr 2001;59(2-B) 425
Fig 1. CT enhanced axialview showing a large het-erogeneous mass arisingfrom the sphenoid bonewith destruction of the eth-moid sinus and sella turcica.
Fig 2. Sagittal magneticresonance in T1-weightedimage without gadoliniumadministration showing anisointense tumor in thesphenoid bone with exten-sion to the sella turcica.
and the nasal packing removed on the third postoperativeday. The patient was discharged from the hospital on theeight postoperative day with recovery of the abducens ner-ve palsy and headache-free. Histologic examination re-vealed an adamantonomatous craniopharyngioma (Fig 4).
The patient was referred for radiotherapy. Conforma-tional radiotherapy consisting of a total of 55Gy (30 frac-tions) was administered 1 month later. At clinical follow-up, 2 year after irradiation, her neurological examinationand hormonal studies were normal.
DISCUSSIONCraniopharyngiomas are benign but aggressive
epithelial neoplasms, which comprises approximately
3% of all intracranial tumors1. They are generallyfound intracranially with a similar frequency in chil-dren and adults but with a slight preponderance inchildren between 5 and 15 years of age2. Equal sexincidence has been noted in groups of children2.Craniopharyngiomas are most commonly located ex-traaxially in the sellar or suprasellar area in 90% ofcases2. They can extend to the anterior (2-5% ofcases), middle (2%), or posterior (1-4%) cranial fossa,and infrasellar extension is found in about 5% ofcases34. Rarely, craniopharyngiomas arise primarilyin unusual locations, such as the nasopharynx, sphe-noid bone, third ventricle, pineal gland, sylvian fis-
426 Arq Neuropsiquiatr 2001;59(2-B)
sure and cerebellopontine angle3-34,37-43. The growthof a craniopharyngioma in the infrasellar region isextremely rare, only 36 cases were found in the lit-erature (Table 1).
The infrasellar craniopharyngioma reported in lit-erature has a similar sex distribution, being 3 timesmore frequent above 15 years of age. The micros-copic appearance of most craniopharyngiomasshows an external layer of high columnar epithe-lium, a variable amount of polygonal cell, and a cen-tral network of epithelial cells. Two clinicopathologicvarieties have been delineated microscopically: the
papillary squamous type, which is seen almost ex-clusively in adults and carries a better prognosis; andthe classical adamantinomatous variant, which de-velops mainly in children and has a worse overalloutcome41.
Treatment of these tumors is mostly excisional,through a craniotomy or transsphenoid approach.Complete removal of the tumor is the preferred pro-cedure2,29. When vital structures are involved andexcision is compromised by significant risks of mor-bidity and mortality, subtotal removal of the tumorfollowed by supplemental radiotherapy is the pre-
Fig 3. T1-weighted gado-linium enhanced axial MRIscans, showing a heteroge-neous enhancing sphenoidsinus mass with extensioninto the ethmoid sinus.
Fig 4. Photomicrographyof adamantinomatous cra-niopharyngioma showinga epithelial componentand peripheral palisadingcells (hematoxylin andeosin, original magnifica-tion x 125)
Arq Neuropsiquiatr 2001;59(2-B) 427
ferred treatment29. Radiotherapy, once consideredineffective, is currently the most often used adju-vant treatment. Craniopharyngiomas have beenfound to be radiosensitive, and radiotherapy plays amajor role in preventing recurrence and improvingsurvival42.
Embryology - According to Warwick and Will-iams43, during the fourth week of gestation anectodermally lined diverticulum (Rathke’s pouch)develops in the roof of the stomodeum just anteriorto the oropharyngeal membrane. This pouch ascendscranially traversing the mesenchyme to meet theneuroectoderm of the infundibulum (neurohypo-physis), which descends as a neural outgrowth fromthe floor of the third ventricle (diencephalon) of theembryonic brain. Rathke’s pouch differentiates intothe anterior lobe of the pituitary, the adenohypo-physis. This course traversed by Rathke’s pouch formsa cord of cells joining the stomodeal ectoderm tothe future adenohypophysis in embryo. Later inembryonic life this cord disintegrates, leaving anobliterated craniopharyngeal canal. It is a tract thatruns from the anterior part of the hypophyseal fossaof the sphenoid bone to the junction of the poste-rior septum of the nose with the palate, which is thestomodeal end of the recess. The pharyngeal hypo-physis (functioning adenohypophyseal tissue), whichremains in adults, is a caudal remnant of this cord.
Types of origin - There are several theories regard-ing the origin of infrasellar craniopharyngioma. Mostof them are based on the embryologic developmentof the adenohypophysis. Mott and Barret35 in 1899were the first to postulate that these tumors mightarise from the remnants of the pharyngeal hypophy-sis. Erdheim36 proposed in 1904 that the craniophar-yngioma originated from the remnants of the oblit-erated craniopharyngeal duct, suggesting that thesetumors can arise anywhere along the tract of migra-tion of Rathke’s pouch from the vomer, the roof ofthe nasopharynx, through the midline sphenoid bonebeneath the floor of the sella turcica.
Clinical presentation - Symptoms and clinical find-ings are related to the craniopharyngioma’s local-ization and mass effect with compression of the sur-rounding structures (Table 1). The symptomatologyof suprasellar tumor is often characterized by de-fects in the visual fields (bitemporal or homonimushemianopsia), varying signs of pituitary insufficiency(diabetes insipidus, amenorrhoea, diminished libidoand cachexia), and the symptoms of increased in-
tracranial pressure (headache, vertigo and cranialnerve deficit) that occur relatively late in the courseaccording to Carmel et al.2 and Sato quoted byFujitani et al.16. The signs of pituitary dysfunctionappear early in the cases in which the tumor expandswithin the sella. The tumor located at the sphenoidsinus usually presents with headache and cavernoussinus syndrome6-8,11,12,15,17,20,23,24,27. The craniopharyn-giomas located in the nasopharyngeal region usu-ally present with frontal headache, nasal obstruction,epistaxis, nasopharyngeal and/or nasal fossa mas-ses3,5,9,10,13,15,18,19,22,25,26,29,32,34. Our case had symptomsof headache, diplopia due to left abducens nervepalsy, without pituitary insufficiency, which suggeststhat the tumor had appeared in the sphenoid boneand, later, extended to the sella turcica region.
Localization - Craniopharyngiomas usually origi-nate intracranially, grow along the pituitary stalk,on the axis of the sella-infundibulum and locatedbelow the brain, above the pituitary, behind the opticchiasma, and within the circle of Willis. Occasionallythere is an intrasellar one and, rarely, one within thebody of the sphenoid or nasopharynx in the tract ofthe former craniopharyngeal canal proper. Since1924, 35 cases of infrasellar involvement by cranio-pharyngioma have been described in the medical lit-erature. In these cases the tumors are situated atthe nasopharyngeal region, the sphenoid sinus, themaxillary sinus and usually involved the sella turcicaand the supra sellar region. The most common loca-tion of infrasellar craniopharyngioma has been thesphenoid sinus either alone, 4 cases, or combinedwith other sites, 28 cases (Table 1). The tumor loca-tion in the case presented could have been exten-sions from the sella turcica or derivatives of pharyn-geal canal remnants. In the former situation, thetumor could be a typical craniopharyngioma thatarose in the sella turcica and then went downwardsinto the sphenoid sinus. In the latter, the tumor origi-nated in the infrasellar region, sphenoid sinus, andthen grew extradurally and superiorly. We suspectthat this tumor was a craniopharyngioma that origi-nated in the infrasellar region because it is situatedmainly in the sphenoid sinus, the pituitary hormoneswere always normal and we could not find the duramater of the sellar floor during the transsphenoidalapproach.
Diagnostic evaluation - Skull x-ray study, tomo-grams, and CT were the most common radiographictechniques employed in evaluation. Infrasellar cran-iopharyngioma cases had the plain skull x-rays and
428 Arq Neuropsiquiatr 2001;59(2-B)
Tabl
e 1.
Inf
rase
llar
cran
ioph
aryn
giom
.
Aut
hor
Year
Patie
nt A
ge/S
exTu
mor
Loc
atio
n S
ymto
ms
& S
igns
Radi
ogra
phic
Inv
esti
gati
on
Boch
(3)
1924
51/M
NPX
Hea
dach
e, n
asal
obs
truc
tion
Skul
l x-r
ay (
Nec
rops
y)
Zeit
lin (
4)19
3540
/MSS
, ST
, m
iddl
e fo
ssa
(L),
Diff
icul
ties
in p
hona
tion,
fac
e pa
in (
L),
anis
ocor
ia,
Nec
rops
y
pet
rous
bon
e (L
)in
tern
al r
ectu
s m
uscl
e pa
lsy
(R),
ptos
is (
R),
loss
or
decr
ease
d vi
sion
, fa
cial
par
alys
is (
L),
tong
ue d
evia
ted
to le
ft s
ide
Dru
mm
ond
(5)
1938
14/F
NPX
, SS
, ST
Nas
al o
bstr
ucti
on,
head
ache
, lo
ss o
r de
crea
sed
Skul
l x-
ray
visi
on,
NPX
and
NF
mas
s
Sato
acc
. Fu
titan
i19
4456
/FN
PX,
SS,
ST,
SV
isua
l lo
ssSk
ull
x-ra
y
et a
l. (1
6) 6
)
Nor
thfie
ld (
6)19
5754
/FSS
Hea
dach
e, v
omiti
ng,
hom
onym
ous,
hem
iano
pia
(L)
Skul
l x-
ray
Ham
berg
er e
t al
. (7
)19
6025
/MSS
Hea
dach
es,
face
hyp
oest
hesi
a in
ter
ritor
ySk
ull
x-ra
y
of in
frao
rbit
al n
erve
(R)
John
son
(8)
1962
39/M
SS,
ST,
SH
eada
che,
dip
lopi
a, v
isio
n lo
ss (
R)Sk
ull
x-ra
y, t
omog
ram
Podo
shin
et
al.
(9)
1970
15/F
NPX
, SS
, ST
Nas
al o
bstr
ucti
on,
NPX
and
NF
mas
sSk
ull
x-ra
y
Isay
ama
(11)
1970
62/F
SS,
ST,
SFa
cial
pai
n, d
iplo
pia
Skul
l x-
ray
Trib
le (
13)
1970
71/M
NPX
, SS
, ES
, ST
Hea
dach
es,
dipl
opia
, na
sal
obst
ruct
ion,
epi
stax
isSk
ull
x-ra
y
Coo
per
& R
anso
hoff
(14
)19
7216
/MN
PX,
SS,
S (m
iddl
e fo
ssa)
Hea
dach
e, e
pist
axis
, CS
F rh
inor
rhea
, di
plop
ia,
lack
of
Skul
l x-
ray
hea
ring
(L),
palp
able
mas
s in
the
zyg
omat
ic a
rea
(L)
Pras
ad &
Kw
i (10
)19
7555
/FN
PX,
SSN
asal
obs
truc
tion
, N
PX a
nd N
F m
ass
Skul
l x-
ray,
tom
ogra
m,
angi
ogra
m
48/M
NPX
, SS
, ST
Nas
al o
bstr
uctio
n, h
eada
che,
dip
lopi
a w
ithSk
ull
x-ra
y, t
omog
ram
late
ral r
ectu
s w
eakn
ess
(L),
faci
al h
ypoe
sthe
sia
in t
errit
ory
of in
frao
rbit
al n
erve
(L)
, N
PX a
nd N
F m
ass
Ishi
yam
a (1
2)19
7741
/FSS
, ST
, S
Faci
al p
ain
Skul
l x-
ray,
tom
ogra
m
25/M
SSH
eada
che,
dip
lopi
aSk
ull
x-ra
y, t
omog
ram
Illum
et
al.
(15)
1977
14/F
NPX
, SS
, ST
Hea
dach
e, d
iplo
pia
wit
h ab
duce
ns n
erve
pal
sy (
L),
Tom
ogra
m,
angi
ogra
m,
tem
pora
l he
mia
nops
ia (
L)pn
eum
o-en
ceph
alog
raph
y
Maj
less
i et
al/ (
18)
1978
17/M
NPX
, SS
, ST
, S
Hea
dach
e, b
item
pora
l he
mia
nops
ia,
Skul
l x-
ray
exop
htha
lmos
, hy
popi
tuit
aris
m,
NPX
mas
s
Arq Neuropsiquiatr 2001;59(2-B) 429
Fujit
ani e
t al
. (
16)
1979
18/F
NPX
, SS
, ES
, M
S, o
rbit
(R)
Exop
htha
lmos
, vi
sual
dis
turb
ance
Skul
l x-
ray,
ang
iogr
am,
scin
tigr
aphy
, cy
stog
raph
y C
T
Phel
ine
et a
l. (2
0)19
8112
/MSS
Hea
dach
e, d
iplo
pia
wit
h ab
duce
nt n
erve
pal
sySk
ull
x-ra
y, a
ngio
gram
pne
umog
ram
Muk
ada
et a
l. (2
1)19
8413
/MN
PX,
SS,
ST,
SV
isua
l di
stur
banc
es,
panh
ypop
itui
tari
smSk
ull x
-ray
, to
mog
ram
s, C
T,
angi
ogra
m,
pneu
mog
ram
Lew
in e
t al
(20
)19
8427
/FN
PXEp
ista
xis,
NPX
mas
sSk
ull x
-ray
, tom
ogra
m, a
ngio
gram
, CT
Mai
er (
23)
1985
77/M
NPX
, SS
, ST
Hea
dach
e, C
SF r
hino
rrhe
aSk
ull
x-ra
y, t
omog
ram
Mai
uri e
t al
. (2
2)19
8725
/FN
PX,
SS,
STH
eada
che,
nas
al o
bstr
ucti
onSk
ull x
-ray
, to
mog
ram
s, C
T
Hill
man
et
al.
(24)
1988
64/M
SS,
ST,
SV
isua
l dis
turb
ance
s, a
bduc
ent
nerv
e pa
lsy
(L)
CT, M
RI
Beni
tez
et a
l. (2
6)19
9829
/MN
PX,
SS,
ESN
asal
obs
truc
tion
, pr
essu
re in
the
nos
eSk
ull x
-ray
, CT
, M
RI,
angi
ogra
ms
and
behi
nd t
he e
yes
Ort
iz e
t al
. (2
5)19
9820
/MN
PXEp
ista
xis
CT
Phar
aboz
et
al.
(26)
1989
27/F
NPX
, SS
Nas
al o
bstr
ucti
onCT
, MRI
Aki
mur
a et
al.
(28)
1989
12/F
NPX
, SS
, ES
, ST
S,
MS
Vis
ual
dist
urba
nce
CT,
MRI
, an
giog
ram
Byrn
e &
Ses
sion
s (2
9)19
9029
/MN
PX,
SS,
ES,
MS
Nas
al o
bstr
uctio
n, N
PX n
a N
F m
ass
Skul
l x-r
ay, C
T, M
RI
Gili
& G
arci
a (1
7)19
919/
MN
PX,
SSN
asal
obs
truc
tion
, di
plop
ia w
ith
Skul
l x-r
ay, C
T, M
RI
abdu
cent
ner
ve p
alsy
(R)
Bret
& B
ezia
t (3
0)19
9316
/FN
PX,
SSN
asal
obs
truc
tion
CT, M
RI
Sene
r ((
34)
1994
8/M
NPX
, SS
, ES
, S
Hea
dach
e, d
ecre
ased
vis
ion,
nas
al o
bstr
ucti
onCT
, MRI
Kanu
ngo
et.
al (
32)
1995
40/F
NPX
, SS
, S
Hea
dach
e, n
asal
obs
truc
tion
Skul
l x-r
ay, C
T, M
RI
Che
ddad
i et
al.
(31)
1996
Prem
atur
e/F
NPX
Resp
irat
ory
insu
ffic
ienc
yN
asal
end
osco
py,
CT
Jiang
et
al.
(33)
1998
7/M
ESEp
ista
xis
CT
Cha
krab
arty
et
al.
(40)
1998
46/M
NPX
, SS
, ST
Nas
al o
bstr
ucti
on,
doub
le v
isio
nCT
, MRI
NPX
, nas
opha
rynx
; SS,
sph
enoi
d si
nus;
ST,
sel
la t
urci
ca; S
, sup
rase
llar;
ES,
eth
moi
d si
nuse
s; M
S, m
axill
ary
sinu
s; N
F, n
asal
fos
sa; C
SF, c
ereb
rosp
inal
flu
id; M
RI, m
agne
tic r
eson
ance
imag
ing;
CT,
com
pute
d to
mog
raph
y; [
L],
left
; [R
], r
ight
.
430 Arq Neuropsiquiatr 2001;59(2-B)
tomograms that showed sinus opacification, tumorexpansion, enlarged sella turcica, cystic lesions, fluidlevel, bony erosions, and calcifications6-10,13-22,27, 30.
Computed tomography in the cranial base cran-iopharyngioma cases reported usually reveals theheterogeneous nature of the tumor with its solidand cystic components, calcification, multicysts, lycticlesions, irregular enhancement. Multiple calcifica-tions are observed within the tumor, especially inyounger patients. In adults, craniopharyngiomas areoften not calcified17,19,21-23,26,27,29,30,32,35,40. Magneticresonance image clearly showed the tumor exten-sion, cystic portions, mixed intensity signal, inhomo-geneous or heterogeneous enhancement17,23,25-
30,32,34,40.
CONCLUSIONCraniopharyngiomas with infrasellar develop-
ment are very rare. Up to now, thirty-five cases hasbeen reported in literature since 1924. The mostcommon location of infrasellar craniopharyngiomahas been the sphenoid sinus either alone or com-bined with other sites. The infrasellar craniopharyn-gioma may then originate anywhere along the tractof the obliterated craniopharyngeal duct, whichwould include the sphenoid bone, vomer, and na-sopharynx.
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