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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