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657

Therapeutic Embolization Juvenile Angiofibroma

Glenn

H. Roberso&’ Ann C. Price3

James

2

M. Davis’

Amar

Gulati’

of

Therapeutic embolization of juvenile angiofibromas was performed in 1 5 boys, aged 1 2-1 8 years, 1 1 of whom subsequently underwent surgery. lntraoperative blood loss was reduced from an average of 2,400 ml in nonembolized patients to 800 ml after embolization. Angiography is of value to confirm the diagnosis prior to excision and to delineate the extent of the tumor. Embolization may be performed at the same sitting as a presurgical adjunct or possibly as a definitive or palliative therapeutic method. The embolization procedure is discussed in detail, emphasizing techniques and potential hazards of such procedures.

Juvenile that arises

nasopharyngeal angiofibroma is a benign, from the nasopharynx almost exclusively

though histologically benign, to recur if not completely epistaxis

and

massive

nasal

obstruction.

hemorrhage.

cryotherapy,

the tumor is locally removed. The most Biopsy

Reported

is

modes

electrocoagulation,

of

hormonal

highly vascular in adolescent

invasive common

hamartoma males. Al-

and has a predilection initial symptoms are

hazardous

due

to

therapy

include

therapy,

embolization,

the

danger

surgery,

of

radiation, and

injection

of sclerosing agents, as well as observation in the hope of spontaneous regression [1 1. Surgical removal is currently the most widely accepted mode of therapy, but this may be accompanied by significant hemorrhage, often greater than 2,000

ml.

Angiography

before

treatment

is indicated

the amount of vascularity, and margins of the tumor, assessment since Received sionJune6,

June 20, 1979.

1 978;

accepted

after

revi-

, Department of Radiology, Massachusetts General Hospital, Boston, MA 02114. 2 Present address: Department of Radiology,

Albany

Medical

1 2208. Address son. 3 Department Center Hospital,

Center reprint

Hospital, requests

Albany,

D.C. 20052.

Roentgen

October 334-0657 Ray

presents

the

extent

of the

lesion,

additional

[1 ]. The

hazards

angiographic

features

and a preoperative diagnosis is usually possible prior to biopsy preoperative embolization of the tumor aids in diminishing blood thereby allowing for more complete excision [3, 4]. We describe

angiographic

findings

in 1 5 patients

and

discuss

the

results

of preoperative

embolization.

NY

1979 $00.00 Society

and Methods

Medical

Present address: Department of Radiology, George Washington University Medical Center,

© American

the

Materials of Radiology, Albany Albany, NY 12208.

AJR 133:657-663, 0361 -803X/79/1

then

to define

nature of the feeding vessels. In defining the of intracranial extent is of particular importance

to G. H. Rober-

4

Washington,

surgery

are characteristic, [2]. In addition, loss at surgery,

the

Radiographic sinuses,

and

preferably and

to define Radiographic

displacement

evaluation often

shows

begins a sizable

pluridirectional, extension findings of the

is usually into

the

plain

necessary

pterygomaxillary

include

posterior

with

nasopharyngeal

expansion wall

of the

films

of the mass

to delineate space

and

of the pterygoid maxillary

nasopharynx

in the

antrum

lateral

the lesion the and

posterior

paranasal

Tomography,

in the frontal

paranasal

maxillary

and view.

sinuses

view (fig.

1).

fissure with anterior displacement

of

ROBERSON

658

ET

AU.

AJR:133,

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Fig.

1 -Case

October

1979

6. Pluridirectional

to-

mography in frontal (A) and sagittal (B) views. Huge right-midline nasopharyngeal mass (arrows), with extension into sphenoid sinus. Right selective external maxillary angiography before (C) and after (D) embolization show vascularity and

the

pterygoid

and

mental

features

increase

rapid

through

nous

channels.

the

the lesion (figs.

2 and

The

patient

should

with

vascularity

have

order bolization

30

anesthesia has

been

mm

were

is

of larger

ye-

homogeneous of

the

retention

and

lesion

within

ing

to techniques the

first

second femoral

necessary

in our

by mouth

the day

suffices, for

to have series.

commencement

embolization

as

but

patients

on with

an intravenous required. The

first

Two method

occasion, emotional line methods

general stress in place

in

of em-

uses large (9 or

by Hilal which

procedures).

catheter

(BD

branch foam The in diameter.

long

and

compressed

and

index

finger,

filled

syringe

catheter.

As

foam and the

6].

This

gelatin used

method

the

was

used

[3].

The

pledgets via transother 1 1 cases (12

Seldinger

is selectively

accord-

described

foam in the

with

Cal.)

technique,

positioned

a

in the

5

exter-

to be embolized.

mm

gelatin

combined

Barbara,

previously

Using

(Gelfoam,

2-3

single

results.

approach, Santa

[5,

were

RPXO65)

available

procedures.

proce-

cases,

direct

Corp.,

embolization technique, catheter approach, was

mercially

is given

of the

via

originated

four

nal carotid

of angiography

catheters

(Heyer-Schulte

on

French

distin-

paranasal

1 00 mg of secobarbital

medication used

seg-

circulation

appearance

fluid

hypertro-

beading, The

relatively

before

usually

It is advisable

to administer

marked

without

periphery

nothing

For premedication,

or instability.

typically

Teflon

spheres

Gelatin

about

anesthesia

1 0 French)

sinus

Silastic

early

concomitant

intramuscularly Local

sphenoid

3).

or embolization. dure.

The

formation.

is

hypervascular from

sinuses

one.

of arteries

or aneurysm

hamartoma The

with

guishing

number

dilation the

except

1 5 cases.

are consistent,

in the

narrowing,

prominent,

cases

in seven of the

Angiographic phy

in all

plates

was involved

postembolization

in pad

Upjohn sterile

is cut The

Co.,

pads into

long

strips strips

by rolling obtaining pledget the embolus

embolus passes

Mich.)

each

used

several are

a final is then

Kalamazoo,

commonly into

dimension is

then

through

surgical

centimeters

cut

fragment

placed

is com-

for

long

and

3-5

mm

segments between of

1 x

the 3-4

thumb mm.

A

in the tip of a 5 ml salinegently the

flushed catheter,

into resistance

the

AJR:133,

Fig. ryngeal

October

2.-Case injections

7. Left ascending before (A) and

(B) embolization. illary sinus

Posterior

displaced

Distal external

Forming

the

proper

desirable

to

have

cm

exchange

659

beshow

curve

at

the

of the

positioning minimal

exits through the catheter tip, to avoid a rapid injection when Injection at a rate exceeding in reflux into proximal arterial could occur; this, of course, is

catheter

catheter

tip

catheter. curve

since

is

important

In general, acute

to

it is

catheter

curves are difficult to manipulate in small caliber vessels, such as the external carotid artery and its branches. The taper of the catheter tip should be minimal in order to permit passage of emboli. It may be necessary to use a catheter with an acute curve to enter either the common carotid or the external carotid trunk and then to exchange the original catheter with a simple curve catheter using a 250

ANGIOFIBROMA

(arrows).

angiograms

(D) embolization

selective

JUVENILE

phaafter

is appreciable and, as the embolus extreme caution must be exercised the resistance is abruptly released. the flow of the artery would result trunks and intracranial embolization imperative to avoid. facilitate

OF

wall of max-

anteriorly

carotid

fore (C) and after desired result.

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EMBOLIZATION

1979

wire.

Maintenance of flow around passage of the emboli; therefore

the catheter assures peripheral use of large catheters and wedging

is

undesirable.

impedes

Likewise,

peripheral

Spasm

be treated

may

Xylocaine The

catheter in

tip

the into

the

catheter

be

avoided.

should

intraarterial

as close carotid temporal

position

superficial

in the

infusion

to the artery artery

is essential

temporal

procedure due

of diluted

lesion

at and

as possible,

the level of the internal maxillary

to prevent

reflux

into

to

with

the

increased

and

rest

middle

of the

caliber

and

meningeal

emboli flow

arteries

entering of

the

the

internal

trunk.

Embolization in each

case,

appears

occluded.

reduction

is advanced

catheter

angiofibroma maxillary

around and

carotid artery and avoid the danger of a stroke. When is in the proper position, the injected emboli lodge in

proximal

early

spasm emboli

by direct

distal external the superficial

Proper

the internal the catheter the

of

or diazepam.

usually bifurcation artery.

arterial

passage

in the

ofthe

internal

continuing As the rate

maxillary until

and

vascular volume

the

component internal

bed of

maxillary

is progressively flow

requires

is the initial component obliterated, a concomitant

step

ROBERSON

660

ET

AU.

AJR:133,

Fig. 3.-Case

October

8. Lateral

nal carotid angiograms. highly vascular hamartoma

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eration of most after embolization rent,

having

with

placement

1979

distal

exter-

Wide extent of (A) and oblit-

of abnormal vascular bed (B). Lesion was recurbeen

previously

resected

seeds

of radon

(arrows).

..1

.J

,b

,

J1(

H_:f##

\

.

.

.‘-‘-“‘#{149}--Tvr:;;T1’ “ .

-

..---.

--

.-.

B

A reduction

of

the

rate

at

which

emboli

and

saline

are

injected

to

through

his

prevent reflux into the intracranial circulation. Arterial pressure progressively forces emboli into the periphery of the tumor so that in time, parts of the vascular bed, initially occluded, become reopacified. A delay of 1 5-30 mm permits peripheral packing of emboli and facilitates more thorough emboli-

operated

zation.

pharynx.

During

ascending

this

cally

evaluated

and

angiography

provide

The

has

For unilateral and

packed

lesions,

ascending

as

may of

gelatin

foam

peripherally completes

the

angiography pharyngeal

the

success

of

initially

lesion.

maxillary

case.

juncture

at which

time

Once

vascular

in the procedure

artery

For those

of the contralateral interarteries was performed

further

embolization bed

the supplying vessel, Usually after 20-40 reduced by 90%-95%

is determining is unnecessary

is obliterated

and

emboli

the end point, or

inadvisable.

accumulate

The

required

3 weeks

mucosa

within

additional embolization is not efficacious. emboli have been introduced, perfusion is and the procedure is terminated.

maxillary

daily

for

2 weeks

of temporary

combined

transantral

air

Case Material

current

angiofibroma

Juvenile angiofibroma was studied in 1 5 patients over a 7 year period; four of these were previously reported [3]. All the patients were boys, aged 1 2-1 8 years. Nasopharyngeal

internal

duration

varied

pharyngeal

from

1 month

to 4 years.

M. E., an 1 8-year old boy, was admitted and Ear Infirmary with a 6 month history

to Massachusetts Eye of difficulty breathing

exclusively

located

a juvenile

from

angiofi-

from

the

the

posterior

returned

for

removal

of

ligation

of the

right

external

transpalatal

excision

was

positioned

gelatin tion

right

(figs.

of flow

distal there

the

right naso-

tumor,

which

carotid

and

with

a

cryosurgery.

right

to the

tumor.

meningeal The (fig.

distal 4D).

right

(fig. After

ascending

were

introduced

The

carotid,

from

catheter

4C).

the

right

by branches

pharyngeal was

re-

ascending

Additional

the artery,

subsequent then

of the

supply

arteriography, with

external

demonstrated

4B) and also

and artery.

in the

internal

arteriography primarily

on the

fragments

external carotid was subsequent middle

4A

artery ophthalmic

foam

bilateral

pharyngeal supplied

artery

the

occluded

Case 5

almost

Transfemoral

ascending

maxillary

fragments.

Case Report

tumor

with

It extended

and

cells.

from

and

Representative

external

nasopharyngeal

Estimated blood loss was 2,500 ml. He was admitted nearly 1 #{189} years later with a 6 month history of recurrent epistaxis. Polytomography of the sinuses demonstrated a lobulated right posterior nasopharyngeal mass with extension into the sphenoid sinus, right maxillary antrum, and possibly the right and

breathing, and epistaxis for admission. Symptom

thickening of in the nasocarotid arterio-

a mass

consistent

supplied

and

consisted

carotid,

difficulty reasons

been

prior to bleeding

pharynx as far forward as the posterior wall of the right maxillary sinus, which was displaced forward superiorly at least as high as the floor of the sphenoid sinus and inferiorly as far as the posterior part of the hard palate. The patient was subsequently discharged on stilbestrol 1 5 mg

ethmoid

mass, nasal obstruction, were the most common

and

bilateral

vascular

branches.

polypoid

antrum

of midline, was

He had

hospitalization.

transfemoral

mass

epistaxis.

demonstrated

maxillary

a large

to the right The

films

right

A selective

internal

and

of additional

sinus

in the

broma.

headaches,

at an outside hospital 6 months was accompanied by massive

‘

surgery

demonstrated

mainly

external

was

the

admission.

that

gram

fluoroscopy

that

into

of the internal

artery

ipsilateral

to the distal

this

the

be angiographi-

Sequential

returned

as the more

the

bifrontal

‘polyps’

On admission,

by embolization.

A critical the

(such tumor

monitoring

is then

embolization

pharyngeal

lesions crossing the midline, nal maxillary and ascending

followed

the

if indicated.

embolization

become

vessels

supply accurate

catheter

for further

injected

other

that

and embolized

embolization. carotid

period

pharyngeal)

nose,

on for

was

catheter and

11

oblitera-

positioned

in the

artery on the right just at its bifurcation and partial occlusion of the superficial temporal arteries after introduction of 1 7 gelatin foam stem After

of the

internal

embolization,

maxillary the

patient

artery underwent

was

also sur-

gery for resection of a recurrent tumor via a medial maxillectomy approach (1 day after angiography). Estimated blood loss was 250 ml. He was admitted for a fifth time nearly 3 years after initial

AJR:133,

Fig.

lective

October

EMBOLIZATION

1979

4.-Case

5. A and

ascending

pharyngeal

B, Right

OF

JUVENILE

661

ANGIOFIBROMA

Se-

injections,

lateral view. Early arterial phase (A), arterial supply. (Ant = anterior). Early yenous filling and dense blush (B). C and

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D, Right

grams.

selective

external

C, Arterial

carotid

angio-

supply from internal

maxillary artery branches. D, Postembolization occlusion of internal maxillary stem (arrow).

I

A

B

hospitalization because of bitemporal headaches and pain behind the eyes and in the back of the head. Transfemoral bilateral external and right internal carotid arteriography demonstrated a small amount of residual juvenile angiofibroma on the right side within the pterygomaxillary fissure. There was no evidence of intracranial extension. Repeat selective right external carotid, right internal carotid,

and

demonstrated of the lower

left

common

carotid

arteriography

1 4 months

later

enlargement of the residual tumor with involvement half of the sphenoid sinus and lateral extension into the

region of the pterygomaxillary fissure. The main blood supply was from the internal maxillary artery on the right. No intracranial extension was demonstrated. The ascending pharyngeal artery was not selectively

catheterized.

radiation

cases. The

arterial

case which

supply

artery,

maxillary

and

in this series, was the major

Embolization

supplied

surgery.

of 1 6 embolic

procedures

in table 1 . In case

1 4, the

on 1 5 patients lesion

recurred

are

consisted the

after

of the

angiofibroma

em-

The rest or further in several internal in every

and the ascending pharyngeal artery, feeding vessel in five of the 1 5 patients.

was

performed

in the

embolization

procedures,

internal

A second

were performed. reembolization was subtotal

to the tumors

which

angiography

eral

summarized

embolization,

last

nine

at the embolic

same

maxillary

1 1 consisted

and

ascending

time

procedures. of unilateral

maxillary embolization, four of unilateral plus ascending pharyngeal embolization,

Results The results

therapy,

bolization and surgical resection of the patients have not required surgery, although the resection

as

initial

Of the 16 internal

internal maxillary and one of bilat-

pharyngeal

emboliza-

tion.

Operative

blood

loss

before

embolization

was

significant,

662

ROBERSON

TABLE

1 : Embolization

Procedures

Tumor

AJR:133,

October

Supply

1979

Estimated

vessel

No.

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AL.

and Results

Method/case

Silastic

ET

Internal Maxillary

Ascending Pharyngeal

Internal carotid

Artery

Artery

Artery

BlOOd Loss at Surgery After

Embolized (no. emboli)

Embolization

[3]: Bilateral

.

.

Left

.

Left internal maxillary

Left

2.

.

.

.

.

.

Left internal

.

maxillary 3.

......

4.

Bilateral

.

.

.

.

.

.

Right

.

.

.

.

.

.

750 ml (36) 500 ml (32)

Right internal maxillary (40) Right internal maxillary

500 ml No surgery

(50)

Gelfoam: 5:

First

Right

admission

.

.

2,500

No embolization

.

ml (no emboli-

zation)

Second

admission:

First attempt

Right

Right

Right

Right ascending pharyngeal (1 1); right

Second Third

attempt attempt

.

6.

.

.

Right Bilateral

.

.

.

.

.

.

.

.

.

.

.

.

.

Bilateral

Left

Left,

right

ml

internal

maxillary (6) No embolization No embolization Left internal maxillary (14);

Left

right

7.

250

anterior

pharyngeal left anterior pharyngeal Left anterior

Left

No surgery

(10); (5)

ml

1,700

pharyngeal (20); left internal maxillary (55) 8: First

admission

Second

(no

2,250 ml (no embolization)

arteriogram)

Left

Left

admission

Left internal maxillary

9.

.

.

.

.

Right

.

.

.

Right internal maxillary (18); right anterior pharyngeal

10.

Right

...

Right

maxillary

.

12.

13.

.

Right,

.

.

Bilateral

Left

500

Left

Left

400-450

ml

1,200

ml

(21)

Left internal maxillary (20); left anterior pharyngeal

ml

(30)

Right internal maxillary

.

.

Left

500 ml

(50)

Right internal

Right

1 ,1 00 ml (postembolization)

(10)

internal

maxillary 11.

(18)

No surgery

(7)

14:

First

attempt

Second

attempt

15

Note-Words . From Albany

.

in

.

boldface

Medical

.

type

center

.

indicate

.

.

.

Right

.

.

.

Left

.

Hospital.

Right

major

blood

supply.

Left

Left

Right internal maxillary (10)

500 ml

Right internal maxillary (40)

No surgery

Left internal maxillary

1,600 (32)

ml

AJR:133,

October

EMBOLIZATION

1979

OF

JUVENILE

ANGIOFIBROMA

Angiography

averaging nearly 2,400 ml. After embolization, the average blood loss at surgery was about 800 ml. In seven cases,

fibroma,

500

to suggest

ml or less

was

lost

at surgery.

although

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benefit

The concept of therapeutic embolization of lesions of the head and neck can be traced to as early as 1 930 when Brooks [7] reported embolization of a carotid-cavernous fistula. The first embolization of an intracerebral lesion, an arteriovenous

malformation,

was

reported

in 1 960

by Leus-

senhop and Spence [8]. This stimulated the exploration of embolization in the management of intracranial lesions deemed surgically untreatable. However, extraaxial vascular lesions of the head, neck, and spinal cord constitute the majority of therapeutic interventional procedures. Juvenile angiofibroma presents an ideal situation for embolization, but reports of embolization of this lesion are scant [3-5, 9-

and

provide

assistance

in planning

surgical

culation

[9].

In no case

of intracerebral

in this

series

was

there

any

in all

our

3.

Roberson

the

profuse

arterial

supply

to

the

scalp

GH,

internal

in these

extent

pluridi-

of

1 5 cases

the

to be

lesion. of major

adjunct.

Biller

maxillary

H, Sessions

artery

DG,

embolization

Ogura

JH:

in juvenile

Presurgical

angiofibroma.

Laryngoscope 82:1524-1532, 1972 Pletcher JD, Newton TH, Dedo HH, Norman D: Preoperative embolization of juvenile angiofibromas of the nasopharynx.

4.

5.

Ann

Oto

Rhino

Hilal

5K,

Michelson

6.

Hilal and

J: Therapeutic

vascular

Mount

1975 percutaneous

of the

J: Therapeutic

of the external

experimental

head,

embolization neck

and

J

spine.

1975

U, Correll

malformations

posium

lesions

43:275-287,

5K,

cular

84:740-746,

Laryngol

extra-axial

Neurosurg

results.

Paper

Neuroradiologicum,

embolization

carotid presented

at the

G#{228}teburg,

of vas-

circulation: Sweden,

clinical

Ninth

Sym-

September

1970 7.

Brooks

8.

B:

9.

The

of traumatic

treatment

South MedJ

23:100-106,

Luessenhop

AJ,

cerebral

arteries.

Djindjian

10.

Spence

WT:

JAMA

Artificial

1 72: 1 1 53-1

R, Cophignon

Lallemant

J, TherOn

Y, Gehanno

.

Biller HF, Sessions

fistula.

embolization 1 55,

of

J, Merland

JJ,

Houdart

JJ, Levesque Otolaryngol

R:

from the femoral

M, Nahum M:

lnt#{233}r#{232}t de l’angiographie Ann

the

1960

arteriography 1973

P. Merland

Fibrome naso-pharyngien. lective et de lembolisation. 92:127-136, 1975 1 1

arterio-venous

1930

Embolization by superselective route. Neuroradiology 6:20-26,

ischemia.

cases

preferably

the

PH, Thompson R, Calcaterra T, Kadin MR: Juvenile angiofibroma: a more rational therapeutic approach based upon clinical and experimental evidence. Laryngoscope 84: 2181-2194, 1974 2. Wilson GH, Hanafee WN: Angiographic findings in 1 6 patients with juvenile nasal angiofibroma. Radiology 92:279-284, 1969

evidence

prevented any necrosis [1 6]. Pain in the scalp, noted in about one-third of the patients, was attributed to transient ischemia. The pain may be sufficient to require narcotic analgesia, but always for only a brief period, with remission after 1 -3 days. Low-grade fever was noted within 48 hr after embolization in three patients, but blood cultures were negative and temperature elevation was ascribed to tissue ischemia in each case.

shown

as a presurgical

for

Developmental variations of the branches of the external carotid arterial tree, notably ophthalmic artery origin from the middle meningeal artery and communications between the posterior division of the ascending pharyngeal artery with the vertebral artery, must be excluded before embolization. The occipital artery may communicate directly with the vertebral artery at the level of the posterior arch of the first cervical vertebra, the so-called proatlantal artery. Although the superficial temporal artery was embolized,

was

define

1 . Ward

therapy.

The primary indication for preoperative embolization is to reduce intraoperative blood loss. In two of our patients, blood loss before embolization averaged nearly 2,400 ml. The overall average intraoperative blood loss after embolization in our series was about 800 ml. Similar conclusions were drawn by Pletcher et al. [4] in Gelfoam embolization of seven cases. No permanent complications of therapeutic embolizations occurred in our series. The dreaded complication that must be prevented is escape of emboli into the intracranial cir-

Tomography, to

References

13].

Several excellent articles discuss the pathologic, clinical, and radiographic (including angiographic) aspects of juvenile angiofibromas[2, 1 2, 1 4, 1 5]. The angiographic findings are sufficiently characteristic to provide a tentative diagnosis

diagnosis.

is important

Embolization Discussion

establishes the diagnosis of juvenile angiothe clinical features are usually sufficient

the

rectional,

663

super

Chir

DG, Ogura JH: Angiofibroma:

so-

Cervicofac

a treatment

approach. Laryngoscope 84:695-706, 1974 1 2. Sessions RB, Wills P1, Alford BR, Harrell JE, Evans RA: Juvenile nasopharyngeal angiofibroma: Radiographic aspects. Laryngoscope 86: 2-1 8, 1976 1 3. Boles R, Dedo H: Nasopharyngeal angiofibroma. Laryngoscope 86:364-372, 1976 1 4. Sternberg 55: Pathology of juvenile nasopharyngeal angiofibroma: A lesion of adolescent males. Cancer 7 : 1 5-28, 1954 1 5.

Hicks

JU,

Nelson

Oral Surg 35:807-81 1 6.

Roberson

management Interventional

JT, Greene ders, 1979

GH,

Gulati

JF:

Juvenile

nasopharyngeal

angiofibroma.

7, 1973 AN:

CNS,

head

and

neck

vascular

lesions:

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