ORIGINAL ARTICLE
Selective Embolization and Resection of a Large
Noninvoluting Congenital Hemangioma of the Lower Eyelid
Kenneth G.-J. Ooi, M.Surg.(Ophth.)*, Jason D. Wenderoth, F.R.A.N.Z.C.R.,
Ian C. Francis, F.A.S.O.P.R.S.*, and Geoffrey A. Wilcsek, F.R.A.N.Z.C.O.*
*Department of Ophthalmology, Prince of Wales Hospital, and Faculty of Medical Sciences, Inflammatory
Diseases Research Unit, University of New South Wales; Save Sight Institute, Sydney Eye Hospital, University of
Sydney; and Department of Radiology, Prince of Wales Hospital, University of New South Wales, Sydney, Australia
Purpose: To present a combined interventional radiologic and resec-
tion technique for the management of a large amblyogenic, highly
vascular lesion of the lower eyelid associated with severe mechanical
ectropion, conjunctival pyogenic granuloma, and recurrent, spontane-
ous conjunctival bleeding.
Methods: Preoperative selective embolization and coil placement were
followed by resection of the tumor.
Results: Embolization, coil placement, and resection were uneventful
with cosmetic and functional visual improvement obtained. Pathology
indicated a noninvoluting congenital hemangioma.
Conclusions: To the authors knowledge, this is the first surgical
technique reported in the ophthalmic literature for the selective embo-
lization of a noninvoluting congenital hemangioma prior to its resec-
tion. Such a technique should be considered in the management of large
lesions of this type.
(Ophthal Plast Reconstr Surg 2009;25:111114)
T he most common orbital tumors of childhood are heman-
giomas.1 They may be congenital hemangiomas (CH) or
infantile hemangiomas (IH). Lesions can be superficial or deep,
small or extensive, with eyelid or orbital involvement, or both.
IH are more common and may develop in 2.6% to 10% of
infants by the age of 1 year.2 There is a female predominance
but no mode of inheritance exists nor are there any causative
environmental factors.2 After appearing, IH undergo rapid
growth for 3 to 6 months, stabilize, usually involute spontane-
ously during the second year of life, and are completely
regressed in 76% of patients by age 7 years.3 Unlike IHs,
proliferation of CHs occurs in utero and they are mature at birth
with an equal sex distribution.4 They may then be either rapidly
involuting CH (RICH)5 or noninvoluting CH (NICH).6
Most hemangiomas involute with no major cosmetic
after-effects or functional ocular deficits. Amblyopia is the
most frequent and serious ocular complication of periorbital
hemangiomas.2 Other complications include strabismus, ptosis,
proptosis, exposure keratopathy, and optic atrophy.2 Large
hemangiomas are also prone to ulceration, which can lead to
sepsis.7 High-output cardiac failure may also occur.8 Any of
these sequelae are an indication for treatment. Surgery has
generally been avoided in the past as these lesions are not
encapsulated and can be difficult to remove en bloc. Piecemeal
Accepted for publication August 18, 2008.
Address correspondence and reprint requests to Geoffrey Wilcsek,
F.R.A.N.Z.C.O., Department of Ophthalmology, Prince of Wales Hospital, Uni-
versity of New South Wales, Sydney, Australia. E-mail: gwilcsek@ bigpond.com
DOI: 10.1097/IOP.0b013e318199dba5
Ophthal Plast Reconstr Surg, Vol. 25, No. 2, 2009
resection can result in pronounced bleeding and severe postop-
erative scarring.8 Successful selective embolization has been
reported9 but not as yet in the ophthalmic literature. We present
a combined interventional, radiologic, and resection technique
for the management of a large amblyogenic, highly vascular
lower eyelid NICH associated with severe mechanical ectro-
pion, conjunctival pyogenic granuloma, and recurrent, spon-
taneous conjunctival bleeding, where preoperative selective
embolization and coil placement was followed by tumor
resection.
METHODS
A 9-year-old Filipino girl presented with a right lower eyelid
tumor. MRI that accompanied her from the Philippines had demon-
strated large flow voids indicating high blood flow (Fig. A). An
angiogram was requested to plan a surgical approach, exclude an
arteriovenous malformation component, and assess the tumors suit-
ability for embolization prior to resection.
The tumor was present at birth and had increased propor-
tionately with general growth until the last 2 years, when the
increase was greater than and out of proportion to her general
growth. She had failed multiple intralesional steroid injections and
it was, therefore, decided that a trial of oral steroid was not
warranted. No pain or visual disturbance was present but she
reported daily bleeding from the tarsal conjunctival surface of her
right lower eyelid. These episodes would stop spontaneously. There
was no other significant medical or surgical history and she was not
on any medication.
On examination there was a large, 4-cm horizontal 3-cm
vertical mass within the right lower eyelid. The lesion was spongy,
nonpulsatile, and firmly adherent to the underlying eyelid. The skin was
mobile over the surface of the mass. There was an associated lobular
ulcerated mass involving the tarsal conjunctiva (Fig. B). Visual acuities
were 6/6 3 OD with no pin hole improvement and 6/5 OS. Pupils
were equal and reactive to light with no relative afferent pupillary
defect detected. Ishihara color plate testing was normal. Cover testing
for distance was normal but a small esophoria for near was present on
the right. There was a full range of ocular rotations and normal fundi.
No systemic abnormalities were detected.
Automated refraction and keratometry with a Retinomax K-plus
along with visual field testing with a Humphrey field analyzer were
conducted preoperatively as were complete blood examination includ-
ing coagulation studies and a group and hold. Preoperative CT, Doppler
ultrasonography, and cerebral angiography were also performed. In-
formed consent was obtained prior to each intervention.
Superselective angiography and embolization were performed
under isoflurane anesthesia with a 5 French Envoy guiding catheter
(Cordis, Miami, FL, U.S.A.) positioned in the right external carotid
artery and placed on continuous heparinized saline flush. Preemboli-
111
K. G.-J. Ooi et al. Ophthal Plast Reconstr Surg, Vol. 25, No. 2, 2009

A, Preoperative coronal view T1-weighted MRI postinjection of contrast (gadolinium) showing a large noninvoluting congenital heman-
gioma (N) with large flow voids present (solid white arrows) consistent with high blood flows. B, Preoperative photo showing large
lower eyelid swelling due to a noninvoluting congenital hemangioma (solid black arrow) and tarsal conjunctival ulcerated lobular mass due
to a pyogenic granuloma (solid white arrow). C, Preembolization lateral view angiogram demonstrating external carotid artery supply prin-
cipally via infraorbital (solid white arrows) and transverse facial (solid black arrows) arteries. This is a vascularized capillary malformation, and
not an arteriovenous malformation, as there is no arteriovenous shunting. D, Postembolization lateral view angiogram showing infraorbital
artery following embolization with 150- to 250-m polyvinyl alcohol particles. There is marked reduction in opacification of the capillary
bed and sluggish flow in the parent vessel. Embolization in this vessel was terminated early to avoid forcible reflux of particles

112 2009 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Ophthal Plast Reconstr Surg, Vol. 25, No. 2, 2009 Eyelid NICH Embolization and Resection

zation angiography of selected vessels was performed with the catheter
advanced as distally as possible to demonstrate any dangerous intraor-
bital or intracranial anastamoses (Fig. C). Slow, gentle embolization of
infraorbital, and transverse facial arteries was achieved with 150- to
250-m polyvinyl alcohol (PVA) particles (Surgica, El Dorado Hills,
CA, U.S.A.) through a Rapid Transit 0.021-inch ID microcatheter
(Cordis, Miami, FL, U.S.A.). A solitary detachable UltiPaq 10 stretch-
resistant platinum microcoil (Micrus, Sunnyvale, CA, U.S.A.) was also
placed at the origin of the infraorbital artery. Feeders from accessory
meningeal artery to ophthalmic artery and from facial artery to the
intraorbital arterial circulation via its alar branch precluded their
embolization. Control angiography postembolization was conducted
(Fig. D).
Four days later, dissection of the tumor was conducted under
general anesthesia supplemented with perilesional local anesthesia
using a mixture of 1:100,000 adrenaline and 0.25% ropivacaine hydro-
chloride. A subciliary approach was used with extensive diathermy and
suture ligation of multiple feeder vessels was required for hemostasis
(Fig. E). The tumor extended through orbital septum and the inferior
retractors and was tightly adherent to the conjunctiva. The plane was
also poorly defined between tumor and orbicularis muscle. Feeding
vessels were tied with 5-0 polyglactin sutures (Vicryl, Ethicon,
Somerville, NJ, U.S.A.) and the tumor was removed for histopa-
thology. A shave biopsy of the lower eyelid tarsal conjunctival
lesion was also taken. After tumor removal, a sulcal deformity
superior to the inferior orbital rim (Fig. F) was evident. Pedicles of
fat were developed from the lateral and central inferior orbital fat pads
and advanced in the defect. Their position was stabilized with 5-0
polyglactin and skin closure was with 7-0 polyglactin (Vicryl, Ethicon,
Somerville, NJ, U.S.A.). A Minivac drain (Promedica, Sydney, NSW,
Australia) was sutured in place with 4-0 (AU-1, Alcon Laboratories,
Fort Worth, TX, U.S.A.) silk. Two days postoperatively, the drain was
removed.
RESULTS
Retinomax K-plus measurements revealed preoperative refrac-
tions and average keratometry readings of 0.25/0.75 84 and
42.25 D OD and 0.50/0.50 28 and 42.25 D OS. Humphrey visual
analysis showed a right inferior visual field defect but numerous
fixation losses were present. The findings from complete blood exam-
ination and coagulation studies were normal.
CT revealed an ovoid mass lesion that appeared to arise in
the soft tissues of the lower right eyelid. This measured 2.9 cm
craniocaudally, 2.9 cm anteroposteriorly, and 3.8 cm transversely.
There was moderate heterogeneous enhancement with contrast.
Superiorly, the mass extended to the level of the inferior border of
the lens and inferiorly to below the orbital rim. Medially and
laterally, the mass appeared to extend to the canthi of the orbit.
Prominent vessels were seen extending from the inferior margin of
the lesion in the subcutaneous fatty layer of the cheek. The appear-
ance was consistent with a hemangioma. The retrobulbar soft tissues
were normally defined. There was no associated bony destruction or
remodelling. The right globe was of normal shape and appearance.
No abnormalities were identified within the brain.
Doppler ultrasonography revealed a large soft-tissue mass
that appeared to be extraorbital. It was heterogeneous in its appear-
ance but predominantly echogenic with large hypoechoic spaces
within it. On color Doppler examination, these hypoechoic spaces
demonstrated arterial and venous flow. No large feeding arteries
were identified but there were high flow arterial vessels within the
mass. At the periphery, there appeared to be draining veins most
prominent at the superior and inferior aspects. Appearance was in
keeping with a hemangioma.
Cerebral angiography showed a moderately dilated right
ophthalmic artery supplying the tumor via feeders that appeared to
arise from anterior ethmoidal and possibly angular arteries. The
component of supply from the ophthalmic artery entered the lesion
superiorly and posteriorly, with vessels virtually encircling the
posterosuperior component of the mass. There was also extensive
external carotid artery supply to the lesion. This arose principally
from the infraorbital, accessory meningeal, and transverse facial
arteries, with a small component arising from the facial artery itself.
There was no arteriovenous shunting; features were in keeping with
a vascularized hemangioma amenable to embolization (Fig. C).
Embolization and coil placement occurred without incident.
Rapid devascularisation of infraorbital (Fig. D) and transverse facial
artery supplies was confirmed on postembolization angiography
with significant reduction in external carotid supply to the lesion.
Facial artery feeders to the inferior aspect of the tumor were not
amenable to embolization as they also joined the intraorbital arterial
circulation. Profound accessory meningeal supply also remained
because of risks of retrograde particle injection in internal carotid
and ophthalmic arteries.
Resection was uneventful with 100 ml blood loss recorded.
The patient had improved cosmetic and functional results (Fig. G)
but developed postoperative pyrexia from an infected peripherally
inserted central catheter line. This resolved with peripherally in-
serted central catheter removal and intravenous ceftriaxone 1 g daily
for 3 days. She was discharged from the hospital on day 5 postop-
eratively on chloramphenicol ointment 3 times per day to the wound
and oral cephalexin 250 mg 4 times per day for 1 week. After her
final 4-week postoperative visit, she returned to the Philippines with
only minor lower eyelid swelling. Her caregiver was given instruc-
tion as to provision of regular photographs to monitor her progress
but unfortunately she has been lost to follow-up to her local
healthcare providers.
Histopathology showed a vascular lesion with both a lobular
and diffuse pattern involving fibrous tissue, fat, and skeletal muscle.
Most vessels were capillary type; however, the lobular areas contained
a relatively large but thin-walled central vessel. The vessels were lined
with flattened endothelium with no obvious hobnailing. Larger vessels,
particularly at the periphery, showed intraluminal refractile material
with giant cell foreign body response, consistent with foreign throm-
bosing agents (Fig. H). The lesion was poorly circumscribed and

FIG. (Continued) through collaterals in the ophthalmic artery. Note the microcatheter tip located distally in the infraorbital artery
(dashed black arrow). A thrombogenic fibered platinum coil was deposited proximally in the infraorbital artery to prevent preoperative
recanalization. E, Intraoperative photo showing subciliary approach (solid black arrow) to exposure of the noninvoluting congenital
hemangioma (N). Extensive feeder vessel ligation and diathermy were used to achieve hemostasis (solid white arrows). F, Intraopera-
tive photo showing dissected-out large volume non-involuting congenital hemangioma (N), measuring 2.5 cm in length, leaving a
sulcal deformity superior to the inferior orbital rim (solid black arrow). Pedicles of fat were developed from the lateral and central in-
ferior orbital fat pads, advanced in the defect, and stabilized with 5 0 polyglactin. G, Postoperative photo showing some residual
postoperative edema at 4 weeks. H, Hematoxylin and eosin stain of noninvoluting congenital hemangioma surgical specimen. A pe-
ripheral section is shown demonstrating polyvinyl alcohol particles within large vessels seen as a pale, lacy material that has partially dis-
solved out during processing (solid black arrows). A surrounding foreign body giant cell reaction is seen (solid white arrows). Fibrin
(dashed black arrow) and red blood cells (R) can also be visualized within the vessels.

2009 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. 113
K. G.-J. Ooi et al.
present at several resection margins. Glucose transporter isoform 1
(GLUT1) staining was negative. There was no evidence of malignancy.
Final diagnosis was NICH. The conjunctival surface showed lobular
hemangioma with surface ulceration and subsequent acute inflamma-
tion, consistent with a pyogenic granuloma resulting from conjunctival
exposure.
Postoperative visual acuity was 6/6 with plano refraction. Cover
testing and ocular rotations remained unchanged from her preoperative
assessment. Lang stereo testing revealed no abnormalities.
DISCUSSION
NICH are much less common than RICH lesions but
both exist as bossed plaques or tumors.7 NICH, RICH, and
IH have overlapping pathologic and clinical features sup-
porting the hypothesis that these vascular tumors may be
variations of a single entity ab initio.4 CHs, however, stain
negatively for the GLUT1 enzyme, which is uniquely ex-
pressed on endothelial cells of IHs.7 RICH lesions involute
much more rapidly compared with IHs5 whereas NICH
lesions grow proportionally with the childs growth.6 De-
spite being clinically evaluated as nonpulsatile, its presence
at birth and growth with age, along with large flow voids on
MRI and high flow arterial vessels as demonstrated on
Doppler ultrasonography and GLUT1 negative staining, is
consistent with this tumor being diagnosed as a NICH.
RICH management is straightforward in patients with
typical presentations where involution may begin within the
first week of life.7 Resection as a primary treatment option has
been presented as definitive treatment for ocular adnexal cap-
illary hemangioma10 and is also proposed as the recommended
treatment for NICH, with preoperative embolization to control
bleeding in larger lesions.7 Rootman11 describes a high flow
eyelid capillary hemangioma with both internal and external ca-
rotid supply necessitating excision under hypotensive anesthesia.
We believe that preoperative embolization to help control intra-
operative bleeding is preferable to hypotensive anesthesia.
Selective embolization with PVA or coils requires suit-
able vascular anatomy.12 There is a risk of infarction of normal
ocular tissue as normal arteries might be occluded along with
target arteries. Feeders from the accessory meningeal and facial
arteries were a potential embolization risk to orbital structures
in our case but were identified with preembolization angiogra-
phy. Intraoperative hemostasis was aided by embolization,
which facilitated uncomplicated NICH excision with satisfac-
tory cosmetic outcome. Balancing the facilitation of resection
114 2009 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Ophthal Plast Reconstr Surg, Vol. 25, No. 2, 2009
with the risks associated with angiography and embolization
requires case-by-case assessment and the availability of an
experienced interventional radiologist.
Our case is novel in its description of a combination
technique of selective embolization followed by resection,
not previously documented for an eyelid NICH. Such a
technique should be considered in the management of large
NICH lesions.
ACKNOWLEDGMENTS
The authors acknowledge Dr. Dianne Reeves from the
Department of Anatomical Pathology, SEALS, Prince of Wales
Hospital, for her contribution to the histopathology figure.
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