CLINICAL CHARACTERISTICS PATIENTS WITH CAPILLARY

HEMANGIOMA AND CAVERNOUS HEMANGIOMA IN KARIADI

HOSPITAL SEMARANG, INDONESIA

Sekar Kumalasari , Trilaksana Nugroho

1 2

1. Student of Ophthalmology Residency Program, Faculty of Medicine Universitas

Diponegoro/Dr. Kariadi General Hospital Semarang
2. Staff of Reconstruction, Oculoplasty, and Oncology, Opthalmology Departement,
Faculty of Medicine Universitas Diponegoro/Dr. Kariadi General Hospital Semarang

ABSTRACT

Introduction: Vascular tumors and malformations of the orbit comprise an

important group of orbital space occupying lesions. Although capillary
hemangioma and cavernous hemangioma are common vascular tumors and
malformations, only a few studies on clinical characteristics were available, which
resulted in a lack of basic data necessary for the proper care and effective
management of treatment to prevent complications.

Objectives: To characterize clinical features and complications of patients with

capillary hemangiomas and cavernous hemangiomas in Kariadi Hospital Semarang.

Methods: Medical records of all patients diagnosed as having capillary

hemangioma and cavernous hemangioma from March 1, 2018 through March 31,
2020 attended in polyclinic ophthalmology of Kariadi Hospital Semarang who met
inclusion criteria were retrospectively reviewed.

Result: Twenty one patients (16 diagnosed as capillary hemangiomas and 5

diagnosed as cavernous hemangioma) were included. Ratio female:male were 3:1
in capillary hemangioma with average age onset was at 3.3 months old. Eyelid
laterality were equal, 75% patients had capillary hemangiomas on upper eyelid.
Complications were refractive errors (43.75%), secondary glaucoma (31.25%) and
amblyopia (18.75%). Ratio female:male were 3:2 in cavernous hemangioma with
average age onset was at 36 years old. Complications were refractive errors
(hyperopia) (80%), secondary glaucoma (60%), limited ocular movement (40%),
and proptosis (100%).

Conclusion: In this study, capillary hemangiomas were most seen in the unilateral
upper eyelid of female patients. Complications include refractive errors, secondary
glaucoma and amblyopia. Complications for cavernous hemangiomas include
refractive errors (hyperopia), secondary glaucoma, limited ocular movement and
proptosis.
INTRODUCTION the slow-developing mass that created
Vascular tumors and many asymptomatic cases. 8

malformations of the orbit comprise Although capillary

an important group of orbital space
occupying lesions, accounted for 6.2
to 12.0% of all histopathologically
documented lesions. 1
Capillary hemangioma is the
most common vascular orbital tumor
in children, accounted for 0.7 to
2.2%. Capillary hemangioma of
1,2,3
eyelid or orbit is usually apparent at
birth or within first 8 weeks of life. 1
Mark may evident at birth in 50%
neonates, this nascent phase is
followed by rapid growth over next
few weeks, up to 6-9 month.
Subsequent to this proliferative phase,
hemangioma stabilize and slowly
regresses over a period of 5-12 years.
Complete involution occurs in 75-
90% cases by age 7-9 years. 4
Ocular complications due to
periocular lesions of capillary
hemangioma include ptosis,
anisometropia, amblyopia,
strabismus, proptosis, secondary
glaucoma, and optic nerve
compression. Main concern for
5
treatment is prevention of amblyopia
and life-threatening systemic
complications. 3
Cavernous hemangioma is the
most common benign orbital tumor in
adult, account for 3.1 to 9.0%. 1
Cavernous Hemangioma is benign,
noninfiltrative, slowly and
progressive. Presents most commonly
in early to middle-aged adults from 20
to 60 years.6
Tumor is
characteristically unilateral and
solitary. Most commonly found
within muscle cone, resulting slowly
progressive proptosis of the globe. 7
Orbit and globe will be able to
accommodate volume expansion of
hemangioma and cavernous
hemangioma are common vascular
tumors and malformations, only a few
studies on clinical characteristics
were available, which resulted in a
lack of basic data necessary for the
proper care and effective
management of treatment to prevent
complications.
The aim of this study was to
characterize clinical features and
complications of patients with
hemangiomas in Kariadi Hospital
Semarang.
MATERIAL AND METHODS
Medical records of all patients
diagnosed as having capillary
hemangioma and cavernous
hemangioma from March 1, 2018
through March 31, 2020 attended in
polyclinic ophthalmology of Kariadi
Hospital Semarang.
Medical records of patients who
met inclusion criteria were
retrospectively reviewed. The 21
medical records were reviewed for
demographics, including sex, onset,
and date of diagnosis, clinical
characteristics including location and
laterality. Ocular findings were
reviewed for the presence of ptosis,
refractive errors, strabismus,
amblyopia, and proptosis.
RESULT
There were a total 21 cases of
hemangiomas included in the study.
76% (16) were diagnosed with
capillary hemangiomas and 24% (5)
were diagnosed with cavernous
hemangioma.
 Characteristics group of Table 2. Location of the Capillary
capillary hemangiomas are given in Hemangioma
Table 1. There were 75% (12) females N=16 %
and 25% (4) males. History of Eyelid Laterality
premature birth and weight £ 2500 Right 8 50.0
grams occurred in 6.25% (1). There Left 8 50.0
were no history of amniocentesis in Bilateral 0 0
all patients. The mean maternal ages Upper Eyelid
recorded at childbirth for each Not further
5 41.7
patients were 27 years. Average age defined
onset of capillary hemangioma was at Nasal 3 25.0
3.3 months old. Temporal 3 25.0
Central 0 0
Table 1. Characteristics of the Central and
1 8.30
Capillary Hemangioma temporal
N=16 % Lower eyelid
Not further
Sex 2 50.0
defined
Female 12 75.0
Nasal 2 50.0
Male 4 25.0
Temporal 0 0
Premature birth 1 6.25
Weight £ 2500
1 6.25
grams
Table 3. Complications of the
Maternal age at
27 Capillary Hemangioma
birth, mean, years
N=16 %
Age of onset, mean,
3.31 Refractive errors 7 43.75
month
were
Secondary 5 31.25
The locations of the capillary
glaucoma
hemangiomas in 16 patients are given
Amblyopia 3 18.75
in Table 2. Eyelid laterality were
Ptosis 5 31.25
equal, there were 50% (8) had
capillary hemangioma on the right
eyelid, and the other 50% (8) on the Among 16 patients with
left eyelid. In this study none of the capillary hemangioma, there were
patients had bilateral capillary 31.25% (5) had ptosis and 60% (3)
hemangioma. There were 75% (12) had closed visual axis due to ptosis.
patients had capillary hemangiomas There were no patients with
on the upper eyelid and 25% (4) had strabismus, proptosis, and optic nerve
on the lower eyelid. compression in this study.
Complications due to capillary Therapy for patient with
hemangioma are given in Table 3. capillary hemangiomas in Kariadi
Refractive errors were 43.75% (7), Hospital were per oral propranolol,
secondary glaucoma were 31.25% steroid injection, and extirpation.
(5), and amblyopia were 18.75% (3). There were 31.25% (5) had
propranolol therapy, 18.75% (3) had
steroid injection, 37.5% (6) had
extirpation and 12.5% (2) had only
need conservative treatment.
Outcome of the therapy were good
with the average of follow up 14.625
months. There were improvement of
therapy such as decreasing of IOP,
axis visual was open because size of
the tumor decreased, and
improvement of visual acuity.
Characteristics group of
cavernous hemangiomas were 60%
(3) females and 40% (2) males.
Average age onset of capillary
hemangioma was at 36 years old.
Eyelid laterality were 60% (3) had
cavernous hemangioma on the right
eyelid, and 40% (2) on the left eyelid.
Table 4. Complications of the
Cavernous Hemangioma
N=5 % In our study, the amblyopia rate
Refractive errors
4 80.0
were
Secondary
3 60.0
glaucoma
Limited ocular
2 40.0
movement
Proptosis 5 100
Complications due to cavernous
hemangioma are given in Table 4.
Refractive errors (hyperopia) were
80% (4), secondary glaucoma were
60% (3), limited ocular movement
were 40% (2), and proptosis were
100% (5).
Therapy of cavernous
hemangiomas were extirpation in 4
patients and observation in 1 patient.
Outcome of the therapy were good
with the average of follow up 9.4
months. There were improvement of
therapy such as decreasing of IOP and
proptosis, improvement of ocular
movement and visual acuity.
DISCUSSION
Aim of this study was to
characterize clinical features and
complications of patients with
hemangiomas in Kariadi Hospital
Semarang.
According to our result, ratio
female:male = 3:1 for capillary
hemangiomas. This clinical result
confirm that females are more likely
to develop hemangiomas than males.
Drolet, Stigmar, and Alniemi report
9 10 11
a similar sex predominance. It is still
unclear why capillary hemangiomas
are more common among females.
Most of lesions in this study
were in the upper eyelid (75% [12]).
Previous studies show similar
11,12,13
finding, although the reason for this
predilection has yet to determine.
was 18.75% (3). Which is lower than
rates described in prior study.
Incidence of amblyopia varies from
76% (older studies) to 21% (recent
studies). Most common mechanism
3
to induce amblyopia in periocular
capillary hemangioma is unequal
refractive error between the eyes.
Typical refractive error that
associated is astigmatism, caused by
pressure on the globe from the lesion
and distortion of cornea. Less
common mechanism for amblyopia is
blockage visual axis by mechanical
ptosis. Result of this study were
5
31.25% (5) had ptosis and 60% (3)
had closed visual axis due to ptosis.
In our study, ratio female:male
= 3:2 for cavernous hemangiomas.
Females are more likely to develop
cavernous hemangioma than males. 14
Complications due to cavernous
hemangioma were refractive errors
(hyperopia) (80% [4]), secondary
glaucoma (60% [3]), limited ocular
movement (40% [2]), and 11. Alniemi AT, Gregory JG, Nancy D, Brian
proptosis (100% [5]). Outcome of the GM. Incidence and Clinical
Characteristics of Periocular Infantile
therapy for both capillary and Hemangiomas. Arch Ophthalmol. 2012
cavernous hemangioma were good. Jul; 130(7): 889–893.
12. Weiss AH, Kelly JP. Reapraisal of
REFERENCES astigmatism Induced by periocular
1. Gunduz Kaan, Zeynel A Karcioglu. capillary hemangioma and treatment with
Vascular Tumors. In: Karcioglu A intralesional corticosteroid injection.
Zeynel, editor. Orbital Tumors: Diagnosis ophthalmology. 2008. Feb;115(2):390-
and Treatment. New Orleans: Elsevier, 397.e1.
2012. 13. Dubois J, Milot J, Jaeger BI, McCUaig C,
2. Bang M Genie, Pete Setabutr. Periocular Rosseau E, Powell J, Orbit and Eyelid
Capillary Hemangiomas: Indications and Hemangima: Is There A Relationship
Options for Treatment. Middle East Afr J Between Location and Ocular Problem?.
Ophthalmol. 2010 Apr-Jun; 17(2): 121– Am Acad Dermatol. 2006 Oct;55(4):614-
128 9.
3. Tavakoli M, Saeid Yadegari, Mahnaz M, 14. McNab AA, Jennifer ST, Jing Xie, Dinesh
Maryam A, Hossein S, Wendy WL. S, Thoms GH, Julia S. Bret O. The
Infantile Periocular Hemangioma. J Natural History of Orbital Cavernous
Ophthalmic Vis Res. 2017 Apr-Jun; Hemangiomas. Ophthal Plast Reconstr
12(2): 205–211 Surg 2015;31:89–93)
4. Codere Francois, Julie Powell. Current
Conceps in the Management of Infantile
Hemangiomas: Steroid, Beta-Blockers, or
Surgery. In: Guthoff RF, Katowitz JA,
editor. Oculoplastics and Orbit. Berlin:
Elsevier: 2010.
5. Samuelov L, Michael K, Karen R,
Meghana K, Sarah LC, Bahram R,
Anthony JM. Risk Factor for Ocular
Complications in Periocular Infantile
Hemangiomas. Pediatr Dermatol.
2018;00:1–5.
6. Dutton J Jonathan.Modern Concepts in
Orbital Imaging. In: Guthoff RF,
Katowitz JA, editor. Oculoplastics and
Orbit. Berlin: Elsevier: 2010.
7. Nasr AM, Grace AC. Ultrasinigraphy in
Orbital Differential Diagnosis. In:
Karcioglu A Zeynel, editor. Orbital
Tumors: Diagnosis and Treatment. New
Orleans: Elsevier, 2012.
8. Golden N. Tjokorda GBM, Christopher
R. Surgical Outcome of Orbital
Cavernosus Hemangioma: a Case Series.
9. Drolet BA, Esterly NB, Frieden IJ.
Hemangiomas in Children. N Engl J Med.
1999 Dec 23;341(26):2018
10. Stigmar G, Crawford JS, Ward CM,
Thomson HG, Ophthalmic sequelae of
infantile hemangiomas of the eyelid and
orbit. Am J Ophthalmol. 1978
Jun;85(6):806-13.