SUBGLOTIC CAVERNOUS HEMANGIOMA EXTIRPATION THROUGH

TRACHEAL STOMA WITH ENDOSCOPY

Sutji Pratiwi Rahardjo

Otorhinolaryngology Department
Faculty of Medicine, Hasanuddin University, Makassar

Abstract

A hemangioma is a benign neoplastic tumour rise from proliferating endothelial

blood vessel, that may occur in any part of the body, such as face, orbital space, liver,
nasopharynx, and larynx/subglotic.
Hemangioma considered vascular tumor that mostly involve the area of head-
neck(60%) and 10% of laryngeal incidence. Jones, e.f reported, from 270 cases of benign
laringeal tumor, hemangioma were found in 5 cases (2%). This tumour rarely found in
larynx , especially subglotic, however, once occurred can lead to significant airway
obstruction.
Male, 39 years old, came with a chief complaint of dysphony since 1 year ago and
worsen slowly, with difficulty in breathing and history of active smoker. From ENT
examination and fiberendoscopic examination showed there was a tumor mass at the right
subglotic, histopathology post-operative described as cavernous hemangioma, unusual
case for 39 years old man. The patient was treated successfully.

Keywords : cavernous hemangioma, subglotic

Abstrak

Hemangioma adalah tumor jinak asal pembuluh darah, dapat terjadi pada dimana
saja pada seluruh tubuh, seperti muka, orbita, hati, nasofaring dan laring/ subglotik.
Hemangioma diperkirakan sebagai tumor pembuluh darah yang tersering
melibatkan daerah kepala-leher (60%) dan 10% predileksinya pada laring. Jones,dkk
melaporkan dari 270 kasus tumor laring jinak, hemangioma ditemukan 5 kasus (2%).
Tumor ini jarang ditemukan pada laring, terutama area glotik, sekali muncul maka resiko
sumbatan jalan napas dapat terjadi.
Laki-laki usia 39 tahun dengan keluhan utama disfoni sejak 1 tahun yang lalu,
tidak progresif, disertai kesulitan bernapas dan riwayat perokok aktif. Dari pemeriksaan
fisis THT dan endoskopi fleksibel tampak massa tumor pada area subglotik kanan, dan
dari histopatologi pasca operasi menunjukkan hemangioma kavernosum, kasus yang
jarang pada usia 39 tahun. Pasien menjalani operasi dan hasilnya memuaskan

Kata kunci : hemangioma kavernosum, subglotik

Introduction
Hemangioma is a vascular
tumour most commonly invovle the
head and neck. Cases on hemagioma
of the larynx are rarely found,
futhermore on adult where generally
hemangioma occurs in infant.(1,2,3)
The etiology of hemangioma
is still vague. It also concerns the
reason why hemangioma undergoes
regression. However, when there is a
defect from angiogenesi thus the
tendency of hemangioma is higher.(4,5)
Although the classification
of vascular tumour varies , mulliken
divides hemangioma into
categories,that is(6,7):
a) Capillary hemangioma with
histologic findings, show
vascular proliferation with the
lumen were lined by the layer of
endothelial cell. In capillaries
lumen, red blood cell were
found. Stroma consist of
connective tissue with a different
density.
b) Cavernous hemangioma
with histologic findings, shows
proliferation of large vessel
sinuses and widening of the thin
wall and covered by single layer
endothelial cell. Vessel sinus
space filled with red blood
cells/erythrocyte.
c) Mixed hemangioma,
combination of cavernous and
capillary hemangioma.
Case Report
Male, 39 years old, came to
ENT policlinic in RSU. Labuang Baji
Makassar with a chief complaint of
dysphony since 1 year ago and
worsen slowly, reside for the last 3
month. Difficulty in breathing started
since 1 month ago, increase on
activities. There is no complaint of
swallowing difficulty, nose and ear
abnormalities. History of hemoptysis,
twice, an active smoker since school.
On physical examination,
patient with good general status and
normal vital, without stridor. From
indirect laryngoscopy and flexible
endoscopy examination shows a
tumour mass on left anterior
commissure, enclose partially1/3 of
the anterior glotic without
abnormalities on vocal cord/vocal
fold movement. Tumour mass is not
fixated, redish-white in colour.
Neck CT-scan investigation, Fig.2b. Tumour mass on examination
0
axial section reveals left glotic with 30 endoscopy through tracheal
tumour with no sign of destruction. stoma
Labaratory and chest x-ray findings
are normal. Post operative evaluation on
the fifth day with indirect
laryngoscopy shows no remaining
tumour mass, slight oedema and
hyperemic of vocal cord and
3 arythenoid. Patient has no sign of
difficulty in breathing untill trachea
decanulation were done and vocal
rest is advised/recommended.
Fig 1. Neck Computed Tomography on
axial view
On 22nd of March 2007,
direct laryngoscopy and tracheostomy
had been performed. On direct
laringoscopy, shows a tumour mass Fig.3a. Vocal cord during respiration
originating from right subglotic and
nonfixated. Base of tumour is not
seen and hard to reach with
instrument. Extirpation later were
done through tracheal stoma with a
helped of 30 endoscope and
intermittent insuflasion anesthesi.
Tumour fully removed and tumour
specimens were sent to pathological
anatomy division.
Fig.3b. Vocal cord during phonation
On 14th days, voice return to
normal and no dyspnea. Flexible
endoscopy show no tumour mass,
hyperemic surface of left vocal cord,
with normal movement. On
histopathological findings demons-
Fig.2a. Tumour mass on examination trate a cavernous hemangioma.
with direct laryngoscopy
Discussion
Imunohistochemical
research demonstrate that angiogenic
factor, specially the basic fibroblast
growth factor (bfgf) and vascular
endothelial growth factor (vegf)
increases during proliferation phases
(0-12 month). Along that periode,
interferon (inhibitor endhothelial
migration) decrease on tumour
surface. After a year, regression phase
will be active. Angiogenic factor
proportion/level will start to
decrease,while endothelial inhibitors
will be produced again and epidermal
interferon will return to the normal
level. Periodically, hyperactive
endothelial cell will be dead, and
remaing are inactive endothelial.(7,8,9)
Subglotic hemangioma in adults
usually asymptomatic when the size
is still small. For one large
hemangioma usually can cause
hoarsness, horse cry, chronic cough,
inspiratory/expiratory stridor until
dyspnea.(10,11,12)
Suspicion of subglotic
hemangioma can be confirmed
through ENT physical
examination. Larynx flexible
endoscopy is essential form the
diagnosis of hemangioma and to
exclude any possibility other
laryngeal anomaly. Lesion ussually
located posteriolaterally to subglotic
submucous, may occurs unilaterally
or bilaterally, or located on upper
region of trachea. Lesiaon usually
blueish red, stalking and non
fixated/easily compressible.(10,13,15)
Ct-scan, shows assymetrical
narrowing of the subglotic area. MRI
could give mora accurate picture,
however, general anesthesis or
sedation may be needed in children.
Whenever diagnosis is still
questionable, biopsy is a must for ther
lesian , with a high risk of
bleeding/hemorrhage.(11,14,16)
In this case, patient
complaints of dysphony for over 1
year. From our investigations, we
diagnosed this patient as subglotic
tumour, most propably a tumour from blood vessel that have a
hemangioma.
Delay diagnosis can occur
due to hemangioma character as
asymptommatic lesion while the size
is still small, after the mass getting
larger, and the manifestation appears,
patientwill come and seek maedical
advice.
Hemangioma in children and
adults prone to be different due to
infantile hemangioma will have a
spontanous regression, so docter and
family members will only perform an
observation. In adult,lesion do not
undrgoes regression, and when it is
located at the airway thus can cause
phonation and respiratory
malfunction.
Management depend on
patient age, location and size of the
lesion and hemangioma
hemodinamic. Optional
management : observation, steroid
tharapy, embolization, chryotherapy,
sclerotherapy, antifibrinolitic agent,
radiation therapy, photocoagulation
therapy, surgical with or without
preoperative embolizationor
combination theraphy.(9,11,15)
Hemangioma surgical
exsicion, from a few references, is
the best option due to its permanent
relief. Lesion that always excise, is a
small lesion that do fail to regress or a
large lesion that disturb the body
function or cause a massive cosmetic
effect.(9,15)
Subglotic hemangioma
extirpation in this patient, nearly
complete with the moderate
instrument with the help of
endoscope. One of the key to
accomplish this technic, is the
expertise of the anasthesiologist in
performing intermittent insuflation
anesthesis. This technic is higly risky,
due to its complication of aspiration,
hypoxia, and hypercarbia. However
this risk does not occurr in this
patient. Two weeks Post operative
follow up results,patient felt that his
voice had return to normal, no
difficulty in breathing and aspiration,
cough, and less sputum.
Due to the origin of the
high tendency to recurs, gradual
observastion are necessary.
REFERENCES
1. Anonymous. Subglotik
hemangioma. In Clinical issues
in pediatric otolaryngology.
Volume 39, issue 21. Available
from : URL: http://www.audio-
digest.org/cgi-bin/html.
2. Shapsay M Stanley, Rebeiz E
Elie. Benign lessions of the
larynx. In: Bailey J. Byron,
Calhoun Karen, editors, Head
and neck surgery –
otolaryngology. 2nd ed. Volume
1. Philadelphia: Lippincott raven
publishers. 1998. p.759-60.
3. Muliken John. Editorial :
Updated hemangioma.
International pediatric journal/
Vol.14/ No.3/ 1999/ p.132-134.
4. Martin G H Regina, Neto L C
Arlindo, Semenzzate Graciela,
Lapate Renan. Case Report :
Laryngeal hemangioma. Rev
Bras otorinolaryngol 2006;72
(4): 574.
5. Tucker M Harvey. The larynx:
Benign neoplasms. 2nd ed. New
York: Thieme medical
publishers. 1993. p.279-282.
6. Dhingra PL. Disease of ear, nose
and throat: Benign tumours of
the larynx. 2nd ed. New Delhi:
B.I.Churcill Livingstone. 2002.
p.299-302.
7. Putz R, Pabst R. Sobotta: Atlas
Anatomi Manusia. Edisi 20. Ali
bahasa : Hadinata I. Jakarta :
EGC Penerbit Buku Kedokteran,
1997 : p.120-130.
8. Ballenger JJ. Penyakit
granulomatosis kronik larung.
Dalam : Penyakit Telinga Hidung
Tenggorok, Kepala dan leher.
Edisi 13. Alih bahasa : Staf THT
FKUI. Jakarta : Binarupa Aksara,
1997 : p.547-551.
9. O’Mailey Bert W.. Hemangioma
of the head and neck. Baylor
college of medicine [serial
online] June 25,1992 [cited
February 8,2006]. Available
from: URL:
http://www.bcm.edu/oto/grand/6
2592.html
10. Tewfick Ted L. Congenital
malformations of the larynx
[online] April 27, 2006 [cited
March 31,2007]. Available from:
URL:
http://www.emedicine.com/ent/to
pic324.htm
11. Sudiona Jati, Kurniadhi Budi,
Hendrawan Adhy, Djimantoro
Bing. Penuntun praktikum
patologi anatomi: Hemangioma
kapiler, hemangioma
kavernosum. Jakarta: Penerbit
buku kedokteran EGC. 2001.
p.53-5.
12. Ludwick James. Hemangioma
and vascular malformations of
the head and neck. Baylor
college of medicine [serial
online] March 21, 2002 [cited
February 8,2006]. Available
from: URL:
http://www.bcm.edu/oto/grand/0
32102.htm
13. Robin PE. Tumour of the larynx.
In: Oloffson Jan, Hibert John
editor, Scott-Brown’s
otolaryngology. 6th ed. Oxford:
Butterworth-heinemann. 1997.
p.4-6.
14. Hyams JV, Heffner DK.
Laryngeal pathology. In Tucker
M Harvey editor, The larynx. 2nd
ed. New York: Thieme medical
publishers. 1993. p.49-60.
15. Banovetz John. Gangguan laring
jinak. In: Adam George,
Lawrence Boeis, Higler Peter
editors, Boeis buku ajar penyakit
THT. Edisi 6. Jakarta: Penerbit
buku kedokteran EGC. 1997.
p.378-91.