Otolaryngologia Polska tom 63, nr 6, listopad–grudzień 2009
KAZUISTYKA / CASE REPORTS 520
Endovascular Treatment of Hereditary Hemorrhagic Telangiectases
of the Tongue
Endowaskularne leczenie wrodzonej naczyniakowatości krwotocznej języka
Assist. Prof. Dragan Stojanov
1
, Prof. Petar Bosnjakovic
1
, Dr. Sasa Ristic
1
, Mila Bojanovic
2
,
Dr. Daniela Benedeto-Stojanov
3
Otolaryngol Pol 2009;
63 (6): 520-522
SUMMARY
Hereditary hemorrhagic telangiectasia, also known as Osler-Weber-Rendu
disease, is an autosomal dominant disorder involving vascular abnormalities
of various organs. Telangiectases are frequently observed, predominantly on
the nasal and oral mucosa. We present a case a of 53-year-old man with the
tonge hemorrhagic telangiectases and epistaxis. Selective catheterization and
embolization of the right lingual artery led to size redution and no bleeding
from tongue telangiectases.
Hasła indeksowe: wrodzona naczyniakowatość krwotoczna, leczenie endowa-
skularne
Key words: hereditary hemorrhagic telangiectases, endovascular treatment
©by Polskie Towarzystwo Otorynolaryngologów
– Chirurgów Głowy i Szyi
Otrzymano/Received:
02.11.2009
Zaakceptowano do druku/Accepted:
08.12.2009
1
Institute of Radiology, Clinical Centre Nis
2
ORL Clinic, Clinical Centre Nis
3
Clinic for Gastroenterology and Hepatology,
Clinical Centre Nis
Wkład pracy autorów/Authors contribution:
Według kolejności
Konflikt interesu/Conflicts of interest:
Autorzy pracy nie zgłaszają konfliktu interesów.
Adres do korespondencji/
Address for correspondence:
imię i nazwisko: Prof. Dragan Stojanov
adres pocztowy:
Institute of Radiology, Clinical Centre Nis
Bul. Dr. Zorana Djindjica 48
18000 Nis
Serbia
tel. +381631094197
fax +381184221469
e-mail drstojanov@gmail.com
Hereditary hemorrhagic telangiectasia (HHT), also
known as Osler-Weber-Rendu disease, is an autosomal
dominant disorder involving vascular abnormalities
of various organs. Telangiectases are frequently ob-
served, predominantly on the nasal and oral mucosa
and the skin of the face and hands [1, 2]. This results
in recurrent and sometimes severe bleeding, of which
epistaxis is the most common. Prolonged hemorrhages
originated from telangiectases of the base of the tongue
and the body of the tongue may requir treatment. To our
knowledge, there are no case reports of endovascular
treatment of the tongue hemorrhagic telangiectases.
The purpose of this report is to present endovascu-
lar treatment of hereditary hemorrhagic telangiectases
of the tongue.
Case report
A 53-year-old man presented with the tonge hemor-
rhagic telangiectases and epistaxisi. He had a family
history of HHT manifesting as telangiectasia of gas-
trointestinal (GI) tract and nasal mucosa. His medi-
cal history was significant for multiple mild to sever
nosebleeds, GI bleeds and a minor cerebral stroke. At
admission on ORL Clinic, patient had severe epistaxis
physical examination revealed multiple telangiectases
of the nasal mucosa (Fig. 1). In addition, scattered
telangiectases on the tongue were found (Fig. 1). The
patient underwent posterior nasal tamponade by ORL
spetialist. As nasal tamponade was not effective, en-
dovascilar treatment of the epistaxis was indicated.
After obtaining informed consent, patient was
brought to the angiography suite. Procedure was per-
formed by using conscious sedation and local an-
esthesia. Using standard technique, procedure was
performed with femoral artery access. After placement
of 5F arterial sheath, a 5F catheter was placed in the
right common carotid artery, and cervical angiography
was performed. Selective internal carotid artery (ICA)
and external carotid artery (ECA) digital subtraction
angiography was then performed with imaging over
the face and skull base.
After placing the catheter in the proximal ECA
trunk and performing an initial control angiogram,
catheter was then advanced over a guidewire into the
internal maxillar artery. After the embolization of the
internal maxillar artery, catheter was advanced in the
lingual artery and a superselective angiogram was
performed. It showed the characteristic patchy mucosal
telangiectasias on the thong apex and body (Fig. 2).
Embolization was performed using 3 ml of polyvinyl
alcohol (PVA) microspheres (Bead Block Terumo) in
range 300-500 μm. Embolization resulted in occlu-
sion of the small branches of the lingual artery and