Vol.2, No.7, 399-401 (2013) Case Reports in Clinical Medicine
http://dx.doi.org/10.4236/crcm.2013.27106
An experience of hyperbaric oxygen and
transurethral coagulation combination therapy for
abnormal vessels and intractable macrohematuria
due to pelvic arteriovenous malformations
Seiya Inoue1,2, Masashi Honda2*, Shinji Hirano2, Noriya Yamaguchi2, Bunya Kawamoto2,
Tsounapi Panagiota2, Katsuya Hikita2, Kuniyasu Muraoka2, Takehiro Sejima2, Chihiro
Takahashi1, At sushi Takenaka2
1Department of Urology, National Hospital Organization, Yonago Medical Center, Yonago, Japan;
2Department of Urology, Tottori University School of Medicine, Yonago, Japan;
*Corresponding Author: [e-mail]
Received 6 August 2013; revised 5 September 2013; accepted 16 September 2013
Copyright © 2013 Seiya Inoue et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
ABSTRACT
A 78-year-old man was admitted to our hospital
with a chief complaint of gross hematuria. The
patient was diagnosed with pelvic arteriovenous
malformations and repeated arterial emboliza-
tion. A cystoscopy demonstrated that bladder
trigone was covered by many clots. At the same
time, bleeding mucosa at the right side of the
bladder dome was confirmed. We administered
hyperbaric oxygen therapy 7 days after admis-
sion. Macrohematuria recovered gradually and
we then performed transurethral coagulation at
the dome with failed mucosa 14 days after ad-
mission. Macrohematuria recovered completely
and there was no recurrence during hospitaliza-
tion. Thirty days after admission, we performed
cystoscopy and found clear mucosa in the tri-
gone. The patient was discharged 32 days after
admission.
Keywords: Hyperbari c O xy g en T h erapy;
Macrohematuria; Pelvic Arteriovenous
Malformations; Tra nsurethral C oagulation
1. INTRODUCTION
Pelvic arteriovenous malformations (P-AVM) are rare,
especially in males. Angiograph y is effective not only for
the diagnosis, but also for embolization therapy of AVM
in the pelvis; howeve r, we sometimes have to repeat em-
bolization many times because of continuous symptoms
or insufficient efficacies. In this report, we attempted hy-
perbaric oxygen therapy (HBO) and transurethral coagula-
tion (TUC) for abnormal vessels and persistent macrohe-
maturia from P-AVM and showed good ef ficacies.
2. CASE REPORT
A 78-year-old man was admitted to our hospital with a
chief complaint of gross hematuria. It started 2 weeks
before his admission. He was diagnosed with P-AVM 7
years before his admission and bilateral iliac artery em-
bolization was performed a few times in another hospital.
He used gabapentin for the continuous supurapubic dull
pain that had continued for 20 years. There was no his-
tory of previous trauma or abdominal surgery. His phy-
sical examination was unremarkable. Laboratory data
showed urinalysis with many red blood cells per high
power field. Prothrombin time, partial thromboplastin
time, and platelet count were normal. Ultrasonography
demonstrated a hypoechoic region over the urinary blad-
der. An abdominal-pelvic computed tomography (CT)
scan demonstrated a large mass consisting of dilated-
tortuous abnormal vessels in the pelvis th at was adherent
to the anterior wall of the bladder and extended to the
bottom of bladder. An enhanced CT scan was performed
and we observed abnormal vessel invasion near the tri-
gone (Figure 1). Cystoscopy revealed bleeding mucosa
at the dome of the bladder wall (Figure 2). In addition,
the bladder trigone was covered by many clots where the
enhanced CT scan revealed abnormal vessel invasion.
We diagnosed macrohematuria from the dome of the
urinary bladder and abnormal vessels in the trigone due

S. Inoue et al. / Case Reports in Clinical Medicine 2 (201 3) 399-401
400
to P-AVM. We treated conservatively with tranexamic
acid and carbazochrome intravenously; then, started
HBO with 2.0 ATA for 60 min/day × 5 days/week for
four weeks. Macrohematuria ameliorated slowly. Thus,
we performed TUC 7 days after starting HBO. In this
surgery, bladder trigone was reddish and full of abnormal
microvessels (Figure 3). The dome still exhibited slight
bleeding from failed mucosa. TUC was performed at the
dome of bladder mucosa. After that, the patient exhibited
no recurrence of macrohematuria and an uneventful pos-
toperative course. On day 30, cystoscopy revealed clear
manifestation in the bladder dome. Surprisingly, most of
the abnormal vessels had disapp eared in the trigon e (Fig-
ure 4). He was discharged 32 days after being admitted.
The patient had no evidence of recurrence through 1
month of follow-up.
3. DISCUSSION
AVM is uncommon vascular lesions built by multi-
ple abnormal communications between arterial and ve-
nous systems without a normal intervening capillary
network [1]. AVM is generally agreed that the majority
of lesions are congenital, with a few that being acquired
Figure 1. A CT scan showed a large pelvic arteriovenous mal-
formation. Enhanced vessels are observed at the trigone of the
urinary bladder (arrow).
Figure 2. A Cystoscopy demon-
strated a mucosal hemorrhage at
the right side of the bladder dome.
Figure 3. A cystoscopy showed red-
dish mucosa and many abnormal ves-
sels in the bladder trigone 14 days after
admission.
Figure 4. A cy stoscopy showed a
normal mucosa in the bladder
trigone 30 days after admission.
after trauma or abdominal surgery. AVM in pelvic space
is rare. Rosenberg et al. reported the first case of AVM
of the bladder [2]. Presenting symptoms of P-AVM in-
clude pelvic discomfort and pain, rectal pain, genitouri-
nary complaints including hematuria, hydronephrosis,
hemospermia, and erectile dysfunction. However, most
are asymptomatic. Physical examinations can reveal a
pulsatile mass, and a harsh noise can be heard. When
P-AVM are suspected, an angiogram and pelvic CT or
magnetic resonance imaging (MRI) are essential to de-
lineate the extent of the disease. Diagnosis is confirmed
by angiography. CT and MRI are useful to demonstrate
the vascular etiology of the lesion, to evaluate its exten-
sion and involvement of adjacent structures, and to de-
monstrate the feeding vessels supplying the malforma-
tion and draining veins [3]. Ultrasonography reveals non-
specific hypoechogenic regions. However, it cannot dif-
ferentiate P-AVM from a cyst or an abscess. Colored
Doppler ultr asonography is useful to detect color images
of the vascular structure. Treatments consist of conven-
tional therapy, arterial embolization, or surgical excision
including transurethral resection, tumor removal, or liga-
tion of the feeding vessels [1]. If sympto ms are relatively
mild or the tumor is not growing, palliative therapy can
be selected. Recently, arterial embolization is the most

S. Inoue et al. / Case Reports in Clinical Medicine 2 (201 3) 399-401 401
common method used as the first line of treatment;
however, it often fails. The most suitable indication of
surgical treatment is still controversial; however, a lower
number of collaterals or a small tu mor volume is easy to
resolve with surgical treatment. HBO therapy is indi-
cated for patients with hemorrhagic cystitis caused by
radiation, intractable interstitial cystitis, or cyclophos-
phamide-induced hemorrhagic cystitis. Nakada et al.
demonstrated that essential macrohematuria patients
treated with HBO (2.0 ATA for 30 days) had a signifi-
cantly superior recovery than those without any treat-
ment. Moreover, the time period of recovery is shorter
than that of non-treated patients [4]. It has been demon-
strated that HBO is associated with vasoconstriction in
the systemic circulation and to a lesser extent in the
coronary vasculature [5]. Activation of plasma kinins
results in vasodilation, edema formation, or hypotension.
Animals treated in the hyperbaric chamber at a pressure
of 2.5 ATA saturated with 100% oxygen showed low
levels of plasma kinins [6]. Such treatment presumably
results in vasoconstriction and contributes to decrease in
hematuria. The biological mechanism of HBO originated
from hypersaturating oxygen dissolved in the plasma due
to the treatment. Subsequently, HBO treatment stimu-
lates leukocyte function characterized by phagocytosis,
producing growth factors that enhance angiogenesis [7].
HBO therapy is well tolerated, and adverse effects of
HBO are unusual, as visual disturbances, eustachian tube
dysfunction, and claustrophobia are rare [8]. To our
knowledge, there have been no reports about the use of
HBO and TUC combination therapy for P-AVM. The
present case showed the difficulties in treating macro-
hematuria in spite of repeated arterial embolization. We
performed HBO and TUC combination therapy for per-
sistent macrohematuria, and confirmed a recovery of
macrohematuria and reduction of abnormal vessels at the
trigone. During HBO therapy, the patient felt ear fulln ess
that resolved itself automatically. We supposed that un-
der a high pressure of oxygen, angiogenesis progressed
and abnormal vessels and damaged mucosa recovered.
As this is a case report of only one patient, our data pro-
vide the possibility of another treatment option for in-
tractable macrohematuria and abnormal vessels patients
who do not respond to conventional therapies. Careful
observations are required.
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