An experience of hyperbaric oxygen and transurethral coagulation combination therapy for abnormal vessels and intractable macrohematuria due to pelvic arteriovenous malformations

doi:10.4236/crcm.2013.27106

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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: [email protected]

Received 6 August 2013; revised 5 September 2013; accepted 16 September 2013

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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