Giant venous aneurysm associated with hypogastric arteriovenous malformation Francisco VALDES, MD, Albrecht KRAMER, MD, Mario FAVA, MD, Francisco CRUZ, MD, Hector CROXATTO, MD, Santiago, Chile Venous aneurysms are extremely rare. They may be congenital or acquired in origin and occasionally related to arteriovenous communications. A 58-year-old man complained of dull left lower quadrant pain and constipation. On physical examination a soft deep mass was palpated. Ultrasonogram and CT scan revealed a cystic formation in the pelvic cavity. Angiograms disclosed an arteriovenous malformation (AVM) at the pelvic floor draining into a large cavity. The patient was successfully managed by intraoperative selective embo- lization of the AVM and partial resection of a 10.6 • 8 • 6.7 cm venous aneurysm. The his- topathologic studies of the wall confirmed a venous structure. Venous dilatation has been reported in high flow vein grafts, blood access V fistulas and rarely, proximal to traumatic AV fistulas of the lower extremities. The etiology of the present case is probably congeni- tal, being tc the best of our knowledge, the first case affecting the hypogastric territory, reported in the English literature. (Ann Vasc Surg, 1986, 1, 143-146). KEY-WORDS : Venous aneurysm. - - Arteriovenous malformation. Although clinically recognized by Osler [1] early this century, true venous aneurysrns are seldom re- ported. They have been found isolated or associated with arteriovenous communications. Excepting those related to trauma, the etiology remains unknown. Clinical presentation depends on its location and as- sociated complications due to local compression or pulmonary embolization. The present report descri- bes our experience with a giant venous aneurysm of the hypogastric territory associated with a pelvic arteriovenous malformation (AVM). Department of Vascular Surgery, Department of Radiology, Department of Pathology, Catholic University Hospital and School of Medicine, Santiago, CHILE. Presented at the XVIlth World Congress of the Internatioxal Society for Cardiovascular Surgery, August 25-20, 1985, Monte- carlo, Monaco. Reprint requests : F. Vald~s, MD, Department of Vascular Sur- gery, Catholic Universi~ Hospital and School of Medicine, Mar- coleta 347, Santiago, CHILE. CASE REPORT A 58 year-old Caucasian male presented with a 3 months his- tory of dull pain of the left lower quadrant, occasionally colicky in character, radiating to the left lumbar area. Associated complaints included impotence and recent onset of constipation. He denied a history of palpitation, dyspnea or trauma. Sixteen years before he had passed a urinary stone. The physical examination disclosed a soft mass deep in the left lower quadrant. Peripheral pulses were normal and without bruits. On the rectal examination the mass was well defined, painless, slightly pulsatile and a thrill could be felt. An intravenous pyelogram (Fig. 1) showed extrinsic compres- sion of the bladder and displacement of the terminal left ureter without dilatation. The ultrasonogram (Fig. 2a) revealed a cystic mass 10.6 x 6.7 x 8 cm occupying most of the pelvic cavity. A CT scan (Fig. 2b) confirmed the vascular nature of the cyst and its relation to the surrounding structures. An abdominal aortogram (Fig. 3a) showed mild dilatation and tormosity of the left common iliac and hypogastric arteries. Selective left iliac catheterization (Fig. 3b) demonstrated an AVM at the pelvic floor fed by hyper- trophied branches originating distal to the superior gluteal artery. Late films showed filling of an aneurysm enclosed by the arterial vessels. Selective films of the right iliac artery failed to show connections with the AVM. Emptying of the aneurysm could not be traced. The patient underwent elective laparotomy. The pelvic ana- tomy was distorted by the tortuous left iliac vessels and the aneu- rysm which displaced the bladder and compressed the rectum.
Transcript
Giant venous aneurysm associated with hypogastric arteriovenous malformation
F r a n c i s c o V A L D E S , M D , A l b r e c h t K R A M E R , M D , M a r i o F A V A , M D , F r a n c i s c o C R U Z , M D , H e c t o r C R O X A T T O , M D , Santiago, Chile
Venous aneurysms are extremely rare. They may be congenital or acquired in origin and occasionally related to arteriovenous communications. A 58-year-old man complained of dull left lower quadrant pain and constipation. On physical examination a soft deep mass was palpated. Ultrasonogram and CT scan revealed a cystic formation in the pelvic cavity. Angiograms disclosed an arteriovenous malformation (AVM) at the pelvic floor draining into a large cavity. The patient was successfully managed by intraoperative selective embo- lization of the AVM and partial resection of a 10.6 • 8 • 6.7 cm venous aneurysm. The his- topathologic studies of the wall confirmed a venous structure. Venous dilatation has been reported in high flow vein grafts, blood access V fistulas and rarely, proximal to traumatic AV fistulas of the lower extremities. The etiology of the present case is probably congeni- tal, being tc the best of our knowledge, the first case affecting the hypogastric territory, reported in the English literature. (Ann Vasc Surg, 1986, 1, 143-146).
A l t h o u g h c l in ica l ly r e c o g n i z e d by O s l e r [1] ea r ly this c e n t u r y , t r u e v e n o u s aneu rys rns a re s e l d o m re- p o r t e d . T h e y h a v e b e e n f o u n d i so l a t ed o r a s s o c i a t e d wi th a r t e r i o v e n o u s c o m m u n i c a t i o n s . E x c e p t i n g t h o s e r e l a t e d to t r a u m a , t h e e t i o l o g y r e m a i n s u n k n o w n . C l in ica l p r e s e n t a t i o n d e p e n d s on its l o c a t i o n and as- s o c i a t e d c o m p l i c a t i o n s d u e to loca l c o m p r e s s i o n o r p u l m o n a r y e m b o l i z a t i o n . T h e p r e s e n t r e p o r t descr i - bes o u r e x p e r i e n c e wi th a g i an t v e n o u s a n e u r y s m of t h e h y p o g a s t r i c t e r r i t o r y a s s o c i a t e d w i t h a p e l v i c a r t e r i o v e n o u s m a l f o r m a t i o n ( A V M ) .
Department of Vascular Surgery, Department of Radiology, Department of Pathology, Catholic University Hospital and School of Medicine, Santiago, CHILE.
Presented at the XVIlth World Congress of the Internatioxal Society for Cardiovascular Surgery, August 25-20, 1985, Monte- carlo, Monaco.
Reprint requests : F. Vald~s, MD, Department of Vascular Sur- gery, Catholic Universi~ Hospital and School of Medicine, Mar- coleta 347, Santiago, CHILE.
C A S E R E P O R T
A 58 year-old Caucasian male presented with a 3 months his- tory of dull pain of the left lower quadrant, occasionally colicky in character, radiating to the left lumbar area. Associated complaints included impotence and recent onset of constipation. He denied a history of palpitation, dyspnea or trauma. Sixteen years before he had passed a urinary stone. The physical examination disclosed a soft mass deep in the left lower quadrant. Peripheral pulses were normal and without bruits. On the rectal examination the mass was well defined, painless, slightly pulsatile and a thrill could be felt. An intravenous pyelogram (Fig. 1) showed extrinsic compres- sion of the bladder and displacement of the terminal left ureter without dilatation. The ultrasonogram (Fig. 2a) revealed a cystic mass 10.6 x 6.7 x 8 cm occupying most of the pelvic cavity. A CT scan (Fig. 2b) confirmed the vascular nature of the cyst and its relation to the surrounding structures. An abdominal aortogram (Fig. 3a) showed mild dilatation and tormosity of the left common iliac and hypogastric arteries. Selective left iliac catheterization (Fig. 3b) demonstrated an AVM at the pelvic floor fed by hyper- trophied branches originating distal to the superior gluteal artery. Late films showed filling of an aneurysm enclosed by the arterial vessels. Selective films of the right iliac artery failed to show connections with the AVM. Emptying of the aneurysm could not be traced.
The patient underwent elective laparotomy. The pelvic ana- tomy was distorted by the tortuous left iliac vessels and the aneu- rysm which displaced the bladder and compressed the rectum.
A N N A L S OF 144 VENOUS A N E U R Y S M A N D H Y P O G A S T R I C A V M A L F O R M A T I O N VASCULAR SURGERY
Fig. 1. ~ Intravenous pyelogram showing displacement o f the bladder and left ureter by a mass located to the left in the pelvic cavity.
The mass was firm and slightly pulsatile. Temporary occlusion of the hypogastric artery was followed by disappearance of pulsati- lity, allowing compression and partial emptying of the aneurysm, facilitating dissection through its base where the AVM became evident. Direct intraoperative embolization was performed with gelfoam flakes injected distal to the superior gluteal artery. This maneuver permitted a safe opening of the aneurysm and to achieve a quick control of venous back bleeding from two large openings draining into the left iliac vein. No mural thrombus was found�9 The left hypogastric artery was then ligated. After suture ligation of the arterial and venous openings, the sac was partially resected. The wall of the aneurvsm was thick, with venous aspect. The histopathologic study of" the specimen revealed intimal thickening and phlebosclerosis (Fig. 4).
Recovery and follow-up for one year has been uneventful. Postoperative angiogram showed absence of residual lesions (Fig. 5).
DISCUSSION
The first venous aneurysm was reported by Beau- mont [2] in 1867, following the autopsy of a patient who suffered a traumatic arteriovenous fistula of the femoral artery. However the first clinical report was done by Osler [1] almost fifty years later. Since then, sporadic cases of venous aneurysms affecting the extremities, visceral veins or superior vena cava have been reported [3]. Based on Abbott 's sugges- tion, venous aneurysms are classified in to congeni- tal, acquired, isolated or associated with arteriove-
% - # ; : _ . , *
2a
Fig. 2. - - a) UItrasonogram of the pelvis. A cystic forma- tion occupies most of the cavity, b) CT scan disclosing a large mass (*) f i l l ed w i th contrast, located between the bladder and rectum.
nous communications [4]. Most isolated venous aneurysms present in young people and involve the superior vena cava. They are usually fusiform and rarely reach more than 5 cm but may be complica- ted by pulmonary embolism from mural thrombus.
Thompson in 1969 reviewed the literature referring to central venous aneurysms associated to arteriove- nous fistulas [5]. However, most of the cases were secondary to longstanding traumatic arteriovenous fistulas of the lower extremities and the venous aneurysm presented in the external iliac vein. In the recent review by Yao [3] only two cases presented in the upper extremity.
The present case, apparently congenital, would be the first case affecting the hypogastric territory. The factors involved in the progressive dilatation of the veins include the flow rate determined by the size and duration of the fistula, the compliance of the
VOLUME 1 N o 1 - 1986 VENOL'S A N E U R Y S M AND H}"POG.4STRIC A!/ M.4LFORMATIO,Y [45
3a 3b
i ~
Fig. 3. - - a) Abdominal aortogram showing tortuosity and dilatation of the left common lilac and hypogastric arteries. Branches distal to the superior gluteal artery fill an arterio- venous malformation and a large aneurysmal sac . b) Selective catheterization of the left common iliac artery.
Fig. 4. - - Microscopic view of the aneurysmal wall. Intimal thickening and increased f ibrous tissue in the outer layers. (Elastic van Gieson stain ; original magnification x 80).
Fig. 5. - - Postoperative abdominal aortogram. There is occlusion of the left hypogastric artery and complete disappearance of the AVM and venous aneurysm.
perianeurysmatic tissues and the hydrostatic pressure determined by the anatomical situation of the abnor- mality. It is interesting that although the fistula may be located in the popliteal area, the dilatation may present in the external iliac vein. Possibly local fac- tors determined by the histologic structure of the
vein are also involved. In our case, the aneurysm arouse in a vein draining into the common lilac, most likely the hypogastric vein. The increased fi- brous tissue and intimal thickening of the aneurys- mal wall are secondary to the longstanding increased pressure. In previous cases, the fistula was known
ANNALS OF 146 VENOUS A N E U R Y S M A N D HYPOGASTR1C A V M A L F O R M A T I O N VASCULAR SURGERY
for up to 36 years [3]. Endophlebosclerosis and endo- ph lebohyper t rophy have been repor ted as patho- logic features in cases of primary venous aneurysms [6]. Due to the slow growing rate, most of the cases have evolved asymptomatically becoming clinically evi- dent as a mass or as an autopsy finding. Our case became evident due to compression of bladder and rectum. Spontaneous rupture has never been repor- ted. Systemic complications are possible if the in- creased flow leads to high cardiac output failure or the aneurysm harbors thrombus which potentially could result in pulmonary embolism.
Currently, the treatment of choice of isolated AVM of the pelvis is intraarterial embolization [7]. The association of a venous aneurysm demands sur- gical excision, t he re fo re a comb ined approach should be the therapy of choice. In the present case, symptomatic relief was achieved by direct intraope- rative selective embolization and partial resection, allowing complete control of the arterial feeding as well as venous drainage.
REFERENCES
I. OSLER W. - - An arteriovenous aneurysm of the axillary vessels of 3(~ years duration. Lancet, 1913, 2, 1248.
2. BEAUMONT W.R. - - Clinical lecture on a case of large circumscribed traumatic aneurysm of the superficial femoral artery, etc. Medical Times and Gazette (London), 1867, 2, 87.
3. YAO J.S.T., VAN BELLEN B.. F L I ~ W.R., BERGAN J.J. - - Aneurysms of the venous system. In : BERGAN J.J., YAO J.S.T. - - Aneurvsms, Diagnosis and Treatment, pp. 515-530. New York, Grune and St'ratton, 1982.
4. ABBOTY B.A. - - Congenital aneurysm of superior vena cava. Report of a case with operative correction. Ann Surg, !950,131, 259-263.
5. THOMPSON N.W., L INDENAUER S.M. - - Central venous aneu- . rysms and arteriovenous fistulas. Ann Surg, 1969, 170, 852-856.
6. SCHATZ I.J., FINE G. - - Venous aneurysms. N Engl J Med, 1962, 266, 1310-1312.
7. OLCO'I-Y C. IV, NEWTON T.H., STONEY R.J., EHRENFELD W.K. - - Intraarterial embolization in the management of arteriovenous malformations. Surgery, 1976, 79, 3-12.
V A L D E S F . , K R A M E R A . , F A V A M . , C R U Z F . , C R O X A T I ' O H. - - A n e u r i s m a v e n o s o asoc iado a m a l f o r m a c i d n A - V hipogast r ica . A n n Vase Su rg , 1986, 1 ,143-146 .
R E S U M E N : Los aneurismas venosos son muy raros. Pueden ser de origen cong6nito o adquirido estando relacionados, f recuentemente, con comuni- caciones arterio-venosas. Se trata de un var6n de 58 afios, afecto de dolor sordo en regi6n iliaca iz- quierda, impotencia y constipaci6n intestinal, con antecedentes de litiasis renal. En la exploraci6n clf- nica se aprecia un masa blanda e indolora, con li- gero latido y thrill a nivel de la fosa iliaca. Tanto la ecograf/a como el TAC ponen de manifiesto una formaci6n qufstica en cavidad pelviana. La arterio- grafia muestra una pequefia dilataci6n de la arteria iliaca primitiva junto a una malformaci6n arterio-ve- nosa pelviana, que drena hacia una gran cavidad sin conexi6n con las arterias iliacas contralaterales. E1 paciente rue tratado quirurgicamente con 6xito me- diante embolizaci6n selectiva peroperatoria de la malformaci6n arterio-venosa y resecci6n parcial del a n e u r i s m a v e n o s o q u e c o n un t a m a r i o de 10,6 x 8 x6,7 cm. ocupaba la mayor parte de la fosa iliaca con desplazamiento del ureter y de la ve- jiga, y compresi6n del recto. E1 estudio anatomopa- tol6gico de la pared confirm6 la estructura venosa de la malformaci6n. No se presentaron complica- ciones postoperatorias. La posibilidad de dilataci6n venosa ha sido descrita en injertos venosos de alto flujo, en las fistulas A-V para accesos vasculares y raras veces en el sector proximal de las fistulas A-V traum~ticas de los miembros inferiores. En el caso descrito la etiologia es probablemente de origen cong6nito y creemos que se trata de la primera pu- blicaci6n en lengua inglesa de un caso de aneurisma venoso que afecta el territorio hipog~strico.
