The Seoul Journal of Medicine Vol. 36, No. 1 :23-28, March 1995
Recurrence of Completely Excised Arteriovenous Malformation : Review of the Literatures
and Possible Explanation
Sang Hyung Lee, MsD. and Dae Hee Han, M.D
De~purrnrc~t~t c j f ' R c ~ ~ r o a r r ~ q c ~ r ~ ~ , Sejolrl Ntrtiorltrl L'tlirc.r.\.i!\, C'ollogr~ o f ' .2fc~rlic,i,10 Sc~olrl 1 1 0 - 7 4 4 , Korc~tr
= Abstract =
The authors report a case of recurrent arteriovenous malformation
(AVM), which has been completely removed and disappeared on postoperative angiography four years ago. Initially, a t the age of nine years, she presented intracerebral hematoma. The angiography demonstrated an aneurysm and AVM nidus located in the posterior frontal area, fed by the branches of the anterior and middle cerebral arteries. Eleven days after the ictus, rebleeding occurred, so left fronto-parietal craniotomy was done emergently. The hematoma and AVM nidus with aneurysm were removed. Her postoperative course was uneventful and postoperative angiography showed that the AVM had been completely excised.
However, four years later, sudden focal motor seizure on right leg developed.
Magnetic resonance(MR) images and angiography demonstrated that the AVM reappeared on the previously operated region. Extirpation of the recurrent AVM was carried out. We do emphasize a long term follow-up MR images or a repeated angiography is essential to confirm the complete absence after excision of the AVM.
Key words :
Arteriovenous malformation, Hematoma, Angograplzj,
,Mugrloric- reson- ance ( M R ) images, Reappear, Extirpation
INTRODUCTION
After complete removal of the AVM whlch was proved by postoperative anglography reported Cerebral anglography has been considered the cases o f the recurrence of AVM on prev~ously best method for dlagnoslng the AVM, and lntra- operated region are very rare (Fuwa et a1 1988, or postoperative anglography was strongly rec- Hlguchl et a1 1991 . Patll, 1982) We hereln pres- ommended for confirming complete exclslon of ent such a case and call attent~on that a long an AVM (Lazar et a / , 1971 , Smlth, 1977) term follow-up MR Images or a repeated anglog-raphy I S essential
CASE REPORT
First admission
Thls 9-year-old glrl suffered abrupt vomltlng
Fig. 1. A, B, C and D Left c a r o t ~ d ang~ography after the ln~tlal hemorrhage demonstrates the aneurysm (small arrow) and the AVM (large arrow) fed by left per~callosal artery left callosomarg~nal artery and pre-roland~c branch of left m ~ d d l e cerebral artery (A B) S~xteen days after o p e r a t ~ o n anglography demonstrates complete removal of the aneurysm and the AVM, and the presence of hemocllp
(C,
D)and dysarthr~a Several hours later, the level o f consslousness deterlorated On adm~sslon, she was sl~ghtly drowsy but properly or~ented Neuro- log~cal e x a m ~ n a t ~ o n revealed rlght hem~pares~s (Grade IIIV), pathologlc reflex on r ~ g h t s ~ d e and neck st~ffness The computer~zed tomography ( C T ) showed ~ntracerebral hematoma In left posterlor frontal area Carot~d ang~ography demonstrated an aneurysm and AVM n ~ d u s on posterlor frontal
area It was fed by w~dened left per~callosal artery left callosomarg~nal artery and p r e - r o a n d ~ c bran ch of left m~ddle cerebral artery (Fig 1 A B ) and d r a ~ n e d to superlor s a g ~ t t a slnus
Eleven days after the onset sudden severe headache occurred and followed by deterloratlor- of consciousness The b r a ~ n CT showed erilarged hematoma so emergent left fronto-par~etal cranl- otomy was done The hematoma and the AVM nl-
Fig. 2. A & B H i s t o o g ~ c f~ndrngs The biopsy rev- ealed small artery and thickened vein ( a r - row )
dus with aneurysm flere removed S~xteen days after the o p e r a t ~ o n postoperat~ve angiography demonstrated complete exclslon of the AVM and aneurysm iFlg 1 C D l a n d she was d ~ s c h a r g e d w ~ t h r ~ g h t h e m ~ p a r e s ~ s (Grade IlllV)
The h~stologic e x a m ~ n a t ~ o n revealed small ar- tery a n d t h ~ c k e n e d veln (Fig 2 A)
Second Admission
Durlng follow-up, her school performances was g o o d and the r ~ g h t hem~paresls Improved u p to Grade IVIV Four years after the exclslon o f AVM.
sudden focal motor selzure o n rrght leg devel- oped MR Images (2 O T , Goldstar, Korea) showed tortuous s ~ g n a l v o ~ d mass w ~ t h s u r r o u n d ~ n g h ~ g h
Fig 3 A & B Magnetrc resonance Images T 2 weighted rnagnet~c resonance lrriage show s signal v o ~ d mass wrth surrounding h ~ g h s ~ g n a l Intensity l e s ~ o n o n p r e v o u s oper- ated reglon ( A ) a n d d ~ s a p p e a r a n c e of sig- nal v o ~ d mass ( B )
s ~ g n a l Intensity l e s ~ o n o n previous operated re- g l o n ( F i g 3 A) Angiography demonstrated that the AVM has rseappeared It was larger than f o r - mer AVM and fed by left callosomarg~nal a n d per-
~callosal arteries a n d pre-roland~c branch o f left m ~ d d l e cerebral artery (Fig 4 A . 6 ) Second oper- atron was done, a n d postopratrve anglography (Fig 4 C D ) and MR I m a g e s i F ~ g 3 B ) showed complete exclsion of the recurrent AVM
Fig. 4. A, B, C a n d D. Left carotid anglography Angiogram after the focal motor seizure demonstrates reappearance of the A V M ( A . B) Fourteen days after o p e r a t ~ o n . angiography demonstrates complete removal of the recurrent AVM
(C
D )The h ~ s t o l o g ~ c e x a m ~ n a t ~ o n revealed same nat- ure o f prevlous examination (Fig 2 B i
DISCUSSION
Cerebral AVM 1s not a true neoplasm but a c o n g e n ~ t a l development of vessels during a n earl1 e m b r y o n ~ c stage Therefore t heoret~cally there could not be true growth of AVM But many das- es s h o w ~ n g progressive enlargement o f cerebral 4VM have been reported(Hook a n d Johanson
1958 K r a y e r i b ~ ~ h l 1977 Morioka et a1 1988 Sano
et
a1 1978 Spetzler a n d W ~ l s o n 1975 Wal t ~ r n o 1973) Several poss~ble rnechan~sms for growth or enlargement o f AVM were sumrnar~zed as f o o w ~ r i g s ( M o r ~ o k ael
a1 19881 ( 1 1 the pro- gresslve vascular d~latatron or pseudoaneurvsnial f o r m a t ~ o n occurlng as a result o f repeated occult hemorrhages that destroy the extravascc~lar neur- al t~ssues i K r a y e n b ~ ~ h l 1977 W a ~ t m o 19731 12) the c o r i t ~ n u o u s her~iodvnarnic stress causing pro gresslve enlargement of the t h ~ r i \vxalled untlfferentiated vessels formlng the f~stulous shunts of the AVM (Hook and Johanson, 1958), (3) the pres- ence of more room for growth, for example, in the sylvian flssure (Hook and Johanson, 1958; Waltlm- o. 1973), ( 4 ) the autonomic growth (Krayenbiihl, 1977; Spetzler and Wllson, 1975), and (5) the pres- ence of a "reserve" nidus, which was not opac- ified initially and becomes visible angiographlcally after a change In hemodynamics(Sano
et a/.,
1978). But. there are a few reports of spon- taneous dlsappearance of cerebral AVM (Golden and Kramer, 1978; Hook and Johanson, 1958;
Levine
et a / ,
1973). It was explained by spon- taneous thrombosis or emboll which occlude feed- ing vessels (Confortl, 1971 ), and at heroscleros~s (Levlneet a / .
1973).An AVM that 1s verlf~ed operatively andlor pat- hologically but I S not detected anglographlcally IS
usually called an anglographically occult AVM.
Several mechanisms why angiography have faded to demonstrate a vascular malformation were speculated and could be summarized. There are (1) a small size (Becker et al. , 1979; Cohen et al. ,
1982), (2) the part~al or complete thrombosis caus- ed by spontaneously or secondary to hemorrhage (Becker et al . 1979; Chin and Harper, 1983; Coh- en et al. , 1982. Davldoff, 1958; Golden and Kram- er, 1978; Kramer and Wing, 1977; Patll, 1982), (3) the compression by adjacent hematoma and edema or destruction in the time of hemorrhage (Cohen et al , 1982; Kramer and Wing, 1977), and (4) the spasm of the feeding vessels (Patil, 1982).
The recurrence of the AVM disappeared on pos- operative anglography was very rarely reported (Fuwa
et a/.,
1988; Higuchiet a/.,
1991 ; Patil, 1982). In the case of Higuchi et al. (Higuchl et al. , 1991), small abnormal vessels on postoperative angiography were noted retrospectively. But it was suggested that small abnormal vascular channels not demonstrated on postoperative angiography mlght have grown to nidus some years later (Fuwa eta/.,
1988; Patil, 1982). Our case would be a well recognized phenomenon namely, failure of angiography to demonstrate an AVM following surglcal excision with subsequent rediscovery of the AVM. So, we might consider that the small thrombosed vascular malforma-tions, which located In the surrounding area of the AVM and was not visualized on postoperative angiography, could have grown to the recurrent AVM four years later. Therefore we suggest that a long term follow-up MR images or a repeated
aggressive angiography is essential to confirm the complete removal after excision of the AVM, particularly in presentation with hemorrhage and in difficulty on hemostasis. Because in presen- tation with hemorrhage, the small vascular mal- formations could not be visualized on angio- graphy due to compression and/or thrombosis.
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