Occipito- Vertebral A nastomosis ; Its Clinical Signifi cance

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1 Neurol Med Chir (Tokyo) 20, 61-66, 1980 Occipito- Verterl A nstomosis ; Its Clinicl Signifi cnce Akinori KONDO Tsunemro KOYAMA, Junichiro ISHIKAwA nd Toshiki YAMASAKI Deprtment of Neurosurgery, Fukui Fukui, Jpn Red Cross Hospitl, Summry A ptient with n occipito-verterl nstomosis ssocited with interesting clinicl symptoms is presented. This 38-yer-old mle hd history of recurrent nd reversile ischemic episodes of the vertero-silr system for more thn 10 yers. Surgicl olitertion of this norml nstomotic chnnel resulted in grdul improvement of most of his clinicl symptoms. The stel phenomenon of lood supply from the vertero-silr system to the externl crotid rtery vi the nstomotic chnnel ws thought to e responsile for clinicl symptoms. Key words: Occipito-verterl nstomosis, vertero-silr insufficiency, stel phenomenon Introduction Since the first report of Kryeniihl nd Richter,") spontneous externl crotidverterl rtery nstomosis hs een frequently reported in the literture. Such extrcrnil-intrcrnil rteril nstomosis is found frequently in ssocition with olitertive lesions of the suclvin, crotid, or verterl rteries. Some reports, however, del with such n nstomotic pthwy even in the sence of olitertive lesions, nd only few uthors hve discussed, to dte, the clinicl significnce of this nstomosis from the functionl point of view.') We recently experienced ptient with spontneous occipito-verterl nstomosis who presented himself with recurrent ischemic vertero-silr insufficiency. Surgicl olitertion of this nstomotic chnnel improved clinicl symptoms. Circultory disturnce in the re supplied y vertero-silr rteries ppered to hve produced clinicl symptoms. diplopi nd dysrthri ut without disturnce of consciousness. Spinl tp reveled norml cererospinl fluid. Neither cervicl nor intrcrnil vsculr normlities were found y ilterl crotid rteriogrms. Three months lter when he ws dischrged, there ws only slight numness in his right finger tips. In 1969, he gin complined of mild hedche nd slight motor wekness with sensory impirment on the right side of the ody. His symptoms lso improved within couple of months with conservtive tretment. In 1970, he experienced three trnsient ttcks of diplopi. Bilterl crotid nd right retrogrde rchil rteriogrms were reported to show no vsculr normlities. He ws dischrged without specific tretment. In 1976, he gin developed motor wekness nd numness on the right side of his ody which were ssocited with diplopi, dysrthri nd git disturnce. He ws finlly dmitted to Fukui Red Cross Hospitl in Mrch, Neurologicl Exmintions Cse Report In 1965, 38-yer-old mle cutely developed motor wekness nd numness on the right side of his ody which were ssocited with Neurologicl exmintion on dmission reveled tht he ws n lert nd well-oriented mn who ws mrkedly dysrthric. Pupils were isocoric nd normlly rective to light nd ccommodtion. Horizontl eye move-

2 62 A. KONDO ET AL. ments were lmost completely restricted on either lterl gze, ut oth verticl eye movement nd convergence remined intct. There ws verticl nystgmus prticulrly on upwrd gze, which did not show ny positionl prepondernce. Otologicl exmintions such s SISI, Bekesy nd speech discrimintion tests showed slight decrement of hering on oth sides which were indictive of retrocochler lesion. Other crnil nerves were intct. There ws slight right hemipresis with mild hyperreflexi, ut without ny pthologicl reflexes. Senstion ws modertely reduced in ll modlities on the right side of the ody. Coordintion ws slightly impired in the right upper nd lower extremities. He ws unle to stnd stright nd on tndem position. His git ws rod-sed nd txic. Lortory studies were ll within norml limits. Cererospinl fluid study ws norml. Rdiologicl Exmintions Trnsfemorl ilterl crotid nd verterl rteriogrms were performed using KIFA red ctheter. The speed of injection nd the mount of contrst dye were 6 ml/sec for verterl rteriogrm (totl 24 ml) nd 4 ml/sec for selective externl crotid rteriogrm (totl 20 ml), respectively. Neither rteriosclerotic chnges nor olitertive lesions were noted in oth cervicl nd intrcrnil vessels. Bilterl posterior communicting rteries were not opcified on oth right nd left common crotic rteriogrms ut were poorly visulized it selective verterl rteriogrm (Fig. 3). A lrge single nstomotic chnnel which linked the left occipitl rtery with the left verterl rtery t the level of C 1 ody ws visulized y the left common crotid rteriogrm. Selective left occipitl rteriogrm opcified the silr rtery more clerly through this nstomotic chnnel nd simultneously visulized the rnches originting from the silr trunk (Fig. l, ) (Fig. 2, ). Selective left verterl rteriogrm conversely visulized this nstomotic chnnel which lso filled the left occipitl rtery (Fig. 3, ). Surgery In April, 1977, under generl nesthesi, the nstomotic chnnel etween the left occipitl nd left verterl rteries ws exposed t the posterior mrgin of the C 1 lmin nd this nstomotic chnnel ws clipped (Fig. 4, ). Postopertive Neurologicl Findings Approximtely two weeks fter the surgery, his right hemipresis improved nd he ws le to write smll letters. Sensory impirment of the right hlf of the ody disppered except Fig.1 Lterl projection of the selective left occipitl rteriogrm. ) Erly phse of this rteriogrm reveled nstomotic chnnel (longer rrows) originting from the occipitl rtery (shorter rrows) t the posterior rim of the formen mgnum. ) Lter phse of the rteriogrm well opcified silr nd verterl rteries f through the nstomotic pthwy (rrows), nd lso rnches originting rom the silr rtery.

3 OCCIPITO-VERTEBRAL ANASTOMOSIS 63 for slight numness t the finger tips on the right side. He ws le to stnd on one foot nd to wlk stright, ut tndem git ws still unstle. He lso ws le to red smll letters more clerly. Dysrthri remined unchnged for while postopertively. An mplitude of verticl nystgmus ws remrkly reduced 2 weeks fter the surgery, ut the frequency of nystgmus remined unchnged. Impired horizontl movements of oth eyells grdully improved, ut monoculr horizontl nystgmus newly ppered on either side of ttempted conjugte lterl gze. In My, 1978 when he ws lst seen, he hd no pprent motor wekness of extremities, ut slight numness of the right finger tips ws still present. Dysrthri improved remrkly, so did horizontl movements of oth eye lls. Fig. 2 ) Hlf xil projection of the selective externl crotid rteriogrm visulized single nstomotic pthwy (rrows) etween the left occipitl nd verterl rteries. Bsilr rtery is poorly opcified in this phse. ) Axil view lso clerly demonstrted lrge single nstomotic chnnel (rrows) which well filled the vertero-silr rteril system in this phse. Fig. 3 Selective verterl rteriogrm. ) Erly phse of this rteriogrm conversely opcified the nstomotic chnnel (thinner rrows) nd peripherl prt of the occipitl rtery (thicker rrows). ) Lter phse of the rteriogrm lso visulized the nstomotic pthwy (thinner rrows) nd more clerly demonstrted the occipitl rtery (thicker rrows). Posterior communicting rtery is poorly opcified y the selective verterl rteriogrm (left upper rrow).

4 64 A.KONDO ET AL. Fig.4 Postopertive rteriogrm (lterl projection). ) Selective occipitl rteriogrm did not opcify either nstomotic chnnel or vertero-silr system. Arrow shows the site of olitertion. ) Selective verterl rteriogrm did not visulize the nstomotic chnnel either. Monoculr horizontl nystgmus, however, ws still noticele on conjugte lterl gze, ut reduced in its mplitude. Discussion There hve een vrious norml ngiogrphicl chnnels known etween crotid nd verterl rteril systems such s persistent primitive trigeminl rtery,28) persistent primitive coustic rtery,3) persistent primitive hypoglossl rtery,16) persistent protlnt rtery nd persistent cervicl intersegmentl rtery.1,5,8) These nstomotic chnnels hve een reported to e ssocited with plsi or hypoplsi of verterl rteries. Externl crotid-verterl rtery nstomosis hve een found incidentlly y ngiogrphy in cses of vsculr occlusive disese, trum, tumors, neurysms nd ostructive hydrocephlus.6,31,33) Creful exmintions of 53 dult cdvers y Schulze nd Suerrey25) demonstrted tht there were direct communictions including smll musculr rnches etween occipitl nd verterl rteries in most cses. Thirty-one reported cses of occipito-verterl nstomosis were summrized y Gito et l.11) in 1975 nd 3 dditionl cses hve een reported to dte.13,27) The ngiogrphic incidences of occipito-verterl nstomosis re, however, reltively low, i.e. 8 cses out of 100 trnsfemorl crotid rteriogrms,11) 4 out of 2108 crotid nd 315 verterl rteriogrms29) nd 2 out of 30 verterl rteriogrms.21) A lrge single nstomotic chnnel verified y ngiogrphy ws shown only in 8 cses in the foregoing literture,9,10,12,14,17,19,20,22) nd ll of those reporters concluded tht such nstomotic chnnels were congenitl. Although the nstomotic chnnel ws seen more frequently in the presence of olitertive lesions of suclvin, crotid, or verterl rteries,26) 17 out of 31 cses with spontneous nstomosis etween crotid nd verterl rtery were reported to hve no concomitnt oliter - tive lesions, which were verified ngio- grphiclly.11) These findings indicte tht this nstomotic chnnel cn e seen either with or without concomitnt olitertive vsculr diseses. According to Alpers et l.2) norml configurtion of the circle of Willis ws found in 52% of routine utopsy cses nd n nomly ws most common in posterior communicting rteries. In the present cse, poor opcifiction of ilterl posterior communicting rteries in crotid nd verterl rteriogrms ppers importnt for the clinicl symptoms, prticu

5 OCCIPITO-VERTEBRAL ANASTOMOSIS 65 lrly when there ws depletion of lood flow in the silr rtery. Lzorthesis) reported tht the externl crotid rteril system supplies the rin stem nd cervicl spinl cord through its communicting chnnel to the vertero-silr system. It is known tht the connection etween externl crotid nd verterl rteries is possily ville for utiliztion when either internl, common or verterl rtery is oliterted. In some cses, this nstomotic chnnel ecme pprent fter surgicl ligtion of crotid rtery. This is proly due to the ltered hemodynmic lnce fter the surgery.7'30) When mstiction or swllowing ecomes very ctive, there my occur steling of lood supply from the vertero-silr system to the externl crotid rtery through the norml nstomosis nd my cuse some reversile ischemic chnges in the rin stem.4,24,32 As shown in the present cse, when no olitertive disese is found in the cervicl or intrcrnil vessels, the direction of flow in the nstomotic chnnel cn e ltered either wy, from the verterl to occipitl or from the occipitl to verterl rteries nd this pthwy my deprive the rin stem of uxilliry flow to externl crotid territory depending on the chnges of intrluminl pressure grdient. Ngshim et l. 181 reported the possiility tht the vestiulr system s well s the oculomotor system in the rin stem re extremely sensitive to reduced lood flow or rupt reduction of intrluminl pressure grdient through the verterl nd silr rteries. In the present cse, preopertive neurologicl findings were suggestive of involvement of the left corticospinl nd spinothlmic trcts, possily t the level of the lower rin stem nd lso of medil longitudinl fsciculus nd prmedin pontine reticulr formtion. Reltively rpid postopertive improvement of clinicl symptoms pprently indictes tht the norml vsculr chnnel might hve cused circultory insufficiency of those structures which resulted in their functionl impirment. References 1) Ae, K. nd Suzuki, T.: Persistence of emryonic crotid-verterosilr nstomoses. Foli Psychitr Neurol Jpn 18: , 1964 [in Jpnese]. 2) Alpers, B. J., Berry, R. G. nd Pddison, R. M.: Antomicl studies of circle of Willis in norml rin. A.M.A. Arch Neurol Psychit 81: , ) Altmnn, F.: Anomlies of the internl crotid rtery nd its rnches; Their emryologicl nd comprtive ntomicl significnce. Lryngoscope 57: , ) Brnett, H. J. M., Wortzmn, G., Gldstone, R. M. nd Lougheed, W. M.: Diversion nd reversl of cererl lood flow; Externl crotid rtery "stel." Neurology 20: 1-14, ) Bloch, S. nd Dnziger, J.: Protlntl intersegmentl rtery. Neurordiology 7: 5-8, ) Bosnik, M. A.: Cervicl rteril pthwys ssocited with rchiocephlic occlusive disese. Am I Roentgenol 91: , ) Cntu, R. C., Ferris, E. J. nd Bker, E. P., Jr.: Collterl circultion to prietl rteriovenous mlformtion following ilterl externl crotid rtery ligtion. J Neurosurg 28: , ) Conforti, P., Armenise, B. nd Glligioni, F.: Anomlous crotid verterl nstomosis; primitive cervicl segmentl rtery. Neurochirurgi (Stuttg) 9: , ) Ito, K., Kwguchi, S. nd Iwuchi, T.: Externl crotid-silr nstomosis: Cse report. Neurol Surg (Tokyo) 3: , 1975 [in Jpnese]. 10) Flynn, R. E.: Externl crotid origin of the dominnt verterl rtery. Cse report. I Neurosurg 29: , ) Gito, Y., Miysk, K., Tkhshi, M. nd Kowd, M.: Occipito-verterl nstomosis. Jpn J Clin Rdiol 20: 17-22, 1975 [in Jpnese]. 12) Hckett, E. R. nd Wilson, C. B.: Congenitl externl crotid-verterl nstomosis. A cse report. Am J Roentgenol 104: 86-89, ) Kmiss, A., Skguchi, S., Kwnum, S. nd Ngshim, C.: Bilterl occipito-verterl nstomosis with internl crotid system. Jpn.7 Clin Rdio! 22: , 1977 [in Jpnese]. 14) Kuchin, M., Ysui, N., Tni, S., Somed, K. nd Mtsumur, H.: Externl crotid-verterl nstomosis. Jpn J Clin Rdio! 22: , 1977 [in Jpnese]. 15) Lzorthes, G., Gouze, A., Bstide, G., Sntini, J., Zdeh, O. nd Burdin, P.: L vsculriztion rterielle de l moelle cervi-

6 66 A. KONDO ET AL. tle. Rev Neurol 115: 1005, ) Lindgren, E.: Percutneous ngiogrphy of the verterl rtery. Act Rdiol 23: 389, ) Miur, I., Ryu, R. nd Mtsudir, J.: A rre cse of externl crotid silr nstomosis. J Keio Med Soc 43: , 1966 [in Jpnese]. 18) Ngshim, C., Iwm, K., Skt, E. nd Miki, Y.: Effects of temporry occlusion of verterl rtery on the humn vestiulr system. J Neurosurg 33: , ) Nimur, A., Sito, A., Nomur, S. nd Hsegw, T.: Externl crotid-silr nstomosis. Brin Nerve (Tokyo) 22: , 1970 [in Jpnese]. 20) Ro, T. S. nd Sethi, P. K.: Persistent protlntl rtery with crotid-verterl nstomosis. J Neurosurg 43: , ) Richter, Hs. R.: Collterls etween the externl crotid rtery nd the verterl rtery in cses of thromosis of the internl crotid rtery. Act Rdio! 40: , ) Smr, K., Scoville, W. B. nd Yghmi, M.: Anstomosis of crotid nd silr rteries: Persistent primitive trigeminl rtery nd hypoglossl rtery. Report of two cses. J Neurosurg 30: , ) Schechter, M. M.: The occipitl-verterl nstomosis. J Neurosurg 21: , ) Schrmek, A., Hshmoni, M., Meir, M. nd Jhel, M.: The suclvin stel syndrome. Isr J Med Sci 10: , ) Schulze, H. A. F. nd Suerrey, A.: Zur Frge der Anstomosen zwischen der A. verterlis nd der A. occipitlis. Zl Neuro. chir 2: 76-81, ) Sole-Llens, J. nd Plns, M.: Occipitl. verterl nstomosis in cse of middle cererl rtery occlusion. Neurordiology 1: 88-91, ) Sueyoshi, S., Jimo, M. nd Tkeuchi, K.: The externl crotid (occipitl)-verterl ns. tomosis. Brin Development (Tokyo) 4: , 1972 [in Jpnese]. 28) Sutton, D.: Anomlous crotid-silr nstomosis. Br J Rdio! 23: , ) Tkhshi, H., Ymguchi, K., Uemur, K., Kowd, M., Mtsuok, S. nd Ito, Z.: Four cses of occipitl-verterl nstomosis. Brin Nerve (Tokyo) 23: , 1971 [in Jpnese]. 30) Tmki, N., Ishikw, M. nd Ymshit, H.: Cererl collterl circultion following occlu. sion of common crotid rtery on one side nd internl crotid rtery on the other side. Brin Nerve (Tokyo) 21: 25-32, 1969 [in Jpnese]. 31) Ttelmn, M.: Pthwys of cererl collterl circultion. Rdiology 75: , ) Toole, J. F. nd Mcgrw, C. P.: The stel syndromes. Ann Rev Med 26: , 1974, 33) Youmns, J. R. nd Scrcell, G.: Extrcr nil l collterl cererl circultion. Neurology 11: , 1961.