Fallberichte - Case reports 54 Cerebral vasospasm with eleva ted creatine phosphokinase following surgery ofunruptured arteriovenous malformation Y. Takabata ke. J. Yamashita. S. ll igashi. T Yamoshitna. Y. Yamamoto Department of'Neurosu rgery. Kanaza wa Universlty. Schoolof'M edicine. Ka nazawa. Japa n Delayed cerebra l vasospasm as sociated with m ark ed eleva tion of seru m crea tine ph osph okin ase (CPK) is reported. A 21-yea r-old man with unruptured arteri oveno us ma lforma tion (AVM) in the left tempor al lobe developed post op erati ve sym ptom atic vaso spa sm lasting for 2 weeks . A postoper ative com puterized tomography sca n showe d intraventr icula r hemat om a without su ba ra chno id hem orrha ge in the basal ciste rn. On day 5 after surge ry, the pa tient developed motor aphasia due to marked vasospas m around the bifur ca tion ofi nternal ca rotid a rtery . On da y 7 and 8, serum CPK was eleva ted as high as 11 75 a nd 137 6 lU/I, resp ectively. On day 35 wh en h e had comp letely recover ed from cere bral vasospasm bot h clinically and angiographica lly, the level ofC PKwas nor malized . The ca usa tive factors of symptomatic cerebral vasospasm following surgery of unruptured AVM ass ociated with CPK elevation a re discussed . Key-Words Arter iovenous malfor mation vasospas m - Creatine pho sp hokinase Cerebral lt is widely acc epted that deiayed cere hral vasospasm following suba rac hnoid hemorrh age is usually as sociated with rupture d an eur ysrns, but rarely with a rt eriovenous m alformations (AVMs) (2, 3,4 ,6). Fu rther more. very few cases ofsym ptom ati c vasos pa srn due to AVMs have been pre viously re por ted. This pap er is to report a case with sympt oma tic cere bra l vas ospas m which was associa ted with m a rked eleva tion of se ru m creatine phosph okinase (CPK)after surge ry of unr uptur ed AVM. Neurochirurgia 35 (1992) 54-56 © Georg Thleme Verlag Stuttgart . New York Zerebraler Vasospasmus bei er höh ter Ph osphokinase nach Operation eine r intakt e n arteriove nösen Mißbild ung Die Verfasser ber icht en üb er einen ve rzögerten zere bra len Vaso spas mus mit eine r a usg eprägten Erhöhung der Serumkreatin phospho kina se . Ein 21jä h riger Mann mit einer intakten arteriovenösen Miß bildung im link en Schläfen la ppen ent wickelte postoperat iv einen 2 Woche n lang anh alten den symptomatischen Vaso spasm us. Postoper ative CT zeigte ein intr aventrikuläres Häm at om ohne suba rachnoida le Blutung in der basalen Ziste rne . Am 5. postoper ati ven Ta g entwickelte der Patient eine motori sche Aphas ie a ufgru nd eines ausgep rägten Vasospa smus a n der Ka rotisb ifurkation . Am 7. und 8. postopera tiven Ta g war die Serumkre atinphosphokinase au f 1175 bzw. 137 6 tEil erhöht. Am 35 . Tag, nach vollständiger klinischer und a ngiogra phischer Erholung vom zerebralen Vasospasrnus, hatt e sich de r Ser umkreatinpho sphokina sespi egel norm alisiert. Die ursä chlichen Fa ktore n des symptomatischen zerebralen Vaso spas m us nach Operation der intakt en a rte riovenöse n Mißbildung bei gleichze itig erhöh tem Kreati nph osph okinasespi egel we rde n erörte rt. Case Rep or t A 21-yea r-old ma n was ad mitted to our hosp ital on September 30. 1988 . because of repeated episodes of dizziness for 7 months. He had a past history offe br ile convulsions a nd head tr aum a with loss of consciousness for a few da ys in his childh ood. Neurological exa minatio n on ad mission revealed no a bnorm ality except for left external str a bismus . A computerize d tomograph y (eT l scan showed a low density area with pa rtial heterogeno us contras t enha ncement in the left tem poral lobe. A left car otid a ngle gram showed a large AVM in the left a nterior tempo ral lobe. The nidus measuring 65 x 40mm in diameter. was fed by the middle cere bra l and a nterior cho roidal arteries . an d dr ained by the veins of Trolard. Hcland o. RosenthaI, a nd l.ab be (Pig. 11. According to the Spetzler's gradi ng (5). it was ranked as grad e IV. T l weighted image or magnetic resonance Imagtng (MHIIshowed a hypointensity a rea with multiple signal vold regions in the a nterio r tempora l lobe. and also revealed porencephalic cavtty du e to old intracere bra l bleeding. Downloaded by: University of Pennsylvania Libraries. Copyrighted material. Summary Cerebm l msospasm with elenated creo tine phasp hokinase Neurochirurgia 35 (19 92) 55 Fig. 1 left carotid angiogram showing a large artenovenous maltormatten (65 x 40 mm in diameter) in the anterior portion of temporal lobe. Anterior view (Ieft) and lateral view (right). Fig. 2 Computenzed tomography sean2days after operanon showingintraventricularhemorrhage (right), but no hemorrhage in the basal cistem(reft). inferior horn ofthe left late ral ventricle was opencd during surgery. a mode ra te a mount ofblood flowed into the later al vent riele. His postoperative recovery was good. A postoperative CT sea n taken on day 2 showed intraven tricular hema toma duc 10 surge ry. but no hemorrhage in the suba rac hnoid spa ce (Fig. 21. Cere bral blood flow (CBF)st udy on day 4 sho wed a diffuse decrease ofCBF in both frontal a nd tempo ral lobos on the left side. On day 5 the patient had a tra nsie nt episode of motor aphas ia lastin g for 5 minutes. Serum CPK on that day was 75 IUlI (norm al ra nge: 0-93 IUII). The value of CPK was clcvated as high as 1155 IU/I on da y 7 and up to 13761U11 on day 8. At the sa me time. hls motor a phas ia became persistent a nd more scvcrc. Phen obarbital was given intra muscularly for the pur pose of sedation in dose of 100 mg thr ee times a day from day 5 to day 7. Intracra nial pressure measured by lumbar puncture on day 9 was 280 mmlt.O. Ccreb rospina l fluid (CSF) was xanthoch rom ic, co nt a i n i n~ 47 mg/dl of proteln. 51 mg/dl of sugar a nd 25 leucocytes/mm . On da y 10, his ne urological conditions further deter iorated with total a phasia. perseveratic n and Iiteral para phasia. Surprisingly a left ceren d anglegra m on day 12 showed severe cerebral vasos pas m around the bifurcation of intern al carotid artery (Fig. 3 leftl. Then CPK isozyme pattern s were a nalyzed twice. Ther e was no CPK· BBat aU an d CP K·~1B was no more than 2% Isee belowl. There were no Fig.3 l eft carotid angiograms showing severe cerebral vasospasm around the bffurcatiön of intemal carobdarteryon day 12 (left), which had substded on day 35. •r I Downloaded by: University of Pennsylvania Libraries. Copyrighted material. A left fronte-t emporal cra niotomy was performed for resection of the AVM on Üctober 13. The superficial sylvian veins were reddish. dilated an d were drai ned to the dilated vein of Trola rd . Three mai n feeders from the middle cereb ral an d a nte ricr ehoroi dal arteries were coagulated a nd elipped via the tra nssylvian rout e. At the rear ofthe nidus was a lar ge poren cenhalic cyst. As the Neurochirurgia 35 (J 992) elec trocardiogra phic changes . e sF study o n day 14 showed diffuse decr eas e in both he mispheres . However, all of his neurological sympto ms gradually disappeared . On day 35, a left ca rotid a ngtogram showed co mplete reco ver y from the vasos pasm (Fig. 3 right). end bot h e SF and e PKwere also normalized . He wa s disch arge d o n day 46 witho ut neur ological deflcits. Comments It is weil known that the amount of hlood in the basal cistern is elosely related to the development of cerebral vasospasm in ruptured aneurysms (7). In case of AVMs, less am ount of blood extends to the basal cistern hecause of their distan t location (3). Accordingly, symptomatic cerebral vasospasm occurs far less frequently following rup ture of AVMs. The incidence of angiogra phic cerebral vasospasm in ruptured AVMs was variously reported as 8-3 1 % (2, 3, 4, 6). However , the majority ofpreviously reported cases were elinically asymptomatic. Although the CT scan on day 2 diselosed no hemorrh age in the basal cistern , the present case developed symptomati c cere bral vasospasm lasting for 2 weeks. Two possible factors may emerge as a cause ofthe vasosp asm. Firstly, as the tempo ral horn was incident alIy opened during surgery thro ugh a pre existing porencephalic cavity in the temporal lobe, a moder ate amount of hlood directly flowed into the tempor al horn . Consequentiy chemical mediators such as serotonin, oxyhemoglobin, prostagland ins , etc, might have dissolved from the elots in the lateral ventriele and entered into the hasal cistern . Secondly, the AVM was located only a few centimete rs dista l to the hifurcation of the middle cerebral artery, surgical procedures might have damaged perforating arte ries to the hypothalamus . The resultant hypothalamic disturb ance might have indu ced the liher ation of various vasoaetive agents into the basal cistern, or stimulated widespread sympathetic discharge with increased level of circulating catecholamines (11). Wilkins reported that increased catecholamines might provoke autonomie dysfunction, myocar dial necrosis (8, 9), and elevated CPK. CPK is a dimer consisting of 2 subunits, M (musele) and B (brain), while isozymes of CPKare MM , BB,and MB. CPK-MMis mainly involved in the skeletal museles, CPK-BB in the brain, and CPK-MB in the myocar dium. Yuu noted marked elevation of CPKas high as 2.1- 12.9 fold ofnormal value, after intramuscular injection of IOOmg of phenobarbital (12). However, we have ohserved only mild elevation of CPK after intramu scular injection of phenobarb ital in our several patients. Greenblatt reported that eerebral vasospasm does not eause an elevation of CPK in the CSF unless it induces cere hral infarction. Elevat- y. Takabatake. 1. Yamashita . S. Higashi. T. Yamashima , Y. Yamamoto ed CPK levels in the CSF was previously ass umed to be indicative of parenchymal damage (I ). Although intramuscular injections of phenobarbital might be one of the causa tive factors, it is unlikely that marked elevation of CPKwas solely due to that. It alone cannot explain dynamic CPK cha nges in this case. Dynamic CPK changes were elosely related with the time course of cerebral vaso spasm in this ease. The exact mechanism of elevated serum CPK in this ease still remains obscure . In summary, we would like to dra w attention to possible relationship between CPK and cerebral vasospasm. References Greenblatt. S. H.: Cerebrosp inal fluid creatin e phosphokinase in acute subarachnoid hemorrhage. J. Neurosu rg. 44 (19 76) 50 -5 4 2 Motsumori. K.. S. Asahi. K. Nakayama. Y. Miyasaka, T. Bepp u: Cerebral vasospasm after subarachnoid hemo rrhag e from arte riovenous malformation . No Shinkei Geka 11 (1983) 829 -83 4 3 Nishimura. K.. T. D. Haw kins: Cerebral vasospasm with subarac hI noid hem orrhage from arteriovenous rna lforma tlons of the brain . Neuroradiology 8 (1975 ) 20 1-2 07 4 Sasaki. T.. Y. Mayanagi. H. Yano, S . Kim: Cerebral vasospas m wit h subarachnoid hemo rrhage from cerebral arteriovenous malformations. Surg. Neurol. 16 (1981) 183-1 87 5 Spetder. R. F.. N. 11. Martin: A proposed grading syste m for arteriovenous malformations. J. Neurosurg . 65 (1986) 476 -48 3 6 St ornelli. S. 11., 1. D. 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Neurosurg. 58 (1983 ) 12 843-850 Yuu, H.: Diagnostic signiflcan ce of serum creatine kinase isoenzyme, MB fraction (CK-MB) in acute myocardial infarction. Nippon Naika Gakkai Zas shi 67 (197 8) 10 - 16 Tetsumori Yamashima. M. D. Dep artrnen t of Neurosurgery Kana zawa Universtty Schoo l ofMe dicine Takaramachi 13-1 Kanazaw a city. 92 0 Japan Downloaded by: University of Pennsylvania Libraries. Copyrighted material. 56