We reported a case of trigonal cavernous malformation (CM) with intraventricular hemorrhage. This 67-year-old woman experienced sudden onset of loss of consciousness and her Glasgow Coma Scale (GCS) was 5 points (E1V1M3) on admission. CT scan demonstrated intraventricular hemorrhage and acute hydrocephalus. Angiography did not demonstrate any vascular abnormality. Ventricular drainage was performed for acute hydrocephalus and the postoperative course was good. CT showed a hyperdense lesion in the left trigone, which was contrast-enhanced on T1-weighted MR. Removal of CM was performed via the left middle temporal sulcus. We conducted a Pub Med search for trigonal CM and found 17 cases. Herein we discuss the symptoms, CT and MR findings and treatment

Kasuya, Hidetoshi

Katsura, Kanade

Nakahara, Yoshikazu

Nanto, Masataka

Ogawa, Takahiro

Ohbuchi, Hidenori

Osaka, Yasuhiko

Tenjin, Hiroshi

Tokushima University Institutional Repository

INTRODUCTIONIntraventricular cavernous malformation (CM) israre, comprising only 2.5-10.3% of patients with cere-bral CMs (1, 2). Of intraventricular CM, only about20% are located in the trigone of the lateral ventricle(3). The first report of trigonal CM was published in1977 by Coin (4). Based on our search of PubMed,trigonal CM has been reported in 17 cases overabout 30 years. We encountered a rare case of trigo-nal CM and discuss the symptoms, CT and MR find-ings and the treatment.CASE REPORTExamination and preoperative courseA 67-year-old right-handed woman with a his-tory of hypertension, experienced a sudden onsetof consciousness disturbance. She was immediatelytransferred to our hospital. On admission, her GCS(Glasgow Coma Scale) was 5 points (E1V1M3), butthere was no paresis. The pupils were equal in sizeand reacted to light. Corneal reflexes were presentbilaterally. CT scan showed an intraventricular hem-orrhage in the left trigone, in the cerebral aqueductand acute hydrocephalus (Figure 1). Angiographydid not show any vascular abnormality. We per-formed ventricular drainage. Seven days after sur-gery, her condition was good. MR imaging showeda lesion measuring 10 mm within the trigone ofthe left lateral ventricle. It was isointense on T1-weighted, hypointense on T2-weighted, hyperin-tense on FLAIR imaging and hypointense on T2*CASE REPORTTrigonal cavernous malformation with intraventricularhemorrhage : A case report and literature reviewHidenori Ohbuchi1) 3), Yasuhiko Osaka1), Takahiro Ogawa1), Masataka Nanto1),Yoshikazu Nakahara1), Kanade Katsura2), Hiroshi Tenjin1), and Hidetoshi Kasuya3)1)Department of Neurosurgery, 2)Department of Pathology, Kyoto Second Red Cross Hospital, Kyoto,Japan ; and 3)Department of Neurosurgery, Tokyo Women’s Medical University Medical Center East,Tokyo, JapanAbstract : We reported a case of trigonal cavernous malformation (CM) with intraventricu-lar hemorrhage. This 67-year-old woman experienced sudden onset of loss of conscious-ness and her Glasgow Coma Scale (GCS) was 5 points (E1V1M3) on admission. CT scandemonstrated intraventricular hemorrhage and acute hydrocephalus. Angiography didnot demonstrate any vascular abnormality. Ventricular drainage was performed for acutehydrocephalus and the postoperative course was good. CT showed a hyperdense lesionin the left trigone, which was contrast-enhanced on T1-weighted MR. Removal of CMwas performed via the left middle temporal sulcus. We conducted a Pub Med search fortrigonal CM and found 17 cases. Herein we discuss the symptoms, CT and MR findingsand treatment. J. Med. Invest. 59 : 275-279, August, 2012Keywords : cavernous malformation, hydrocephalus, intraventricular hemorrhage, trigoneReceived for publication March 29, 2012 ; accepted May 18,2012.Address correspondence and reprint requests to HidetoshiKasuya, Department of Neurosurgery, Tokyo Women’s MedicalUniversity Medical Center East, 2 -1 -10 Nishiogu, Arakawa-ku,Tokyo 116-8567, Japan and Fax : +81-3-3819-8680.The Journal of Medical Investigation Vol. 59 2012275imaging and well enhanced with gadolinium on T1-weighted imaging (Figure 1).SurgeryThe patient underwent total removal of the massvia the left middle temporal sulcus. The middletemporal sulcus was incised and the left ventricleopened. The choroidal plexus was hypertrophic, red,and led to the trigonal lesion. The mass was bluish-red and packed with thin-walled vessels. A drainingvein was seen leading out of the lesion to the innersurface of the lateral ventricle. The mass was totallyremoved.Postoperative courseThe patient transiently demonstrated mild dys-lexia and mild acalculia, with no visual field defects.She did not show any neurological findings 3 weekslater. Postoperative MR imaging confirmed that thelesion in the trigone of the left lateral ventricle hadbeen removed.Pathological FindingsHistologically, the lesion was composed of dilatedvessels filled with blood. The vascular walls werelined by endothelial cells with fibrous stroma. Therewas no glial tissue between the vessels. The patho-logical diagnosis was CM (Figure 2).DISCUSSIONIntraventricular CMs occur in only 2.5-10.3% ofpatients with cerebral CM (1, 2). Trigonal CM iseven rarer. Among the 17 cases of trigonal CM thatwe have collected, the median patient age was 36.1years and there were eight males and nine females(Table). Lesion size was reported to be around 1 cmor larger. Our case was a so-called microangioma(5).Figure 1 Admission CT scan showing a hyperdense lesion in the trigone of the left lateral ventricle, and a small mount of hemor-rhage in the cerebral aqueduct along with acute hydrocephalus (A). The lesion was isointense on T1-weighted (B) ; hypointense onT2-weighted (C) ; hyperintense on FLAIR (D) ; and hypointense on T2* MR (E). Contrast -enhanced MR image showing Gd-DTPAenhancement (F).Figure 2 Dilated vessels (*) lined by endothelial cells withfibrous stroma. There was no glial tissue between the vessels.H & E,100H. Ohbuchi, et al. trigonal cavernous malformation276The first symptom was seizure in three cases(17.6%), intraventricular hemorrhage in three(17.6%) and mass lesion in eleven (64.7%) (Table).The symptoms of mass lesion showed a higher in-cidence than seizure or intraventricular hemorrhagesin trigonal CMs. Katayama et al. reported that thereason for the higher incidence of the mass effectmay be direct compression of vital structures. Cav-ernous malformation is often voluminous as it growsin a preexisting cavity (6).Among 138 cases of intracranial symptomatic CMsreviewed by Simard et al. 40 (29%) presented withextralesional hemorrhage. A similar incidence (27%)of subarachnoid and/or intraventricular hemorrhagewas reported in intraventricular CMs (7). Amongthe 77 cases of intraventricular cereberal CMs re-viewed by Kivelev et al. , 11 (14%) presented withintraventricular hemorrhage and intraventricularCMs had a higher tendency for rebleeding thanCMs in other locations (8, 9). It is expected that ourcase showing intraventricular hemorrhage wouldalso have a higher tendency to rebleed ; therefore,we selected surgical removal.Typical CT findings of intraventricular CM includemoderately hyperdense intraventricular nodular le-sions showing mild contrast enhancement and mod-erate signs of a mass effect (10). The CT findings in9 cases of trigonal CM that reported CT density in-dicated that the lesion was hyperdense. Typical find-ings of CMs on MRI are : central area of hypersig-nal, corresponding to the presence of methemoglo-bin, associated with areas of hyposignal due to calci-fication and extense fibrosis within the lesion on T1and T2 weighted images ; marked enhancement ;TableCase Year Author Reference Age Sex Symptoms Side Tumor size(cm) Angiography CT scan Surgery Outcome1 1977 Coin et al. 4 36 F Seizures Rt NA normal hyperdensity NA Hemianopia2 1977 Numaguchi et al. 22 43 M Mass lesion Rt NA venous stain andvenous pooling NA NAHemiplegia,hemianopia3 1979 Pau andOrynesu et al. 23 56 F IVH Lt NA not recognizable NA inoperable Dead4 1983 Iwasa et al. 24 8 F Mass lesion Lt 3 NA hyperdensity temporal craniotomy Improvement5 1983 Handa et al. 7 73 M Mass lesion Rt 3 no tumor stain NA lower posteriorparietal lobe incision Improvement6 1986 Yamasaki et al. 25 73 M Mass lesion Rt 3 no feeding artery,tumor stain, draining veinhyperdensity,calcification NA Improvement7 1989 Andoh et al. 16 62 F Mass lesion Lt 3 no revealed homogenousovershadowing hyperdensitymiddle temporalgyrus Dead8 1990 Tatagiba et al. 7 35 M Seizures Rt 4 normal hyperdensity calcarine fissure Improvement9 24 M Mass lesion Lt 3 draining vein hyperdensity transcorticotomy(behind angular gyrus) Improvement10 1993 Miyagi et al. 17 3 F Mass lesion Lt NA no abnormal staining hyperdensity transsylviantransventricular Improvement11 2003 Nieto et al. 26 11 F Seizures Lt 5 NA NA temporal craniotomy Improvement12 2006 Kumar et al. 15 8 M Mass lesion Rt 5 NA brilliantenhancementinferior temporal,superior temporal sulcus Improvement13 19 F Mass lesion Lt 5 no tumor blush orevidence of AVM NAmiddle temporalgyrus Hemiparesis14 20 M Mass lesion Rt 5 no abnormality NA middle temporalgyrus Improvement15 2007 Gonzalez -Darder 27 25 M IVH Rt 3 not pathological hyperdensity trans -sulcustemporal posterior Improvement16 2008 Stavrinou et al. 18 52 F Mass lesion Rt 3 unremarkable NA low postcentraltranscortical Improvement17 2012 Ohbuchi et al. 67 F IVH Lt 1 no vessel malformation hyperdensity middle temporalgyrus ImprovementIVH : intraventricular hemorrhage, NA : not available, F : female, M : male, Rt : right, Lt : leftThe Journal of Medical Investigation Vol. 59 August 2012 277peripheral hyposignal area due to the presence ofhemosiderin ; moderate mass effect compared to thesize of lesion ; and slight perifocal edema (10). Cere-bral angiography rarely shows abnormalities otherthan a nonspecific avascular mass (9, 11, 12, 13, 14).Angiography plays a role in excluding AVM (15).Differential diagnoses are choroid plexus papil-loma, teratoma, astrocytoma, ependymoma, neuro-cytoma, metastasis, meningioma and arteriovenousmalformation (6, 16, 17, 18).The surgical approach for trigonal lesions de-pends on the size of the lesion and whether thelesion is in the dominant hemisphere. We choosethe transtemporal approach to the lesion even indominant hemispheres. This approach has manytheoretical advantages : it is close to the tumor ; theanterior choroidal artery can be managed early (19,20) ; and sufficient decompression and a wide opera-tive field are obtainable because the inferior hornof the lateral ventricle is generally dilated. The dis-advantages are (21) : homonymous hemianopsiamay occur at least slightly, even though the ap-proach is made parallel to the optic radiation ; indi-vidual differences have been noted at the site ofWernicke’s area, so fluent aphasia might occur evenwhen there is no invasion in the posterior 1/3 ofthe superior temporal gyrus on the dominant side ;and tumors that develop on the posterior body arehardly removable (16). In the literature, twelvecases (70%) showed improvement. Our patient in-itially demonstrated mild dyslexia and mild acalcu-lia, which completely disappeared within 3 weeks.DISCLOSUREThere is no COI status to disclose. Hidenori OhbuchiThere is no COI status to disclose. Yasuhiko OsakaThere is no COI status to disclose. Takahiro OgawaThere is no COI status to disclose. Masataka NantoThere is no COI status to disclose. Yoshikazu NakaharaThere is no COI status to disclose. Kanade KatsuraThere is no COI status to disclose. Hiroshi TenjinThere is no COI status to disclose. 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Trigonal cavernous malformation with intraventricular hemorrhage : A case report and literature review

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Trigonal cavernous malformation with intraventricular hemorrhage : A case report and literature review

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