Management of patients with cavernous angiomas presenting epileptic seizures

Noto, Sadami; Fujii, Masami; Akimura, Tatsuo; Imoto, Hirochika; Nomura, Sadahiro; Kajiwara, Koji; Kato, Shoichi; Fujisawa, Hironori; Suzuki, Michiyasu

doi:10.1016/j.surneu.2005.03.045

Cited by 42 publications

(24 citation statements)

References 15 publications

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“…In all recent pediatric series involving patients with cerebral CMs, children with long-standing seizures were rare [168], because when lesions appear clinically, a curative and definitive therapy is usually considered. The early surgical removal of superficial or not critically located CMs causing seizures may offer real curative treatment of epilepsy, preventing psychosocial disability in patients on long-term medical therapy and avoiding the risk of neurological deficits from growth and hemorrhage of the cavernomas [169, 170]. Surgery may also improve the efficiency of anticonvulsant therapy in patients with epilepsy who are resistant to medication [171,172,173].…”

Section: Resultsmentioning

confidence: 99%

Cavernous Malformations of the Central Nervous System in the Pediatric Age Group

et al. 2009

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Objective: The main clinico-diagnostic features, risk factors and associated diseases of cavernous malformations (CMs), also called cavernous angiomas or cavernomas, of the central nervous system (CNS) in children are described, and the most relevant differences compared to the affected adult population are pointed out, focusing on the management of pediatric patients harboring cranial and spinal CMs. Materials: This was a retrospective study of a series of 42 children symptomatic for CMs of the cranial and spinal compartments (35 supratentorial brain lesions, 5 infratentorial and 2 in the spinal region) operated on between 1975 and 2005, with a clinical follow-up ranging from 12 to 192 months. The results were compared with those found in the most recent literature dealing with pediatric CMs of the CNS. Results: Surgical treatment produced excellent or good results in 69% of our 42 children. Unchanged neurological deficits were observed in 23.8% of cases, while morbidity from surgical procedures was 7.14%. Mortality was absent in this series. These surgical results are comparable with those found in the literature, where morbidity and mortality rates from surgery are 8.8 and 1.13%, respectively, and are mostly associated with procedures for the excision of deep, critically located cavernomas. Conclusion: CMs represent the most common CNS vascular lesion in children, although their incidence is 4 times lower than that of the adult population. The natural history of pediatric CMs throughout the neuraxis seems to be more aggressive than in adult patients; these lesions have higher rates of growth and hemorrhage, larger dimensions and often atypical radiological pictures at diagnosis. Beside the familial form of the disease, which is more often associated with multiple lesions and an earlier age of clinical presentation, the major risk factor for CMs in children seems to be radiotherapy for CNS tumors. Furthermore, a greater number of CMs coexistent with mixed angiomatous lesions have been reported in children than in adults. Surgical results are related to the preoperative neurological status of the children; symptomatic patients who are operated on early, before they develop severe neurological deficits or long-standing seizures, may achieve the best clinical outcome. Radiosurgery does not seem to be advisable in children as an alternative treatment for deep CMs or those causing epilepsy.

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Section: Resultsmentioning

confidence: 99%

Cavernous Malformations of the Central Nervous System in the Pediatric Age Group

et al. 2009

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“…patients, which found a beneficial effect of CCM excision on seizure freedom 12 (that almost reached the threshold for a statistically significant and "dramatic" effect in an observational study 13 ), the other 10 studies did not show clinically or statistically significant differences in death or functional outcome from CCM excision. Therefore, we compared clinical outcomes after CCM excision and conservative management in an observational study.…”

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confidence: 92%

“…First, we updated our systematic review 11 of studies involving more than 20 adults, which compared treatment with neurosurgery to conservative management in a concurrent or historical control group and reported objective clinical outcomes. We found 5 comparative studies involving 205 patients with brainstem CCM that had caused ICH/FND 24-28 and 9 studies involving 278 patients with CCM that had caused seizures, 13,[29][30][31][32][33][34][35][36] which did not demonstrate dramatic beneficial or harmful effects on death or functional outcome, 12 apart from one study that found a beneficial effect of CCM excision on seizure freedom, although we judged it to be at high risk of bias. 13 Second, we quantified the risk of adverse events after CCM excision using the largest published case series from individual institutions, which included more than 20 adults who underwent excision of CCMs that were diagnosed by MRI or pathologic examination, and which described both patients' clinical presentation as well as objective clinical outcomes, stratified by CCM location.…”

mentioning

confidence: 99%

“…We found 5 comparative studies involving 205 patients with brainstem CCM that had caused ICH/FND 24-28 and 9 studies involving 278 patients with CCM that had caused seizures, 13,[29][30][31][32][33][34][35][36] which did not demonstrate dramatic beneficial or harmful effects on death or functional outcome, 12 apart from one study that found a beneficial effect of CCM excision on seizure freedom, although we judged it to be at high risk of bias. 13 Second, we quantified the risk of adverse events after CCM excision using the largest published case series from individual institutions, which included more than 20 adults who underwent excision of CCMs that were diagnosed by MRI or pathologic examination, and which described both patients' clinical presentation as well as objective clinical outcomes, stratified by CCM location. We included 26 case series (figure e-4, table e-1, table e-2, e-references) reporting the outcome after CCM excision from 1,839 adults over average postoperative follow-up ranging from Figure 3 Postoperative adverse events in published studies of predominantly supratentorial or exclusively brainstem cerebral cavernous malformation Meta-analysis of proportions of patients with postoperative adverse events (death within 30 days, symptomatic intracranial hemorrhage (ICH), or new or worsened focal neurologic deficit) in studies of predominantly supratentorial or exclusively brainstem cerebral cavernous malformation (CCM).…”

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confidence: 99%

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Outcome after surgical or conservative management of cerebral cavernous malformations

Moultrie¹,

Horne²,

Josephson³

et al. 2014

Neurology

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Objective: There have been few comparative studies of microsurgical excision vs conservative management of cerebral cavernous malformations (CCM) and none of them has reliably demonstrated a statistically and clinically significant difference. Methods:We conducted a prospective, population-based study to identify and independently validate definite CCM diagnoses first made in 1999-2003 in Scottish adult residents. We used multiple sources of prospective follow-up to assess adults' dependence and to identify and independently validate outcome events. We used univariate and multivariable survival analyses to test the influence of CCM excision on outcome, adjusted for prognostic factors and baseline imbalances.Results: Of 134 adults, 25 underwent CCM excision; these adults were younger (34 vs 43 years at diagnosis, p 5 0.004) and more likely to present with symptomatic intracranial hemorrhage or focal neurologic deficit than adults managed conservatively (48% vs 26%; odds ratio 2.7, 95% confidence interval [CI] 1.1-6.5). During 5 years of follow-up, CCM excision was associated with a deterioration to an Oxford Handicap Scale score 2-6 sustained over at least 2 successive years (adjusted hazard ratio [HR] 2.2, 95% CI 1.1-4.3) and the occurrence of symptomatic intracranial hemorrhage or new focal neurologic deficit (adjusted HR 3.6, 95% CI 1.3-10.0).Conclusions: CCM excision was associated with worse outcomes over 5 years compared to conservative management. Long-term follow-up will determine whether this difference is sustained over patients' lifetimes. Meanwhile, a randomized controlled trial appears justified. Classification of evidence:This study provides Class III evidence that CCM excision worsens shortterm disability scores and increases the risk of symptomatic intracranial hemorrhage and new focal neurologic deficits.

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“…Epilepsy in the setting of a lesion is usually resistant to medical management [18,32]. Unfortunately, achieving seizure control is underappreciated in the medical community.…”

Section: Discussionmentioning

confidence: 99%

Temporal lobe epilepsy and cavernous malformations: surgical strategies and long-term outcomes

et al. 2015

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Patients with temporal lobe CCM should be offered resection for durable seizure control, prevention of secondary epileptogenic foci, and elimination of hemorrhage risk. The preoperative work-up should follow a team approach. Surgical intervention should include complete lesionectomy in all cases. Intra or extra-operative ECoG for neocortical lesions may be beneficial. Management of mesial temporal CCMs (archicortex) should consider resection of a well-defined epileptogenic zone (including mesial structures) due to high probability of pathologically proven MTS. The use of this treatment algorithm is useful for the education and treatment of these patients.

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Management of patients with cavernous angiomas presenting epileptic seizures

Cited by 42 publications

References 15 publications

Cavernous Malformations of the Central Nervous System in the Pediatric Age Group

Cavernous Malformations of the Central Nervous System in the Pediatric Age Group

Outcome after surgical or conservative management of cerebral cavernous malformations

Temporal lobe epilepsy and cavernous malformations: surgical strategies and long-term outcomes

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