Cerebral cavernous malformation presenting in childhood: a single-centered surgical experience of 29 cases

Aslan, Ayfer; Börcek, Alp Özgün; Demirci, Harun; Erdem, Mehmet

doi:10.1016/j.clineuro.2020.105830

Cited by 10 publications

(7 citation statements)

References 30 publications

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“…Additionally, we observed a (re)hemorrhage risk per year per patient of 7.1% and 6.2% in patients with BSCM and familial history of CCM, respectively, compared with 4.1% in the entire cohort and 0.4% in asymptomatic patients. While some small trials on pediatric populations suggest higher risk of hemorrhage compared with adults, 4,14–16 we found equal risks independent of age.…”

Section: Discussioncontrasting

confidence: 64%

“…In addition, existing studies are limited by short follow-up periods, small sample sizes or the inclusion of patients of up to 25 years of age. 2,8,11,15,16,23,27 There also tend to be divergent conclusions, whereby some state higher bleeding rates in children compared with adults and report the presence of DVA as a risk factor for recurrent hemorrhage, whereas others observed equal rates. 1,4,11,14,19,23 Our study analyzed a set of risk factors for (re)hemorrhage in pediatric CCM patients.…”

Section: Discussionmentioning

confidence: 99%

“…2022;53:817-824. DOI: 10.1161/STROKEAHA.121.035338 adults, 4,[14][15][16] whose estimated overall cumulative 5-year risk of symptomatic ICH is ≈20%. 6,[17][18][19] This risk may be further influenced by factors, such as sex, CCM location, and history of previous ICH.…”

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confidence: 99%

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Natural Course of Cerebral Cavernous Malformations in Children: A Five-Year Follow-Up Study

Santos

Rauschenbach

Saban

et al. 2022

Stroke

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Background and Purpose: The purpose of this study was to investigate the natural course of cerebral cavernous malformations (CCM) in the pediatric population, with special emphasis on the risk of first and recurrent bleeding over a 5-year period. Methods: Our institutional database was screened for patients with CCM treated between 2003 and 2020. Patients ≤18 years of age with complete magnetic resonance imaging data set, clinical baseline characteristics, and ≥1 follow-up examination were included. Surgically treated individuals were censored after CCM removal. We assessed the impact of various parameters on first or recurrent intracerebral hemorrhage (ICH) at diagnosis using univariate and multivariate logistic regression adjusted for age and sex. Kaplan-Meier and Cox regression analyses were performed to determine the cumulative 5-year risk for (re)hemorrhage. Results: One hundred twenty-nine pediatric patients with CCM were analyzed. Univariate logistic regression identified brain stem CCM (odds ratio, 3.15 [95% CI, 1.15−8.63], P =0.026) and familial history of CCM (odds ratio, 2.47 [95% CI, 1.04−5.86], P= 0.041) as statistically significant predictors of ICH at diagnosis. Multivariate logistic regression confirmed this correlation (odds ratio, 3.62 [95% CI, 1.18−8.99], P= 0.022 and odds ratio, 2.53 [95% CI, 1.07−5.98], P =0.035, respectively). Cox regression analysis identified ICH as mode of presentation (hazard ratio, 14.01 [95% CI, 1.80−110.39], P= 0.012) as an independent predictor for rehemorrhage during the 5-year follow-up. The cumulative 5-year risk of (re)bleeding was 15.9% (95% CI, 10.2%−23.6%) for the entire cohort, 30.2% (20.2%−42.3%) for pediatric patients with ICH at diagnosis, and 29.5% (95% CI, 13.9%−51.1%) for children with brain stem CCM. Conclusions: Pediatric patients with brain stem CCM and familial history of CCM have a higher risk of ICH as mode of presentation. During untreated 5-year follow-up, they revealed a similar risk of (re)hemorrhage compared to adult patients. The probability of (re)bleeding increases over time, especially in cases with ICH at presentation or brain stem localization.

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Section: Discussioncontrasting

confidence: 64%

Section: Discussionmentioning

confidence: 99%

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Natural Course of Cerebral Cavernous Malformations in Children: A Five-Year Follow-Up Study

Santos

Rauschenbach

Saban

et al. 2022

Stroke

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“…Ten studies were identified after screening, involving 216 pediatric patients ( 4 , 9 – 17 ). All 10 studies were published between 1995 and 2020.…”

Section: Resultsmentioning

confidence: 99%

“…If the two researchers cannot reach an agreement, a senior researcher (Xiaofan Jiang) will be consulted. Finally, 10 out of 1,586 articles met the inclusion criteria ( 4 , 9 – 17 ). The two researchers extracted the following data from the 10 studies: publication date, first author’s last name, total number of patients, number of female patients, mean age at surgery, mean duration of follow-up, mean duration of seizure, number of patients with drug-resistant epilepsy, type of epilepsy, number of patients with multiple CCMs, lesion location, postoperative seizure outcome, case fatality, and temporary and permanent symptomatic adverse effects ( Table 1 ).…”

Section: Methodsmentioning

confidence: 99%

A systematic review and meta-analysis of surgeries performed for cerebral cavernous malformation-related epilepsy in pediatric patients

et al. 2022

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BackgroundThe clinical benefit of surgery for the treatment of cerebral cavernous malformation (CCM)-related epilepsy in pediatric patients is still controversial. Although surgical treatment of CCM-related epilepsy in children is widely recognized, the clinical benefits of controlling the seizure rate must be balanced against the risk of leading to perioperative morbidity.MethodsWe conducted a comprehensive search to identify relevant studies via Ovid Medline, Web of Science and PubMed (January 1995–June 2020). The following search terms were used: “hemangioma, cavernous, central nervous system,” “brain cavernous hemangioma,” “cerebral cavernous hemangioma,” “CCM,” “epilepsy,” and “seizures.” The seizure control rate and the risk of postoperative adverse outcomes along with their 95% confidence intervals (CIs) were calculated.ResultsA total of 216 patients across 10 studies were included in meta-analysis. The results showed that the control rate of epilepsy was 88% (95% CI: 76–95%). Four percent (95% CI: 2–10%) of the patients experienced temporary symptomatic adverse effects following surgical resection, and 3% (95% CI: 0–26%) of the patients developed permanent symptomatic adverse effects in the long-term follow-up after surgical excision of the CCMs. None of the patients died as a result of the CCMs or surgical treatment.ConclusionSurgery is an effective and safe treatment for CCM –related epilepsy in pediatric patients with a low risk of postoperative complications and death.

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Cavernous Malformations

Karsy

Schmidt

Bollo

2022

Pediatric Neurosurgery for Clinicians

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Cerebral cavernous malformation presenting in childhood: a single-centered surgical experience of 29 cases

Cited by 10 publications

References 30 publications

Natural Course of Cerebral Cavernous Malformations in Children: A Five-Year Follow-Up Study

Natural Course of Cerebral Cavernous Malformations in Children: A Five-Year Follow-Up Study

A systematic review and meta-analysis of surgeries performed for cerebral cavernous malformation-related epilepsy in pediatric patients

Cavernous Malformations

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