Pediatric Cerebral Cavernous Malformations

Paddock, Michael; Lanham, Sarah; Gill, Kanwar R.; Sinha, Saurabh; Connolly, Daniel

doi:10.1016/j.pediatrneurol.2020.11.004

Cited by 16 publications

(13 citation statements)

References 67 publications

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“…Cerebral cavernous malformations are enlarged vascular channels in the brain parenchyma lined by a single layer of endothelial cells. Most are diagnosed with magnetic resonance imaging 1 . Occurrence in children is uncommon, with reported prevalence rates ranging from 0.30% to 0.53%.…”

Section: Discussionmentioning

confidence: 99%

“…Most are diagnosed with magnetic resonance imaging. 1 Occurrence in children is uncommon, with reported prevalence rates ranging from 0.30% to 0.53%. However, 25% of children with a cerebral cavernous malformation will experience epilepsy, hemiplegia, headache, other symptoms, or multiple episodes of acute bleeding, which is associated with high rates of mortality and disability.…”

Section: Discussionmentioning

confidence: 99%

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Cranial ultrasonography in diagnosis of infantile cerebral cavernous malformation

Kaneko

Ikeno

Wada

2023

Clinical Case Reports

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Cranial ultrasonography in diagnosis of infantile cerebral cavernous malformation

Kaneko

Ikeno

Wada

2023

Clinical Case Reports

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“…CMs with lobar locality predominately present with seizures and surgery aims to achieve seizure freedom in case of pharmaco-resistance. Only severe hemorrhage due to a cavernoma necessitates urgent surgical removal [ 21 ]. In our cohort, 72% of patients had seizures at the time of diagnosis, and seizure freedom was achieved in 86% of these patients.…”

Section: Discussionmentioning

confidence: 99%

“…No hemorrhage was seen in any of the conservatively managed patients (median follow-up time: 59.5 months). Although deep location is reported to be a risk factor for hemorrhage, lesion size is likely to be crucial [ 10 , 21 ]. According to a study by Li et al, which included 85 pediatric patients with untreated brainstem CMs, a lesion diameter of > 2 cm is associated with higher hemorrhage risk [ 16 ].…”

Section: Discussionmentioning

confidence: 99%

“…Pediatric cerebral cavernous malformations account for approximately 25% of all cases and are a major cause of brain hemorrhage in children [ 11 , 26 ]. Since the introduction of magnetic resonance imaging (MRI), a rise in the incidence of CMs has been reported, appearing as “mulberry-like” lesions in the brain [ 2 , 10 , 21 ]. The spectrum of the clinical presentation encompasses no symptoms in cases with incidental diagnosis of chronic symptoms in cases with epileptic seizures, or acute severe neurologic impairments caused by hemorrhage [ 9 ].…”

Section: Introductionmentioning

confidence: 99%

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How can we optimize the long-term outcome in children with intracranial cavernous malformations? A single-center experience of 61 cases

et al. 2022

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The objective is to provide a treatment algorithm for pediatric patients with intracranial cavernous malformations (CMs) based on our experience. Patients < 18 years of age who were treated either surgically or conservatively at the authors’ institution between 1982 and 2019 were retrospectively evaluated. A total of 61 pediatric patients were treated at the authors’ institution: 39 with lobar CMs; 18 with deep CMs, including 12 in the brainstem and 6 in the basal ganglia; and 4 with CMs in the cerebellar hemispheres. Forty-two patients underwent surgery, and 19 were treated conservatively. The median follow-up time was 65 months (1–356 months). In surgically treated patients, lesions were larger (2.4 cm vs 0.9 cm, p < 0.001). In patients with lobar CMs, seizures were more common (72% vs 21%, p = 0.003) in the surgery group than in conservatively managed patients. In deep CMs, modified Rankin scale (mRS) was higher (4 vs 1, p = 0.003) in the surgery group than in conservatively treated patients. At the time of last follow-up, no differences in Wieser outcome class I were seen (86% vs 67%) in lobar CMs, and mRS scores had aligned between the treatment groups in deep CMs (1 vs 0). We encountered no new permanent neurological deficit at time of last follow-up. We propose a treatment algorithm according to lesion location and size, burden of symptoms, epilepsy workup, and further clinical course during observation. A conservative management is safe in pediatric patients with asymptomatic CMs. Gross total resection should be the aim in patients with symptomatic lobar CMs. A less aggressive approach with subtotal resection, when required to prevent neurological compromise, sustainably improves neurological outcome in patients with deep CMs.

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Outcomes after surgical and nonsurgical treatment of pediatric cerebral cavernous malformation

Zhang,

Yu,

Mao

et al. 2024

Pediatric Investigation

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ImportancePediatric cerebral cavernous malformation (CCM) is a rarely encountered vascular entity. Comparative study on surgical excision and nonsurgical management outcomes of CCM in pediatrics is limited.ObjectiveTo determine the demographic characteristics, hemorrhage rate, and long‐term outcomes of pediatric patients with CCM.MethodsA retrospective study of pediatric patients with CCM in Chinese PLA General Hospital was conducted between January 2004 and January 2019. We compared the clinical characteristics, radiological features, and outcomes of the surgical and nonsurgical groups.ResultsSeventy‐nine children were included, with 69.6% being boys, and the average age was 11.8 ± 5.5 years. The annual retrospective hemorrhagic rate was 5.7% per patient per year. Fifty‐six children (70.9%) underwent surgical excision, and they were more likely to present with seizure symptoms (P = 0.011), have a higher proportion of larger lesion size (P = 0.008), less likely to have durations ≤10 days (P = 0.048), and less likely to have supratentorial deep CCM (P = 0.014) compared to children who received nonsurgical management. Total resection was achieved in most surgical cases (55, 98.2%). During the 143.9 ± 50.8 months of follow‐up, 44 patients (78.6%) achieved improvement, 12 patients (17.8%) remained the same, and two (3.6%) experienced worsening. In the nonsurgical management group, 14 children (60.9%) experienced symptom improvement, eight (34.8%) remained the same, and one (4.3%) worsened, with a re‐hemorrhagic risk of 8.7%.InterpretationSurgical removal of pediatric CCM can eliminate the risk of hemorrhage and lead to satisfactory outcomes. For children undergoing nonsurgical management, long‐term close monitoring is essential due to the life‐long risk of hemorrhage.

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Pediatric Cerebral Cavernous Malformations

Cited by 16 publications

References 67 publications

Cranial ultrasonography in diagnosis of infantile cerebral cavernous malformation

Cranial ultrasonography in diagnosis of infantile cerebral cavernous malformation

How can we optimize the long-term outcome in children with intracranial cavernous malformations? A single-center experience of 61 cases

Outcomes after surgical and nonsurgical treatment of pediatric cerebral cavernous malformation

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