ArticleNeuroradiology2023
Natural history of familial cerebral cavernous malformation syndrome in children: a multicenter cohort study.
Article in Neuroradiology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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Who cites it
9 citing papers in PubMed, 11 citations in OpenAlex.
- Pediatric Familial Cerebral Cavernous Malformation Associated With a Novel KRIT1 Initiation-Region Frameshift Variant.Molecular genetics & genomic medicine · 2026Article
- The skin as the mirror of the soul: familial multiple cavernous malformation syndrome in an infant with a ruptured intracranial cavernoma and multiple skin lesions.Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery · 2026Article
- Familial Cerebral Cavernous Malformations: Pathophysiology, Genetics, Biomarkers, and Treatment Perspectives.Journal of neurochemistry · 2026Review
- Familial Cerebral Cavernous Malformations : A Clinical Series and Literature Review.Journal of Korean Neurosurgical Society · 2025Article
- Resection of multiple intramedullary spinal cord cavernous malformations in infancy: illustrative case.Journal of neurosurgery. Case lessons · 2025Article
- Prevalence, genetic and clinical characteristics in first-degree relatives of patients with familial cerebral cavernous malformations in China.Stroke and vascular neurology · 2025Article
- Review
- Outcomes after surgical and nonsurgical treatment of pediatric cerebral cavernous malformation.Pediatric investigation · 2024Article
- The Brainstem Cavernoma Case Series: A Formula for Surgery and Surgical Technique.Medicina (Kaunas, Lithuania) · 2023Article
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Authors and funding
16 authors at 7 institutions in 4 countries.
Funding
Abstract
purposeThere is limited data concerning neuroimaging findings and longitudinal evaluation of familial cerebral cavernous malformations (FCCM) in children. Our aim was to study the natural history of pediatric FCCM, with an emphasis on symptomatic hemorrhagic events and associated clinical and imaging risk factors.
methodsWe retrospectively reviewed all children diagnosed with FCCM in four tertiary pediatric hospitals between January 2010 and March 2022. Subjects with first available brain MRI and [Formula: see text] 3 months of clinical follow-up were included. Neuroimaging studies were reviewed, and clinical data collected. Annual symptomatic hemorrhage risk rates and cumulative risks were calculated using survival analysis and predictors of symptomatic hemorrhagic identified using regression analysis.
resultsForty-one children (53.7% males) were included, of whom 15 (36.3%) presenting with symptomatic hemorrhage. Seven symptomatic hemorrhages occurred during 140.5 person-years of follow-up, yielding a 5-year annual hemorrhage rate of 5.0% per person-year. The 1-, 2-, and 5-year cumulative risks of symptomatic hemorrhage were 7.3%, 14.6%, and 17.1%, respectively. The latter was higher in children with prior symptomatic hemorrhage (33.3%), CCM2 genotype (33.3%), and positive family history (20.7%). Number of brainstem (adjusted hazard ratio [HR] = 1.37, P = 0.005) and posterior fossa (adjusted HR = 1.64, P = 0.004) CCM at first brain MRI were significant independent predictors of prospective symptomatic hemorrhage.
conclusionThe 5-year annual and cumulative symptomatic hemorrhagic risk in our pediatric FCCM cohort equals the overall risk described in children and adults with all types of CCM. Imaging features at first brain MRI may help to predict potential symptomatic hemorrhage at 5-year follow-up.
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