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Pediatric cerebral cavernous malformations and stereotactic radiosurgery: an analysis of 50 cases from a multicentric study

G. Mantziaris, C. Dumot, S. Pikis, S. Peker, Y. Samanci, GD. Ardor, AM. Nabeel, WA. Reda, SR. Tawadros, K. Abdel Karim, AMN. El-Shehaby, RM. Emad Eldin, AH. Elazzazi, D. Sheehan, K. Sheehan, N. Martínez Moreno, R. Martínez Álvarez, R. Liscak, J....

. 2024 ; 33 (4) : 315-322. [pub] 20240105

Jazyk angličtina Země Spojené státy americké

Typ dokumentu multicentrická studie, časopisecké články

Perzistentní odkaz   https://www.medvik.cz/link/bmc24014496

OBJECTIVE: Cerebral cavernous malformations (CCMs) are the second most common vascular anomaly affecting the CNS in children. Although stereotactic radiosurgery (SRS) has been proposed as an alternative to microsurgery in the management of selected cases in adults, there is a paucity of studies focusing on pediatric patients. The aim of this study was to present the outcomes and associated risks of SRS in this subgroup of patients. METHODS: This retrospective multicenter study included pediatric patients treated with single-session SRS for CCMs. The annual hemorrhage rate (AHR) was calculated before and after SRS in hemorrhagic lesions. The Engel classification was used to describe post-SRS epileptic control. Adverse radiation effects (AREs) and the occurrence of new neurological deficits were recorded. RESULTS: The study included 50 patients (median age 15.1 [IQR 5.6] years) harboring 62 CCMs. Forty-two (84%) and 22 (44%) patients had a history of hemorrhage or epilepsy prior to SRS, respectively. The AHR from diagnosis to SRS excluding the first hemorrhage was 7.19 per 100 CCM-years, dropping to 3.15 per 100 CCM-years after treatment. The cumulative risk of first hemorrhage after SRS was 7.4% (95% CI 0%-14.3%) at 5 years and 23.6% (95% CI 0%-42.2%) at 10 years. Eight hemorrhagic events involving 6 CCMs in 6 patients were recorded in the post-SRS follow-up period; 4 patients presented with transient symptoms and 4 with permanent symptoms. Of the 22 patients with pre-SRS seizures, 11 were seizure free at the last follow-up (Engel class I), 6 experienced improvement (Engel class II or III), 5 had no improvement (Engel class IVA or IVB), and 1 experienced worsening (Engel class IVC). Radiographic AREs were documented in 14.5% (9/62) of CCMs, with 4 being symptomatic. CONCLUSIONS: Single-session SRS reduces the CCM hemorrhage rate in the pediatric population and provides adequate seizure control.

Citace poskytuje Crossref.org

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$a Pediatric cerebral cavernous malformations and stereotactic radiosurgery: an analysis of 50 cases from a multicentric study / $c G. Mantziaris, C. Dumot, S. Pikis, S. Peker, Y. Samanci, GD. Ardor, AM. Nabeel, WA. Reda, SR. Tawadros, K. Abdel Karim, AMN. El-Shehaby, RM. Emad Eldin, AH. Elazzazi, D. Sheehan, K. Sheehan, N. Martínez Moreno, R. Martínez Álvarez, R. Liscak, J. May, M. Tripathi, A. Rajput, N. Kumar, R. Kaur, JD. Alzate, D. Kondziolka, S. Dayawansa, JP. Sheehan
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$a OBJECTIVE: Cerebral cavernous malformations (CCMs) are the second most common vascular anomaly affecting the CNS in children. Although stereotactic radiosurgery (SRS) has been proposed as an alternative to microsurgery in the management of selected cases in adults, there is a paucity of studies focusing on pediatric patients. The aim of this study was to present the outcomes and associated risks of SRS in this subgroup of patients. METHODS: This retrospective multicenter study included pediatric patients treated with single-session SRS for CCMs. The annual hemorrhage rate (AHR) was calculated before and after SRS in hemorrhagic lesions. The Engel classification was used to describe post-SRS epileptic control. Adverse radiation effects (AREs) and the occurrence of new neurological deficits were recorded. RESULTS: The study included 50 patients (median age 15.1 [IQR 5.6] years) harboring 62 CCMs. Forty-two (84%) and 22 (44%) patients had a history of hemorrhage or epilepsy prior to SRS, respectively. The AHR from diagnosis to SRS excluding the first hemorrhage was 7.19 per 100 CCM-years, dropping to 3.15 per 100 CCM-years after treatment. The cumulative risk of first hemorrhage after SRS was 7.4% (95% CI 0%-14.3%) at 5 years and 23.6% (95% CI 0%-42.2%) at 10 years. Eight hemorrhagic events involving 6 CCMs in 6 patients were recorded in the post-SRS follow-up period; 4 patients presented with transient symptoms and 4 with permanent symptoms. Of the 22 patients with pre-SRS seizures, 11 were seizure free at the last follow-up (Engel class I), 6 experienced improvement (Engel class II or III), 5 had no improvement (Engel class IVA or IVB), and 1 experienced worsening (Engel class IVC). Radiographic AREs were documented in 14.5% (9/62) of CCMs, with 4 being symptomatic. CONCLUSIONS: Single-session SRS reduces the CCM hemorrhage rate in the pediatric population and provides adequate seizure control.
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$a Dumot, Chloe $u 1Department of Neurological Surgery, University of Virginia, Charlottesville, Virginia $u 2Department of Neurological Surgery, Hospices Civils de Lyon, France
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$a Pikis, Stylianos $u 1Department of Neurological Surgery, University of Virginia, Charlottesville, Virginia
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$a El-Shehaby, Amr M N $u 4Gamma Knife Center Cairo, Nasser Institute Hospital, Cairo, Egypt $u Departments of6Neurosurgery and
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