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

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作者
Mantziaris, Georgios [1 ]
Dumot, Chloe [1 ,2 ]
Pikis, Stylianos [1 ]
Peker, Selcuk [3 ]
Samanci, Yavuz [3 ]
Ardor, Gokce D. [3 ]
Nabeel, Ahmed M. [4 ,5 ]
Reda, Wael A. [4 ,6 ]
Tawadros, Sameh R. [4 ,6 ]
Karim, Khaled Abdel [4 ,7 ]
El-Shehaby, Amr M. N. [4 ,6 ]
Eldin, Reem M. Emad [4 ,8 ]
Elazzazi, Ahmed H. [9 ]
Sheehan, Darrah [1 ]
Sheehan, Kimball [1 ]
Moreno, Nuria Martinez [10 ]
Alvarez, Roberto Martinez [10 ]
Liscak, Roman [11 ]
May, Jaromir [11 ]
Tripathi, Manjul [6 ,12 ]
Rajput, Akshay [6 ,12 ]
Kumar, Narendra [13 ]
Kaur, Rupinder [6 ,12 ]
Alzate, Juan Diego [14 ]
Kondziolka, Douglas [14 ]
Dayawansa, Sam [1 ]
Sheehan, Jason P. [1 ]
机构
[1] Univ Virginia, Dept Neurol Surg, Charlottesville, VA USA
[2] Hosp Civils Lyon, Dept Neurol Surg, Lyon, France
[3] Koc Univ, Sch Med, Dept Neurosurg, Istanbul, Turkiye
[4] Nasser Inst Hosp, Gamma Knife Ctr Cairo, Cairo, Egypt
[5] Benha Univ, Fac Med, Neurosurg Dept, Qalubya, Egypt
[6] Ain Shams Univ, Dept Neurosurg, Cairo, Egypt
[7] Ain Shams Univ, Dept Clin Oncol, Cairo, Egypt
[8] Cairo Univ, Natl Canc Inst, Dept Radiat Oncol, Cairo, Egypt
[9] Ain Shams Univ, Extended Modular Program, Fac Med, Cairo, Egypt
[10] Ruber Int Hosp, Dept Radiosurg, Madrid, Spain
[11] Na Homolce Hosp, Dept Stereotact & Radiat Neurosurg, Prague, Czech Republic
[12] Postgrad Inst Med Educ & Res, Dept Neurosurg, Chandigarh, India
[13] Postgrad Inst Med Educ & Res, Dept Radiat Therapy, Chandigarh, India
[14] NYU Langone, Dept Neurosurg, New York, NY USA
关键词
pediatric; cerebral cavernous malformation; stereotactic radiosurgery; vascular disorders; NATURAL-HISTORY; HEMORRHAGE; GUIDELINES; ANGIOMAS; RISK;
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中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
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 classifi - cation 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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