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儿童脑动静脉畸形的放射外科治疗:57例病例系列

Radiosurgery of cerebral arteriovenous malformations in children: a series of 57 cases.

作者信息

Nataf François, Schlienger Michel, Lefkopoulos Dimitri, Merienne Louis, Ghossoub May, Foulquier Jean Noel, Deniaud-Alexandre Elisabeth, Mammar Hamid, Meder Jean-François, Turak Baris, Huart Judith, Touboul Emmanuel, Roux François-Xavier

机构信息

Department of Neurosurgery, Sainte-Anne Hospital, Paris, France.

出版信息

Int J Radiat Oncol Biol Phys. 2003 Sep 1;57(1):184-95. doi: 10.1016/s0360-3016(03)00445-0.

Abstract

PURPOSE

To evaluate the efficacy and outcome of Linac radiosurgery (RS) as treatment of cerebral arteriovenous malformations (cAVM) in a series of 57 children.

METHODS AND MATERIALS

Between 1984 and 2000, we used Linac radiosurgery to treat 792 patients with cAVM. This series included 57 children (7.2%) under the age of 15 years at the time of RS (range = 7-15 years, median = 12 years). We were able to evaluate 49 of the children (86%) by angiography, 21 boys and 28 girls (sex ratio = 0.75). First symptoms were: hemorrhage, 34 patients (69.4%); seizures, 6 patients (12.5%); headache, 6 patients (12.5%); and progressive neurologic deficit, 1 patient (2.1%). Nidus size ranged from 5 to 50 mm (median = 20 mm). Nidus volume ranged from 0.6 to 16 cc (median = 3.5 cc). Patient distribution according to Spetzler-Martin grade was as follows: Grade 1, n = 5 (11%); Grade 2, n = 18 (35%); Grade 3, n = 21 (40%,); Grade 4, n = 5 (14%); and Grade 5, n = 0%. Twenty-seven patients (55.1%) had other treatment before RS: embolization, n = 14 (31.1%); neurosurgery, n = 9 (20.5%); embolization and neurosurgery, n = 3 (6.1%). RS was performed with the system used for adults. Patients were seated in a Betti armchair. Circular 15 MV X-ray minibeams (6 to 20 mm) were delivered in coronal arcs by a GECGR Saturne 43 Linac. Planification and dosimetry were carried out using the Associated Target Methodology and Dosigray TPS dosimetric systems. The dose at the peripheral isodose (50-70%) ranged from 18 to 28 Gy. Median and mean doses were 25 Gy and 23.8 Gy. Mono-isocentric planification was used in 25 patients (53.2%) and multi-isocentric in 24 patients (2 to 5 isocenters). The overall follow-up ranged from 7 to 172 months (mean 40 months, median 34 months).

RESULTS

The overall rate of obliteration (OR) was 30/49 (61.2%). Mean time to obliteration was 34 months (range = 7 to 172 months). OR varied according to nidus size and volume: OR was 80% for nidus <15 mm, 67% for nidus between 15 and 25 mm, and 42% for nidus >25 mm (p = 0.058). OR was 100% for nidus <1 cc, 73% for nidus between 1 and 4 cc, and 40% for nidus of 4 to 10 cc (p = 0.019). OR according to patient gender was 84.2% for boys and 40% for girls. OR according to minimum dose (Dmin) was 44% for Dmin < 15 Gy (p = 0.01), 89% for D min from 15 to 20 Gy, and 100% for Dmin > 20 Gy (p = 0.01). OR was 62% in nonembolized AVM and 58% in previously embolized AVM (NS). OR according to the number of isocenters was 68.2%, 55.6%, 80%, 50%, and 0% for 1, 2, 3, 4, and 5 isocenters, respectively. After multivariate analysis, only Dmin closely correlated with OR (beta = 0.462; SE = 0.244, p = 0.057). Of the 6 patients with seizures before RS, 5 (80%) were seizure-free without medication after RS. One patient died of pneumonia.

MORBIDITY

Four patients (8.2%) had bleeding after RS at 39, 45, 51, and 59 months. No new neurologic deficit was found during the follow-up period. Twenty-nine patients underwent magnetic resonance imaging. Thirteen patients (44.8%) showed no parenchymal changes. Thirteen patients (44.8%) had Grade 2 changes, that is, T2 hypersignals. Two patients (6.9%) had Grade 3 changes, and 1 patient (3.4%) had Grade 4 "necrosis-like" changes.

CONCLUSION

In our experience, Linac RS has proven to be a safe and effective method to treat cerebral AVM in children <15 years, whether used alone or in association with embolization and/or neurosurgery.

摘要

目的

评估直线加速器放射外科(RS)治疗57例儿童脑动静脉畸形(cAVM)的疗效和结果。

方法和材料

1984年至2000年间,我们使用直线加速器放射外科治疗792例cAVM患者。该系列包括57例在接受RS治疗时年龄小于15岁的儿童(7.2%)(范围=7 - 15岁,中位数=12岁)。我们能够通过血管造影评估其中49例儿童(86%),21例男孩和28例女孩(性别比=0.75)。首发症状为:出血,34例患者(69.4%);癫痫发作,6例患者(12.5%);头痛,6例患者(12.5%);进行性神经功能缺损,1例患者(2.1%)。病灶大小范围为5至50毫米(中位数=20毫米)。病灶体积范围为0.6至16立方厘米(中位数=3.5立方厘米)。根据Spetzler - Martin分级的患者分布如下:1级,n = 5(11%);2级,n = 18(35%);3级,n = 21(40%);4级,n = 5(14%);5级,n = 0%。27例患者(55.1%)在接受RS治疗前曾接受其他治疗:栓塞,n = 14(31.1%);神经外科手术,n = 9(20.5%);栓塞和神经外科手术,n = 3(6.1%)。RS采用成人使用的系统进行。患者坐在Betti扶手椅上。通过GECGR Saturne 43直线加速器在冠状弧中输送圆形15 MV X射线微束(6至20毫米)。使用相关靶区方法和Dosigray TPS剂量测定系统进行计划和剂量测定。外周等剂量线(50 - 70%)处的剂量范围为18至28 Gy。中位数和平均剂量分别为25 Gy和23.8 Gy。25例患者(53.2%)采用单等中心计划,24例患者(2至5个等中心)采用多等中心计划。总体随访时间范围为7至172个月(平均40个月,中位数34个月)。

结果

总体闭塞率(OR)为30/49(61.2%)。平均闭塞时间为34个月(范围=7至172个月)。OR根据病灶大小和体积而有所不同:病灶<15毫米时OR为80%,病灶在15至25毫米之间时为67%,病灶>25毫米时为42%(p = 0.058)。病灶<1立方厘米时OR为100%,病灶在1至4立方厘米之间时为73%,病灶为4至10立方厘米时为40%(p = 0.019)。根据患者性别OR分别为男孩84.2%,女孩40%。根据最小剂量(Dmin),Dmin < 15 Gy时OR为44%(p = 0.01),Dmin为15至20 Gy时为89%,Dmin > 20 Gy时为100%(p = 0.01)。未栓塞的AVM的OR为62%,先前栓塞过的AVM的OR为58%(无显著差异)。根据等中心数量,1、2、3、4和5个等中心的OR分别为68.2%、55.6%、80%、50%和0%。多因素分析后,只有Dmin与OR密切相关(β = 0.462;标准误=0.244,p = 0.057)。RS治疗前有癫痫发作的6例患者中,5例(80%)RS治疗后无需药物治疗即无癫痫发作。1例患者死于肺炎。

并发症

4例患者(8.2%)在RS治疗后39、45、51和59个月时发生出血。随访期间未发现新的神经功能缺损。29例患者接受了磁共振成像检查。13例患者(44.8%)未显示实质改变。13例患者(44.8%)有2级改变,即T2高信号。2例患者(6.9%)有3级改变且1例患者(3.4%)有4级“坏死样”改变。

结论

根据我们的经验,直线加速器RS已被证明是治疗15岁以下儿童脑AVM的一种安全有效的方法,无论单独使用还是与栓塞和/或神经外科手术联合使用。

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