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儿童眼眶淋巴管畸形的图像引导下经皮博来霉素和贝伐单抗硬化治疗

Image-Guided Percutaneous Bleomycin and Bevacizumab Sclerotherapy of Orbital Lymphatic Malformations in Children.

作者信息

Abdelaziz Omar, Hassan Farouk, Elessawy Kareem, Emad-Eldin Sally, Essawy Rania El

机构信息

Department of Diagnostic and Interventional Radiology, Cairo University Hospitals, Cairo, Egypt.

Department of Ophthalmology, Cairo University Hospitals, Cairo, Egypt.

出版信息

Cardiovasc Intervent Radiol. 2019 Mar;42(3):433-440. doi: 10.1007/s00270-018-2128-4. Epub 2018 Nov 28.

Abstract

PURPOSE

To evaluate the effectiveness and safety of image-guided percutaneous sclerotherapy using bleomycin for macrocystic and bevacizumab (Avastin™) for microcystic orbital lymphatic malformations in children.

MATERIALS AND METHODS

Between October 2015 and July 2018, we prospectively evaluated 10 pediatric patients who presented clinically and radiologically with lymphatic malformations and were treated with percutaneous sclerotherapy. Patients with venous malformations were excluded. Eight females and two males with ages ranging from 3 to 17 years (mean: 8.8, SD: 4.9) were included. Guided with ultrasound and fluoroscopy, macrocysts were treated with bleomycin instillation. For microcystic components in three patients, bevacizumab was injected intralesional. All patients underwent ultrasound and non-contrast MRI to evaluate response to treatment after 6 weeks.

RESULTS

The malformations were macrocystic in seven patients and complex (macro/microcystic) in three. Twenty sclerotherapy sessions were performed, (range: 1-3 sessions, mean: 2, SD: 0.8). Clinically, there was a significant reduction in the proptosis after treatment (P = 0.007) and dystopia (P = 0.018). The local radiological response showed a reduction in the maximum lesions diameters and volumes after treatment (P = 0.005 and 0.005, respectively). Two of the three patients treated with bevacizumab showed a reduction in the lesions volumes by 90.4% and 63.4%, respectively, whereas one patient did not show volume reduction. Transient periorbital edema and ecchymosis occurred following the procedure with no major complications encountered. Follow-up ranged from 9-33 months, mean: 20.3, SD: 7.4.

CONCLUSION

Bleomycin sclerotherapy is a safe and effective treatment for orbital macrocystic lymphatic malformations. Further use of bevacizumab for microcystic lesions in a larger series is required to outline its efficacy and safety.

摘要

目的

评估使用博来霉素进行影像引导下经皮硬化治疗儿童眼眶大囊型淋巴管畸形以及使用贝伐单抗(阿瓦斯汀™)治疗微囊型淋巴管畸形的有效性和安全性。

材料与方法

2015年10月至2018年7月期间,我们对10例临床和影像学表现为淋巴管畸形并接受经皮硬化治疗的儿科患者进行了前瞻性评估。排除静脉畸形患者。纳入8名女性和2名男性,年龄范围为3至17岁(平均:8.8岁,标准差:4.9岁)。在超声和荧光透视引导下,对大囊肿进行博来霉素注射治疗。对于3例患者的微囊成分,将贝伐单抗注射到病变内。所有患者在6周后接受超声和非增强MRI检查以评估治疗反应。

结果

7例患者的畸形为大囊型,3例为复杂型(大囊/微囊型)。共进行了20次硬化治疗(范围:1 - 3次,平均:2次,标准差:0.8次)。临床上,治疗后眼球突出(P = 0.007)和眼球异位(P = 0.018)明显减轻。局部放射学反应显示治疗后最大病变直径和体积减小(分别为P = 0.005和0.005)。接受贝伐单抗治疗的3例患者中有2例病变体积分别减小了90.4%和63.4%,而1例患者病变体积未减小。治疗后出现短暂的眶周水肿和瘀斑,未发生重大并发症。随访时间为9 - 33个月,平均:20.3个月,标准差:7.4个月。

结论

博来霉素硬化治疗是眼眶大囊型淋巴管畸形的一种安全有效的治疗方法。需要在更大规模的系列研究中进一步使用贝伐单抗治疗微囊型病变,以明确其疗效和安全性。

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