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低剂量平阳霉素眼眶淋巴管畸形和淋巴管静脉畸形注射治疗:多中心病例系列研究。

Low-dose Bleomycin Injections for Orbital Lymphatic and Lymphatic-Venous Malformations: A Multicentric Case Series Study.

机构信息

Goldschleger Eye Institute, Sheba Medical Center, Tel Hashomer, Israel.

Division of Ophthalmology, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.

出版信息

Ophthalmic Plast Reconstr Surg. 2021;37(4):361-365. doi: 10.1097/IOP.0000000000001870.

Abstract

PURPOSE

Orbital lymphatic malformations (LM) are associated with ocular morbidity and facial disfigurement. Surgery is challenging and may not be effective. We describe the outcome of bleomycin injections for venous LM and lymphatic-venous malformation (LVM) malformations of the orbit in 5 tertiary referral centers between January 2010 and December 2018.

METHODS

Multicenter retrospective case series, 5 oculoplastic referral centers: Sheba and Rabin Medical Centers, Israel; Mulago Hospital, Uganda; Sri Sankaradeva Nethralaya, India; and Clinique Ophtalmologique de Tunis, Tunisia. All patients diagnosed with orbital LM/LVM were assigned to successive (range 1-6) intralesional 5 international units bleomycin injections. They all underwent complete ophthalmic and orbital evaluations, orbital imaging, and ancillary testing as needed. Clinical photographs were assessed pre- and posttreatment along with objective assessments of clinical improvement. Additional injections were provided in cases of incomplete response.

RESULTS

A total of 21 patients (17 women, mean ± standard deviation age 18 ± 13 years, range 2-48 years) underwent bleomycin injections. The mean injection dose was 12 ± 10 international units in 1-3 injections. There was a dramatic improvement in lesion size, appearance, proptosis, and ocular motility in 20/21 patients (95%) after a mean follow-up of 18 months. Visual acuity slightly improved after treatment (20/50-20/30; P = 0.076). No side effects were noted after bleomycin injections.

CONCLUSIONS

Bleomycin injections for LM/LVM of the orbit are effective; local or systemic side effects were not seen in this series. To the best of our knowledge, this is the largest reported series of this treatment.

摘要

目的

眼眶淋巴管畸形(LM)与眼部发病率和面部畸形有关。手术具有挑战性,并且可能效果不佳。我们描述了在 2010 年 1 月至 2018 年 12 月期间,5 个三级转诊中心对静脉性 LM 和淋巴管静脉畸形(LVM)的眼眶病变进行博来霉素注射治疗的结果。

方法

多中心回顾性病例系列研究,涉及 5 个眼整形转诊中心:以色列的 Sheba 和 Rabin 医疗中心、乌干达的 Mulago 医院、印度的 Sri Sankaradeva Nethralaya 以及突尼斯的 Clinique Ophtalmologique de Tunis。所有诊断为眼眶 LM/LVM 的患者均接受了连续(范围 1-6)次病灶内 5 国际单位博来霉素注射治疗。所有患者均接受了完整的眼科和眼眶评估、眼眶成像以及根据需要进行的辅助检查。治疗前后评估了临床照片以及临床改善的客观评估。对于反应不完全的病例提供了额外的注射。

结果

共有 21 名患者(17 名女性,平均年龄为 18 ± 13 岁,范围为 2-48 岁)接受了博来霉素注射治疗。平均注射剂量为 12 ± 10 国际单位,分 1-3 次注射。21 例患者(95%)在平均 18 个月的随访后,病变大小、外观、眼球突出度和眼运动均有显著改善。治疗后视力略有改善(20/50-20/30;P=0.076)。博来霉素注射后未见任何副作用。

结论

博来霉素注射治疗眼眶 LM/LVM 是有效的;本系列中未观察到局部或全身副作用。据我们所知,这是此类治疗的最大报告系列。

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