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硬化治疗联合平阳霉素不会对面神经功能造成不良影响,适用于儿童头颈部囊性淋巴管畸形。

Sclerotherapy with bleomycin does not adversely affect facial nerve function in children with cervicofacial cystic lymphatic malformation.

机构信息

Department of Pediatric Surgery, Hacettepe University Faculty of Medicine, Ankara 06100, Turkey.

出版信息

J Pediatr Surg. 2010 Aug;45(8):1627-32. doi: 10.1016/j.jpedsurg.2009.12.014.

Abstract

BACKGROUND AND PURPOSE

Sclerotherapy with bleomycin sulfate (BS) is currently used in the management of cervicofacial cystic lymphatic malformations in children. Neurotoxic adverse effects of BS after intraventricular or intracavitary administration have been reported; however, the effects of intralesionally administered BS on the adjacent peripheral neural structures have not been previously investigated. The authors conducted a clinical experimental study to evaluate facial nerve function in children who have undergone BS sclerotherapy for cervicofacial cystic lymphatic malformation.

MATERIALS AND METHODS

Twelve patients who underwent BS sclerotherapy for cervicofacial lymphatic malformation were included in the study. The hospital records were reviewed, and the following data were recorded: age at admission and at the time of motor nerve conduction study (MNCS) and electromyography (EMG) study, sex, time elapsed between sclerotherapy and the EMG study, and the outcome. The MNCS/EMG study was performed by neurologists blinded to the side of sclerotherapy. Bilateral facial MNCS and needle-EMG study of the orbicularis oris muscle on the treated side were performed. The previously treated and untreated sides constituted the study and control groups, respectively. In the MNCS, compound muscle action potential (CMAP) amplitude and distal latencies were recorded from the orbicularis oculi and orbicularis oris muscles on both sides, and needle-EMG of the orbicularis oris muscle was performed on the treated side.

RESULTS

The male-to-female ratio was 2, and age at the time of sclerotherapy ranged from 1 month to 16 years (median, 19.5 months). The lymphatic malformations were located in the right submandibular (n = 5), left submandibular (n = 6), and in the right buccal (n = 1) areas. Bleomycin sulfate was injected 1 to 4 times, and the time elapsed between injections varied from 1 to 6 months. The results of sclerotherapy were excellent, with residual disease observed in only 1 patient. The MNCS/EMG study was performed 6 months to 10 years after completion of sclerotherapy, and ages of the patients at the time of the study ranged from 4 to 17 years. Side-to-side CMAP amplitude difference did not exceed 50% for orbicularis oculi and orbicularis oris muscles. The mean CMAP amplitude of orbicularis oculi and orbicularis oris muscles on the treated and untreated sides (1219.0 +/- 842.0 vs 1202.4 +/- 923.8 microV and 1866.3 +/- 911.5 vs 1921.0 +/- 910.0 microV, respectively) did not differ between groups (P = .76 and P = .80). Distal latencies recorded from orbicularis oculi and orbicularis oris muscles on treated and untreated sides (2.64 +/- 0.46 vs 2.68 +/- 0.47 milliseconds and 3.10 +/- 0.35 vs 3.10 +/- 0.25 milliseconds, respectively) also did not differ between groups (P = .71 and P = .80). Needle-EMG orbicularis oris muscle (n = 11) on the treated side showed normal findings at rest, and there was no spontaneous activity. During mild voluntary contraction, the amplitude and duration of motor unit action potentials were within normal limits except in one case. Interference patterns were also normal in all cases.

CONCLUSION

Bleomycin sulfate did not adversely affect facial nerve function in children who underwent sclerotherapy for cervicofacial cystic lymphatic malformation when it was applied according to our protocol.

摘要

背景与目的

硫酸博来霉素(BS)硬化疗法目前用于治疗儿童头颈部囊性淋巴管畸形。虽然已有 BS 脑室或囊内给药后出现神经毒性不良反应的报道,但尚未研究过腔内给药对邻近周围神经结构的影响。作者进行了一项临床实验研究,以评估接受 BS 硬化疗法治疗头颈部囊性淋巴管畸形的儿童的面神经功能。

材料与方法

本研究纳入了 12 例接受 BS 硬化疗法治疗头颈部淋巴管畸形的患者。回顾了患者的病历记录,包括入院时和行运动神经传导研究(MNCS)和肌电图(EMG)检查时的年龄、性别、硬化治疗与 EMG 检查之间的时间间隔以及结果。MNCS/EMG 研究由不了解硬化治疗侧别的神经科医生进行。对治疗侧的眼轮匝肌进行双侧面神经 MNCS 和针极肌电图检查。治疗侧和未治疗侧分别构成研究组和对照组。在 MNCS 中,记录双侧眼轮匝肌和口轮匝肌的复合肌肉动作电位(CMAP)幅度和远端潜伏期,并对治疗侧的口轮匝肌进行针极肌电图检查。

结果

男女比例为 2:1,硬化治疗时的年龄为 1 个月至 16 岁(中位数,19.5 个月)。淋巴管畸形位于右侧下颌下(n=5)、左侧下颌下(n=6)和右侧颊部(n=1)区域。BS 注射 1 至 4 次,注射间隔为 1 至 6 个月。硬化治疗效果良好,仅 1 例患者残留病变。MNCS/EMG 研究在硬化治疗完成后 6 个月至 10 年内进行,患者年龄为 4 至 17 岁。眼轮匝肌和口轮匝肌的双侧 CMAP 幅度差异均不超过 50%。治疗侧和未治疗侧眼轮匝肌和口轮匝肌的平均 CMAP 幅度(分别为 1219.0±842.0μV 和 1202.4±923.8μV,1866.3±911.5μV 和 1921.0±910.0μV)无组间差异(P=0.76 和 P=0.80)。治疗侧和未治疗侧眼轮匝肌和口轮匝肌记录的远端潜伏期(分别为 2.64±0.46 毫秒和 2.68±0.47 毫秒,3.10±0.35 毫秒和 3.10±0.25 毫秒)也无组间差异(P=0.71 和 P=0.80)。治疗侧口轮匝肌的针极肌电图(n=11)在静息时表现正常,无自发性活动。在轻度随意收缩时,运动单位动作电位的幅度和持续时间均在正常范围内,除 1 例外。所有病例的干扰模式也均正常。

结论

根据我们的方案,BS 腔内给药治疗头颈部囊性淋巴管畸形不会对儿童面神经功能产生不良影响。

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