Boop Scott, Nistal Dominic, Barrios-Anderson Adriel, Cherny W Bruce, Chang Irene J, Shelkowitz Emily, Kho Terry, Goldstein Hannah E, Hauptman Jason
Division of Neurological Surgery, Department of Neurological Surgery, Seattle Children's Hospital, University of Washington, Seattle, WA, USA.
Department of Pediatric Neurosurgery, St. Luke's Children's Hospital, Boise, ID, USA.
Childs Nerv Syst. 2025 Apr 29;41(1):172. doi: 10.1007/s00381-025-06822-4.
Neuronal ceroid lipofuscinosis type 2 (CLN2) is a genetic disease caused by deficiency of the enzyme tripeptidyl peptidase 1 (TPP1), resulting in seizures, vision/cognitive decline, and early death. Cerliponase alfa is an enzyme replacement therapy approved as treatment for CLN2 disease, administered by intracerebroventricular infusion via a surgically implanted device. Maintaining sterility for prolonged scalp-based infusions can be difficult in children and damage devices. We report the development of a technique connecting the intraventricular device and catheter to a chest port to mitigate some of these challenges.
We describe a seven-patient cohort who underwent implantation of our modified infusion system connecting a ventricular catheter distally to a central venous access device (CVAD) port implanted in the anterior chest.
Between March 2019 and April 2024, seven (n = 7) patients aged 2-19 years underwent the placement of this system or had an old device converted to a CVAD port system. None of our patients has experienced significant access issues, infection, or required revision since implantation.
The use of an intraventricular access device connected to a CVAD allows for safe and efficacious long-term infusion of cerliponase alfa therapy and provides a more stable and well-tolerated alternative to scalp-based infusions.
2型神经元蜡样脂褐质沉积症(CLN2)是一种由三肽基肽酶1(TPP1)缺乏引起的遗传性疾病,可导致癫痫发作、视力/认知功能下降和过早死亡。阿法赛洛尼酶是一种被批准用于治疗CLN2疾病的酶替代疗法,通过手术植入的装置进行脑室内输注。对于儿童来说,长时间进行头皮输注时保持无菌状态可能很困难,并且可能会损坏装置。我们报告了一种将脑室内装置和导管连接到胸部端口的技术的开发,以缓解其中一些挑战。
我们描述了一个由7名患者组成的队列,他们接受了我们改良的输注系统的植入,该系统将脑室导管向远侧连接到植入前胸的中心静脉通路装置(CVAD)端口。
在2019年3月至2024年4月期间,7名年龄在2至19岁的患者接受了该系统的植入,或已将旧装置转换为CVAD端口系统。自植入以来,我们的患者均未出现严重的通路问题、感染或需要进行翻修。
使用连接到CVAD的脑室内通路装置可实现阿法赛洛尼酶治疗的安全、有效长期输注,并为头皮输注提供了一种更稳定且耐受性良好的替代方案。