دورية أكاديمية

A survival analysis of ventricular access devices for delivery of cerliponase alfa

التفاصيل البيبلوغرافية
العنوان: A survival analysis of ventricular access devices for delivery of cerliponase alfa
المؤلفون: Craven, Claudia L, Gissen, Paul, Bower, Rebecca, Lee, Laura, Aquilina, Kristian, Thompson, Dominic NP
المصدر: Journal of Neurosurgery: Pediatrics , 29 (1) pp. 115-121. (2022)
بيانات النشر: American Association of Neurological Surgeons
سنة النشر: 2022
المجموعة: University College London: UCL Discovery
مصطلحات موضوعية: Cerliponase alfa, intracerebroventricular, drug delivery, neurosurgery, neuronal ceroid lipofuscinosis, ventricular access devices, punctures, complications, surgical technique
الوصف: OBJECTIVE: Late infantile neuronal ceroid lipofuscinosis type 2 (CLN2) is a rare autosomal recessive disease caused by tripeptidyl peptidase 1 enzyme deficiency. At the authors' center, the medication cerliponase alfa is administered every 2 weeks via the intracerebroventricular (ICV) route. This requires the placement of a ventricular access device (VAD) or reservoir and frequent percutaneous punctures of this device over the child's lifetime. In this study, the authors audited the longevity and survival of these VADs and examined the causes of device failure. METHODS: A single-center survival analysis of VAD insertions and revisions (January 2014 through June 2020) was conducted. All children received cerliponase alfa infusions through a VAD. Patient characteristics and complications were determined from a prospectively maintained surgical database and patient records. For the VAD survival analysis, the defined endpoint was when the device was removed or changed. Reservoir survival was assessed using Kaplan-Meier curves and the log-rank (Cox-Mantel) test. RESULTS: A total of 17 patients had VADs inserted for drug delivery; median (range) age at first surgery was 4 years 4 months (1 year 8 months to 15 years). Twenty-six VAD operations (17 primary insertions and 9 revisions) were required among these 17 patients. Twelve VAD operations had an associated complication, including CSF infection (n = 6) with Propionibacterium and Staphylococcus species being the most prevalent organisms, significant surgical site swelling preventing infusion (n = 3), leakage/wound breakdown (n = 2), and catheter obstruction (n = 1). There were no complications or deaths associated with VAD insertion. The median (interquartile range) number of punctures was 59.5 (7.5-82.0) for unrevised VADs (n = 17) versus 2 (6-87.5) for revised VADs (n = 9) (p = 0.70). The median survival was 301 days for revisional reservoirs (n = 9) versus 2317 days for primary inserted reservoirs (n = 17) (p = 0.019). CONCLUSIONS: In the context of the current ...
نوع الوثيقة: article in journal/newspaper
وصف الملف: text
اللغة: English
العلاقة: https://discovery.ucl.ac.uk/id/eprint/10145763/2/Gissen_Battens%20manuscript.pdfTest; https://discovery.ucl.ac.uk/id/eprint/10145763Test/
الإتاحة: https://discovery.ucl.ac.uk/id/eprint/10145763/2/Gissen_Battens%20manuscript.pdfTest
https://discovery.ucl.ac.uk/id/eprint/10145763Test/
حقوق: open
رقم الانضمام: edsbas.9DC3D245
قاعدة البيانات: BASE