14800-DvRappard

66 Chapter 4 Variables, data sources and measurement Of all included patients we collected age, sex, age at pump implantation and height of catheter tip. In MLD, we noted clinical subtype, age at diagnosis and at wheelchair dependency, and whether spasticity (SMLD) or dyskinesia (DMLD) was the predominant motor impairment. Mobility status was scored via GMFC-MLD in MLD patients, and GMFCS in SCP patients. The goals of ITB were noted. Dose pump settings at six months after implantation, including dose of baclofen in micrograms (μg) per day and baclofen pump dosing mode (simple continuous or flexible program with bolus administration) were used as outcome parameter. The starting dose after pump implantation was used as first measurement (t = 0). Data analysis The correlation between age at pump implantation and baclofen dosage at six months was calculated. An Independent Samples T-Test was used to search for a difference in baclofen dosage between DMLD and SMLD and between SMLD and SCP patients at 6 months after pump implantation. For the mode of baclofen dose and number of boluses, the Chi Square test was used. P < 0.05 was considered statistically significant. Analyses were performed using SPSS version 22.0. Figure 1. Study scheme MLD = metachromatic leukodystrophy; ITB = intrathecal baclofen; SMLD = spastic MLD; DMLD = dyskinetic MLD; SCP = spastic cerebral palsy. Cohort of MLD patients with ITB pump implantation from 2006 – 2016 N = 11 Pump removal < 3 months N = 1 MLD patients with ITB pump N = 10 Matched group of SCP patients with ITB pump implantation from 2002 – 2016 N = 8 DMLD N=2 SMLD N=8 Figure 1. Study scheme MLD = metachromatic leukodystrophy; ITB = intrathecal baclofen; SMLD = spastic MLD; DMLD = dyskinetic MLD; SCP = spastic cerebral palsy.

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