Duchenne muscular dystrophy (DMD) is the second most common single gene disorder in humans, affecting 1 in 3500 live male births (1). It results from mutation in the dystrophin gene causing the absence of the normal full length dystrophin product (2). Apart from its symptoms of muscle wasting, a significant cognitive impairment has also been reported in DMD patients (3,4). Here, we investigate the effect of the absence of dystrophin on the frequency and amplitude of spontaneous miniature inhibitory postsynaptic currents (mIPSCs) in cerebellar Purkinje cells of the dystrophin-deficient mdx mouse compared to littermate controls. Whole-cell patch-clamp recordings from Purkinje cells were performed in saggital cerebellar slices from mdx (n = 7) and littermate control (n = 6). Purkinje cells were held at -80mV and to prevent action potential-evoked events, TTX (1μM) was added to the bathing solution (composition in mM: NaCl 124, KCl 3.2, CaCl₂ 2.5, MgCl₂ 1.3, NaHCO₃ 26, NaH₂PO₄ 1.25 and D-glucose 25; bubbled with 95% O₂ and 5% CO₂). Spontaneous mIPSCs were analysed using MiniAnalysis (Synaptosoft), for details of the experimental protocol see (5). We found a significant difference (all errors are expressed as SEM) between the frequency of spontaneous mIPSCs in mdx (1.38 ± 0.46 Hz; n = 8 cells) and littermate control mice (2.74 ± 1.00 Hz; n = 8 cells) (two-tailed t test, p = 0.0036). The average amplitude of mIPSCs from littermate controls (74.81 ± 7.840 pA, n=9) were also significantly different from mdx mice (53.81 ± 4.621 pA, n=8), p = 0.041, two tailed unpaired t test. The reduction in the frequency and amplitude of mIPSCs in dystrophin deficient mice could be due to a mislocalization of the GABA_A receptor channels at post-synaptic regions. This finding suggests that dystrophin may play an important role in ion channel localization in cerebellar Purkinje cells.