Background: CIPN is a potentially permanent side effect of cisplatin chemotherapy. CIPN remains a major clinical challenge due to lack of effective treatment, impact on quality of life and unexplained inter-individual variability. Methods: Testicular cancer patients (n = 847) given cisplatin-based therapy (median dose: 400 mg/m²) were assessed for responses to the validated EORTC QLQ-CIPN20 questionnaire. Associations were evaluated between frequency of sensory neuropathy and cumulative cisplatin dose, smoking history and age. Using the Illumina HumanOmniExpressExome chip and with imputation, 5.1 million SNPs passed quality control (QC) for GWAS inclusion. A gene-based method, PrediXcan, was used to consider associations between the genetically determined component of gene expression and CIPN. Results: Sensory neuropathy was common (57% having any symptom). CIPN sensory items (n = 8) indicated excellent internal consistency (alpha coefficient = 0.88). Using each patient’s mean sensory neuropathy score, we sorted them into 3 ordinal groups according to severity: 43% none, 41% a little, 16% quite a bit/very much. Only age and smoking status, not dose, were significantly related to sensory neuropathy (OR = 1.04 [95% CI 1.03-1.06] and 1.5 [95% CI 1.2-2.0], respectively). The top 2 SNPs (P = 5 x 10^-7) from a GWAS of 677 patients that passed QC, were each associated with greater risk of sensory neuropathy (OR = 1.9 [95% CI 1.5-2.4] and 1.9 [95% CI 1.5-2.5]). These SNPs are expression QTLs for LYPD3 (rs12797447), SNX8 and GSTT1 (rs4757366) in lymphoblastoid cell lines. PrediXcan identified lower predicted expression of RPRD1B in whole blood (5453 genes tested) met genome-wide significance for association with increased sensory neuropathy risk (P = 3 x 10^-6). Conclusions: Our traditional GWAS identified several SNPs associated with CIPN that were functionally important due to their association with gene expression. Our gene-based test implicated RPRD1B, known to play a role in cell cycle regulation, as associated with CIPN. Future studies will focus on functional validation, analysis of a replication set and meta-analysis with other CIPN studies to identify drug-independent variants.