Attention to the ocular significance of CYP2U1 was brought about by the ocular phenotype of patients with spastic paraplegia 56, an autosomal recessive neurodegeneration characterized by early-onset progressive lower-limb spasticity and weakness due to mutations in CYP2U1 (Tesson et al., 2012). This evidence concerns the gene CYP2U1 and Spastic paraplegia.