© 2021 Sociedad Anatomica Espanola. All rights reserved.Our aim was to describe electron microscopic findings of anterior lens capsule (ALC) of a patient with granulomatosis with polyangiitis (GPA). A 73-year-old woman referred to our clinic due to decreased vision. The best-corrected visual acuity was counting fingers in 3 m distance with her right eye and in 2 m distance with her left eye. Slit-lamp examination revealed grade-4 nuclear sclerosis in both eyes. An operation was planned for each eye one month apart. The ALCs were obtained during cataract surgery from each eye. Ultrathin sections of ALCs were stained with uranyl acetate and lead citrate and examined under the transmission electron microscope. The lens epithelium was found to be thin and there were some small vacuoles in the cytoplasm. The epithelial surface showed irregularities. There was subepithelial and intercellular oedema, and in some focal areas the lens epithelium was detached from the capsule. ALC obtained from cataract surgery of a subject without any systemic disease showed intracellular vacuoles in some areas with a normal basement membrane. This report showed that the ALC in GPA have significant and demonstrative changes, especially in the epithelial level compared to normal eye. Our findings may have a guiding effect on the pathogenesis of GPA. However, there is still no clear evidence about the existence and pathogenesis of GPA on lens epithelium, and additional studies are needed.