Purpose. To report the histopathological features of anterior subcapsular cataract associated with atopic dermatitis. Case Report. A 29-year-old man with atopic dermatitis presented with bilateral anterior subcapsular cataract. After routine cataract surgery, the anterior subcapsular cataractous tissue was obtained as an anterior capsulorhexis flap and prepared as a wholemount for histological analysis. The wholemount consisted of a well-demarcated central grayish-white plaque surrounded by transparent capsule, corroborating the slit-lamp biomicroscopic appearance. Higher magnification of the plaque revealed a fibrous and amorphous mass, most likely extracellular matrix owing to the presence of irregularly arranged bundled strands of fibrils, typical of collagen. Lens epithelial cells at the plaque were densely packed and myofibroblast-like and immunoreactive for alpha-smooth muscle actin. In contrast, lens epithelial cells more distant from the plaque retained their regular cuboidal arrangement and regular spacing, and were not labeled for alpha-smooth muscle actin, similar to lens epithelial cells obtained from a non-cataractous case. Conclusions. The presence of alpha-smooth muscle actin-reactive elongated cells at the plaque suggests that the cuboidal lens epithelial cells making up the anterior subcapsular cataract have transdifferentiated into spindle-shaped myofibroblastic cells that produce and deposit aberrant extracellular matrix. This transdifferentiation process, more commonly known as an epithelial-mesenchymal transition, contributes to a fibrotic response leading to the development of human anterior subcapsular cataract.