This case report describes the appearance of fibrillary lines in the anterior stroma of a 39-year-old Asian woman wearing overnight orthokeratology (OK) lenses. The fibrillary lines were fine, slightly curved and sub-epithelial, arranged in a band-like annulus in the corneal mid-periphery. The lines were not associated with epithelial staining, although a marked Fischer-Schweitzer corneal mosaic was noted after blinking. Fibrillary lines are a relatively common finding in normal and keratoconic corneas and have been reported previously accompanying OK lens wear. Their origin is unknown and epithelial neural remodelling, corneal biomechanical stress and abrupt corneal curvature changes have been suggested as contributing factors. The appearance of fibrillary lines in our OK patient had no adverse consequences on vision or ocular health, at least in the medium term.