Orthokeratology is a procedure involving the use of reverse-geometry lenses to temporarily correct refractive errors. Switching from spectacles to orthokeratology lenses may influence binocular visual function and accommodation. This is the first report of orthokeratology lens-associated acquired esotropia, a form of distance esotropia, in an adolescent with myopic anisometropia. [J Pediatr Ophthalmol Strabismus. 2022;59(1):e7-e10.].