IMPORTANCE: Measurement of retinal nerve fiber layer (RNFL) thickness using optical coherence tomography can aid in the diagnosis and management of glaucoma. We observed a previously unreported phenomenon in eyes with uveitis-associated glaucoma in which paradoxical changes in RNFL thickness were noted. OBSERVATIONS: Four eyes of 3 patients with uveitis-associated glaucoma had a relatively normal RNFL measurement on presentation during periods of active uveitis and raised intraocular pressure. Subsequent control of uveitis and intraocular pressure was associated with a paradoxical thinning of the RNFL and increased cupping. CONCLUSIONS AND RELEVANCE: Normal-appearing measurements of RNFL thickness in patients with uveitis should be interpreted cautiously in those with a raised intraocular pressure. Physicians should recognize that continued thinning of the RNFL and increased cupping, despite good intraocular pressure control in such eyes, may be due to resolution of edema of the RNFL.