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The optimal test for identifying and monitoring ON in FD is unknown. Optical coherence tomography (OCT) is an imaging modality that detects retinal nerve fiber layer (RNFL) thinning, a sign of optic nerve atrophy. The purpose of this study was to (i) assess the ability of OCT RNFL thickness measurements to identify ON in FD; (ii) compare the performance of RNFL thickness to computed tomography measurements; and (iii) examine changes in RNFL thickness over time to assess disease progression. A retrospective cohort study was performed to assess subjects (n =\u200970) who underwent neuro-ophthalmologic examination, including OCT. The diagnostic utility of RNFL thickness was determined using receiver operator characteristic (ROC) curve analysis, and the accuracy was compared with computed tomography measurements. The relationship between RNFL thickness and age was assessed cross-sectionally, using generalized estimating equation methodology, and longitudinally, using a generalized mixed model. Eleven subjects were identified with ON. RNFL thickness identified ON (area under curve = 0.997, p &amp;lt;\u20090.0001) with sensitivity and specificity of 100% and 95%, respectively, when using the diagnostic criterion of \u226471\u2009\u03bcm. RNFL thickness outperformed computed tomography measurements of optic canal narrowing and optic nerve stretch. Subjects with ON exhibited a greater decrease in RNFL thickness with each year of age (\u22120.70\u2009\u03bcm\/year, p &amp;lt;\u20090.001) than subjects with normal vision (\u22120.16\u2009\u03bcm\/year, p &amp;lt;\u20090.05). When assessed longitudinally, subjects with normal vision demonstrated an increase in RNFL thickness until approximately age 20\u2009years that decreased thereafter. In contrast, subjects with ON exhibited an earlier decrease in RNFL thickness during adolescence. In conclusion, RNFL thickness of \u226471\u2009\u03bcm accurately identified ON in this population. By establishing the difference in rate of RNFL thinning in patients with and without ON, clinicians may distinguish between patients at risk for ON and intervene before irreversible damage. \u00a9 2020 American Society for Bone and Mineral Research.<\/jats:p>","DOI":"10.1002\/jbmr.4129","type":"journal-article","created":{"date-parts":[[2020,7,9]],"date-time":"2020-07-09T08:53:08Z","timestamp":1594284788000},"page":"2199-2210","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Utility of Optical Coherence Tomography in the Diagnosis and Management of Optic Neuropathy in Patients with Fibrous Dysplasia"],"prefix":"10.1093","volume":"35","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9559-0124","authenticated-orcid":false,"given":"Kristen S","family":"Pan","sequence":"first","affiliation":[{"name":"Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research National Institutes of Health Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edmond J","family":"FitzGibbon","sequence":"additional","affiliation":[{"name":"Laboratory of Sensorimotor Research National Eye Institute, National Institutes of Health Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Susan","family":"Vitale","sequence":"additional","affiliation":[{"name":"Division of Epidemiology and Clinical Applications National Eye Institute, National Institutes of Health Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janice S","family":"Lee","sequence":"additional","affiliation":[{"name":"Craniofacial Anomalies and Regeneration Section National Institute of Dental and Craniofacial Research, National Institutes of Health Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3676-7644","authenticated-orcid":false,"given":"Michael T","family":"Collins","sequence":"additional","affiliation":[{"name":"Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research National Institutes of Health Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alison M","family":"Boyce","sequence":"additional","affiliation":[{"name":"Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research National Institutes of Health Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2020,7,9]]},"reference":[{"issue":"6","key":"2024031419002314000_jbmr4129-bib-0001","first-page":"1587","article-title":"Fibrous dysplasia of bone in the McCune-Albright syndrome: abnormalities in bone formation","volume":"151","author":"Riminucci","year":"1997","journal-title":"Am J Pathol."},{"issue":"6","key":"2024031419002314000_jbmr4129-bib-0002","doi-asserted-by":"crossref","first-page":"355","DOI":"10.1677\/JME-10-0097","article-title":"Skeletal progenitors and the GNAS gene: fibrous dysplasia of bone read through stem cells","volume":"45","author":"Riminucci","year":"2010","journal-title":"J Mol Endocrinol."},{"issue":"2","key":"2024031419002314000_jbmr4129-bib-0003","doi-asserted-by":"crossref","first-page":"249","DOI":"10.1002\/(SICI)1096-9896(199901)187:2<249::AID-PATH222>3.0.CO;2-J","article-title":"The histopathology of fibrous dysplasia of bone in patients with activating mutations of the Gs alpha gene: site-specific patterns and recurrent histological hallmarks","volume":"187","author":"Riminucci","year":"1999","journal-title":"J Pathol."},{"key":"2024031419002314000_jbmr4129-bib-0004","volume-title":"GeneReviews\u00ae","author":"Boyce","year":"1993"},{"issue":"5","key":"2024031419002314000_jbmr4129-bib-0005","doi-asserted-by":"crossref","first-page":"1011","DOI":"10.1227\/01.NEU.0000254440.02736.E3","article-title":"Long-term outcome of optic nerve encasement and optic nerve decompression in patients with fibrous dysplasia: risk factors for blindness and safety of observation","volume":"59","author":"Cutler","year":"2006","journal-title":"Neurosurgery."},{"issue":"6","key":"2024031419002314000_jbmr4129-bib-0006","first-page":"631","article-title":"Compressive optic neuropathy caused by fibrous dysplasia","volume":"26","author":"Messaoud","year":"2003","journal-title":"J Fr Ophtalmol."},{"issue":"7","key":"2024031419002314000_jbmr4129-bib-0007","first-page":"263","article-title":"Acute monocular blindness secondary to fibrous dysplasia of the skull: a case report","volume":"24","author":"Bland","year":"1992","journal-title":"Ann Ophthalmol."},{"issue":"2","key":"2024031419002314000_jbmr4129-bib-0008","doi-asserted-by":"crossref","first-page":"350","DOI":"10.3171\/jns.2000.92.2.0350","article-title":"Visual loss associated with fibrous dysplasia of the anterior skull base. 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