Utility of Optical Coherence Tomography in the Diagnosis and Management of Optic Neuropathy in Patients with Fibrous Dysplasia
ABSTRACT Optic neuropathy (ON) is a highly disabling complication of fibrous dysplasia (FD). 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 at...
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| Veröffentlicht in: | Journal of bone and mineral research 2020-11, Vol.35 (11), p.2199-2210 |
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| container_title | Journal of bone and mineral research |
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| creator | Pan, Kristen S FitzGibbon, Edmond J Vitale, Susan Lee, Janice S Collins, Michael T Boyce, Alison M |
| description | ABSTRACT
Optic neuropathy (ON) is a highly disabling complication of fibrous dysplasia (FD). 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 = 70) 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 |
| doi_str_mv | 10.1002/jbmr.4129 |
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Optic neuropathy (ON) is a highly disabling complication of fibrous dysplasia (FD). 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 = 70) 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 < 0.0001) with sensitivity and specificity of 100% and 95%, respectively, when using the diagnostic criterion of ≤71 μm. 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 (−0.70 μm/year, p < 0.001) than subjects with normal vision (−0.16 μm/year, p < 0.05). When assessed longitudinally, subjects with normal vision demonstrated an increase in RNFL thickness until approximately age 20 years that decreased thereafter. In contrast, subjects with ON exhibited an earlier decrease in RNFL thickness during adolescence. In conclusion, RNFL thickness of ≤71 μm 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. © 2020 American Society for Bone and Mineral Research.</description><identifier>ISSN: 0884-0431</identifier><identifier>EISSN: 1523-4681</identifier><identifier>DOI: 10.1002/jbmr.4129</identifier><identifier>PMID: 32644197</identifier><language>eng</language><publisher>Hoboken, USA: John Wiley & Sons, Inc</publisher><subject>Age ; Atrophy ; Computed tomography ; FIBROUS DYSPLASIA ; Optic nerve ; OPTIC NEUROPATHY ; OPTICAL COHERENCE TOMOGRAPHY ; Optics ; RETINAL NERVE FIBER LAYER ; Thinning ; Tomography ; Vision</subject><ispartof>Journal of bone and mineral research, 2020-11, Vol.35 (11), p.2199-2210</ispartof><rights>2020 American Society for Bone and Mineral Research</rights><rights>2020 American Society for Bone and Mineral Research.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c3889-2b7f86ff6028286255809e856180c125f8a0963723ad84c1256f5b4eb0ebe1733</citedby><cites>FETCH-LOGICAL-c3889-2b7f86ff6028286255809e856180c125f8a0963723ad84c1256f5b4eb0ebe1733</cites><orcidid>0000-0003-3676-7644 ; 0000-0002-9559-0124</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1002%2Fjbmr.4129$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Fjbmr.4129$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,780,784,1417,27923,27924,45573,45574</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/32644197$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Pan, Kristen S</creatorcontrib><creatorcontrib>FitzGibbon, Edmond J</creatorcontrib><creatorcontrib>Vitale, Susan</creatorcontrib><creatorcontrib>Lee, Janice S</creatorcontrib><creatorcontrib>Collins, Michael T</creatorcontrib><creatorcontrib>Boyce, Alison M</creatorcontrib><title>Utility of Optical Coherence Tomography in the Diagnosis and Management of Optic Neuropathy in Patients with Fibrous Dysplasia</title><title>Journal of bone and mineral research</title><addtitle>J Bone Miner Res</addtitle><description>ABSTRACT
Optic neuropathy (ON) is a highly disabling complication of fibrous dysplasia (FD). 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 = 70) 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 < 0.0001) with sensitivity and specificity of 100% and 95%, respectively, when using the diagnostic criterion of ≤71 μm. 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 (−0.70 μm/year, p < 0.001) than subjects with normal vision (−0.16 μm/year, p < 0.05). When assessed longitudinally, subjects with normal vision demonstrated an increase in RNFL thickness until approximately age 20 years that decreased thereafter. In contrast, subjects with ON exhibited an earlier decrease in RNFL thickness during adolescence. In conclusion, RNFL thickness of ≤71 μm 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. © 2020 American Society for Bone and Mineral Research.</description><subject>Age</subject><subject>Atrophy</subject><subject>Computed tomography</subject><subject>FIBROUS DYSPLASIA</subject><subject>Optic nerve</subject><subject>OPTIC NEUROPATHY</subject><subject>OPTICAL COHERENCE TOMOGRAPHY</subject><subject>Optics</subject><subject>RETINAL NERVE FIBER LAYER</subject><subject>Thinning</subject><subject>Tomography</subject><subject>Vision</subject><issn>0884-0431</issn><issn>1523-4681</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2020</creationdate><recordtype>article</recordtype><recordid>eNp1kU1P3DAQhi3UCpaFQ_9AZakXOIQdO7bXOZYF-iG2IATnyMlOdr1K4mA7Qrnw25vtUg6Vehpp5plHo3kJ-cTgggHw2bZo_IVgPDsgEyZ5mgil2QcyAa1FAiJlR-Q4hC0AKKnUITlKuRKCZfMJeX2KtrZxoK6id120panpwm3QY1sifXSNW3vTbQZqWxo3SK-sWbcu2EBNu6JL05o1NtjG9336C3vvOhP3O_cm2nEc6IuNG3pjC-_6QK-G0NUmWHNCPlamDnj6Vqfk6eb6cfE9ub379mPx9TYpU62zhBfzSquqUsA114pLqSFDLRXTUDIuK20gU-mcp2alxa6jKlkILAALZPM0nZKzvbfz7rnHEPPGhhLr2rQ4HpRzwTmAFuPzpuTLP-jW9b4drxspmQkOcjROyfmeKr0LwWOVd942xg85g3wXSr4LJd-FMrKf34x90eDqnfybwgjM9sCLrXH4vyn_ebl8-KP8DbF8liY</recordid><startdate>202011</startdate><enddate>202011</enddate><creator>Pan, Kristen S</creator><creator>FitzGibbon, Edmond J</creator><creator>Vitale, Susan</creator><creator>Lee, Janice S</creator><creator>Collins, Michael T</creator><creator>Boyce, Alison M</creator><general>John Wiley & Sons, Inc</general><general>Wiley Subscription Services, Inc</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7QP</scope><scope>7TS</scope><scope>K9.</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0003-3676-7644</orcidid><orcidid>https://orcid.org/0000-0002-9559-0124</orcidid></search><sort><creationdate>202011</creationdate><title>Utility of Optical Coherence Tomography in the Diagnosis and Management of Optic Neuropathy in Patients with Fibrous Dysplasia</title><author>Pan, Kristen S ; FitzGibbon, Edmond J ; Vitale, Susan ; Lee, Janice S ; Collins, Michael T ; Boyce, Alison M</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3889-2b7f86ff6028286255809e856180c125f8a0963723ad84c1256f5b4eb0ebe1733</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2020</creationdate><topic>Age</topic><topic>Atrophy</topic><topic>Computed tomography</topic><topic>FIBROUS DYSPLASIA</topic><topic>Optic nerve</topic><topic>OPTIC NEUROPATHY</topic><topic>OPTICAL COHERENCE TOMOGRAPHY</topic><topic>Optics</topic><topic>RETINAL NERVE FIBER LAYER</topic><topic>Thinning</topic><topic>Tomography</topic><topic>Vision</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Pan, Kristen S</creatorcontrib><creatorcontrib>FitzGibbon, Edmond J</creatorcontrib><creatorcontrib>Vitale, Susan</creatorcontrib><creatorcontrib>Lee, Janice S</creatorcontrib><creatorcontrib>Collins, Michael T</creatorcontrib><creatorcontrib>Boyce, Alison M</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>Calcium & Calcified Tissue Abstracts</collection><collection>Physical Education Index</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><jtitle>Journal of bone and mineral research</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Pan, Kristen S</au><au>FitzGibbon, Edmond J</au><au>Vitale, Susan</au><au>Lee, Janice S</au><au>Collins, Michael T</au><au>Boyce, Alison M</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Utility of Optical Coherence Tomography in the Diagnosis and Management of Optic Neuropathy in Patients with Fibrous Dysplasia</atitle><jtitle>Journal of bone and mineral research</jtitle><addtitle>J Bone Miner Res</addtitle><date>2020-11</date><risdate>2020</risdate><volume>35</volume><issue>11</issue><spage>2199</spage><epage>2210</epage><pages>2199-2210</pages><issn>0884-0431</issn><eissn>1523-4681</eissn><abstract>ABSTRACT
Optic neuropathy (ON) is a highly disabling complication of fibrous dysplasia (FD). 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 = 70) 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 < 0.0001) with sensitivity and specificity of 100% and 95%, respectively, when using the diagnostic criterion of ≤71 μm. 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 (−0.70 μm/year, p < 0.001) than subjects with normal vision (−0.16 μm/year, p < 0.05). When assessed longitudinally, subjects with normal vision demonstrated an increase in RNFL thickness until approximately age 20 years that decreased thereafter. In contrast, subjects with ON exhibited an earlier decrease in RNFL thickness during adolescence. In conclusion, RNFL thickness of ≤71 μm 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. © 2020 American Society for Bone and Mineral Research.</abstract><cop>Hoboken, USA</cop><pub>John Wiley & Sons, Inc</pub><pmid>32644197</pmid><doi>10.1002/jbmr.4129</doi><tpages>12</tpages><orcidid>https://orcid.org/0000-0003-3676-7644</orcidid><orcidid>https://orcid.org/0000-0002-9559-0124</orcidid><oa>free_for_read</oa></addata></record> |
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| source | Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; Oxford University Press Journals All Titles (1996-Current); Wiley Online Library All Journals |
| subjects | Age Atrophy Computed tomography FIBROUS DYSPLASIA Optic nerve OPTIC NEUROPATHY OPTICAL COHERENCE TOMOGRAPHY Optics RETINAL NERVE FIBER LAYER Thinning Tomography Vision |
| title | Utility of Optical Coherence Tomography in the Diagnosis and Management of Optic Neuropathy in Patients with Fibrous Dysplasia |
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