Retina tissue validation of optical coherence tomography determined outer nuclear layer loss in FTLD-tau

Abstract Alzheimer’s disease (AD) is associated with inner retina (nerve fiber and ganglion cell layers) thinning. In contrast, we have seen outer retina thinning driven by photoreceptor outer nuclear layer (ONL) thinning with antemortem optical coherence tomography (OCT) among patients considered t...

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Autores principales:	Benjamin J. Kim, Vivian Lee, Edward B. Lee, Adrienne Saludades, John Q. Trojanowski, Joshua L. Dunaief, Murray Grossman, David J. Irwin
Formato:	article
Lenguaje:	EN
Publicado:	BMC 2021
Materias:	Neurology. Diseases of the nervous system RC346-429
Acceso en línea:	https://doaj.org/article/9b9e6585afe94301bd7742334141f114
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spelling	oai:doaj.org-article:9b9e6585afe94301bd7742334141f1142021-11-21T12:07:33ZRetina tissue validation of optical coherence tomography determined outer nuclear layer loss in FTLD-tau10.1186/s40478-021-01290-82051-5960https://doaj.org/article/9b9e6585afe94301bd7742334141f1142021-11-01T00:00:00Zhttps://doi.org/10.1186/s40478-021-01290-8https://doaj.org/toc/2051-5960Abstract Alzheimer’s disease (AD) is associated with inner retina (nerve fiber and ganglion cell layers) thinning. In contrast, we have seen outer retina thinning driven by photoreceptor outer nuclear layer (ONL) thinning with antemortem optical coherence tomography (OCT) among patients considered to have a frontotemporal degeneration tauopathy (FTLD-Tau). Our objective was to determine if postmortem retinal tissue from FTLD-Tau patients demonstrates ONL loss observed antemortem on OCT. Two probable FTLD-Tau patients that were deeply phenotyped by clinical and genetic testing were imaged with OCT and followed to autopsy. Postmortem brain and retinal tissue were evaluated by a neuropathologist and ocular pathologist, respectively, masked to diagnosis. OCT findings were correlated with retinal histology. The two patients had autopsy-confirmed FTLD-Tau neuropathology and had antemortem OCT measurements showing ONL thinning (66.9 μm, patient #1; 74.9 μm, patient #2) below the 95% confidence interval of normal limits (75.1–120.7 μm) in our healthy control cohort. Postmortem, retinal tissue from both patients demonstrated loss of nuclei in the ONL, matching ONL loss visualized on antemortem OCT. Nuclei counts from each area of ONL loss (2 – 3 nuclei per column) seen in patient eyes were below the 95% confidence interval (4 – 8 nuclei per column for ONL) of 3 normal control retinas analyzed at the same location. Our evaluation of retinal tissue from FTLD-Tau patients confirms ONL loss seen antemortem by OCT. Continued investigation of ONL thinning as a biomarker that may distinguish FTLD-Tau from other dementias is warranted.Benjamin J. KimVivian LeeEdward B. LeeAdrienne SaludadesJohn Q. TrojanowskiJoshua L. DunaiefMurray GrossmanDavid J. IrwinBMCarticleNeurology. Diseases of the nervous systemRC346-429ENActa Neuropathologica Communications, Vol 9, Iss 1, Pp 1-8 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Neurology. Diseases of the nervous system RC346-429
spellingShingle	Neurology. Diseases of the nervous system RC346-429 Benjamin J. Kim Vivian Lee Edward B. Lee Adrienne Saludades John Q. Trojanowski Joshua L. Dunaief Murray Grossman David J. Irwin Retina tissue validation of optical coherence tomography determined outer nuclear layer loss in FTLD-tau
description	Abstract Alzheimer’s disease (AD) is associated with inner retina (nerve fiber and ganglion cell layers) thinning. In contrast, we have seen outer retina thinning driven by photoreceptor outer nuclear layer (ONL) thinning with antemortem optical coherence tomography (OCT) among patients considered to have a frontotemporal degeneration tauopathy (FTLD-Tau). Our objective was to determine if postmortem retinal tissue from FTLD-Tau patients demonstrates ONL loss observed antemortem on OCT. Two probable FTLD-Tau patients that were deeply phenotyped by clinical and genetic testing were imaged with OCT and followed to autopsy. Postmortem brain and retinal tissue were evaluated by a neuropathologist and ocular pathologist, respectively, masked to diagnosis. OCT findings were correlated with retinal histology. The two patients had autopsy-confirmed FTLD-Tau neuropathology and had antemortem OCT measurements showing ONL thinning (66.9 μm, patient #1; 74.9 μm, patient #2) below the 95% confidence interval of normal limits (75.1–120.7 μm) in our healthy control cohort. Postmortem, retinal tissue from both patients demonstrated loss of nuclei in the ONL, matching ONL loss visualized on antemortem OCT. Nuclei counts from each area of ONL loss (2 – 3 nuclei per column) seen in patient eyes were below the 95% confidence interval (4 – 8 nuclei per column for ONL) of 3 normal control retinas analyzed at the same location. Our evaluation of retinal tissue from FTLD-Tau patients confirms ONL loss seen antemortem by OCT. Continued investigation of ONL thinning as a biomarker that may distinguish FTLD-Tau from other dementias is warranted.
format	article
author	Benjamin J. Kim Vivian Lee Edward B. Lee Adrienne Saludades John Q. Trojanowski Joshua L. Dunaief Murray Grossman David J. Irwin
author_facet	Benjamin J. Kim Vivian Lee Edward B. Lee Adrienne Saludades John Q. Trojanowski Joshua L. Dunaief Murray Grossman David J. Irwin
author_sort	Benjamin J. Kim
title	Retina tissue validation of optical coherence tomography determined outer nuclear layer loss in FTLD-tau
title_short	Retina tissue validation of optical coherence tomography determined outer nuclear layer loss in FTLD-tau
title_full	Retina tissue validation of optical coherence tomography determined outer nuclear layer loss in FTLD-tau
title_fullStr	Retina tissue validation of optical coherence tomography determined outer nuclear layer loss in FTLD-tau
title_full_unstemmed	Retina tissue validation of optical coherence tomography determined outer nuclear layer loss in FTLD-tau
title_sort	retina tissue validation of optical coherence tomography determined outer nuclear layer loss in ftld-tau
publisher	BMC
publishDate	2021
url	https://doaj.org/article/9b9e6585afe94301bd7742334141f114
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Retina tissue validation of optical coherence tomography determined outer nuclear layer loss in FTLD-tau

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