Acute retinal pigment epitheliitis using adaptive optics imaging: a case report

Abstract Background Acute Retinal Pigment Epitheliitis (ARPE, Krill’s disease) is a rare inflammatory retinal disorder commonly affecting young adults. It often presents unilaterally with central vision disruption, and typically resolves with vision restoration within 6 to 12 weeks. The pathogenesis...

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Main Authors: P.A.T. Heutinck, S. Wooning, K. Liman, M. Durand, L. Sanchez Brea, C.C.W. Klaver, V.J.M. Verhoeven, D. Andrade De Jesus, A.A.H.J. Thiadens
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Language:English
Published: BMC 2024-11-01
Series:BMC Ophthalmology
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Online Access:https://doi.org/10.1186/s12886-024-03768-0
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author P.A.T. Heutinck
S. Wooning
K. Liman
M. Durand
L. Sanchez Brea
C.C.W. Klaver
V.J.M. Verhoeven
D. Andrade De Jesus
A.A.H.J. Thiadens
author_facet P.A.T. Heutinck
S. Wooning
K. Liman
M. Durand
L. Sanchez Brea
C.C.W. Klaver
V.J.M. Verhoeven
D. Andrade De Jesus
A.A.H.J. Thiadens
author_sort P.A.T. Heutinck
collection DOAJ
description Abstract Background Acute Retinal Pigment Epitheliitis (ARPE, Krill’s disease) is a rare inflammatory retinal disorder commonly affecting young adults. It often presents unilaterally with central vision disruption, and typically resolves with vision restoration within 6 to 12 weeks. The pathogenesis of ARPE remains a subject of ongoing debate. Adaptive Optics Flood Illumination Ophthalmoscopy (AO-FIO) imaging has emerged as a valuable tool capable of detecting early cone photoreceptor changes and recovery. This case study presents two patients with ARPE, with longitudinal follow-up using multimodal imaging, including optical coherence tomography (OCT) and AO-FIO. Case presentations A 30-year-old male presented with sudden vision loss in both eyes. The best corrected visual acuity (BCVA) was 20/33 and 20/40 Snellen in the right and left eye, respectively. OCT showed interruption of the ellipsoid zone (EZ) band and outer nuclear layer (ONL) in both eyes; AO-FIO imaging revealed a foveal lesion and diminished parafoveal cone density in both eyes compared to two age-matched controls. After 6 months, BCVA was restored to 20/20, and OCT showed recovery of the ONL and EZ. On AO-FIO, the foveal lesion was still present and the parafoveal cone density increased but remained reduced even up to 15 months after onset when compared to the controls. The second patient, a 30-year-old woman, presented with a unilateral drop in vision to 20/63 Snellen. OCT showed discontinuation of the EZ and hyperreflectivity within the ONL and retinal pigment epithelium in the affected eye. The unaffected eye showed no abnormalities. After 3 months, the BCVA improved to 20/16 Snellen and OCT showed recovery of the EZ. AO-FIO was conducted 9 months after onset and revealed reduced parafoveal cone density in the affected and non-affected eye compared to the controls while OCT still showed recovery of all retinal layers. Conclusions ARPE is a self-limiting disease with recovery of BCVA and OCT retinal layers within 6 months. However, our 2 cases showed that parafoveal cone density recovered during follow-up but did not reach levels observed in controls. AO-FIO is an imaging modality that enhances sensitivity in measurements and can therefore be used as a complementary tool for follow-up.
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spelling doaj-art-5a7096fe8b224b999a1e9286b337e5e92024-12-01T12:14:56ZengBMCBMC Ophthalmology1471-24152024-11-0124111110.1186/s12886-024-03768-0Acute retinal pigment epitheliitis using adaptive optics imaging: a case reportP.A.T. Heutinck0S. Wooning1K. Liman2M. DurandL. Sanchez Brea3C.C.W. Klaver4V.J.M. Verhoeven5D. Andrade De Jesus6A.A.H.J. Thiadens7Department of Ophthalmology, Erasmus MC, University Medical CenterEye Image Analysis Group Rotterdam, Department of Radiology & Nuclear Medicine, Erasmus MC, University Medical CenterDepartment of Ophthalmology, Erasmus MC, University Medical CenterEye Image Analysis Group Rotterdam, Department of Radiology & Nuclear Medicine, Erasmus MC, University Medical CenterDepartment of Ophthalmology, Erasmus MC, University Medical CenterDepartment of Ophthalmology, Erasmus MC, University Medical CenterEye Image Analysis Group Rotterdam, Department of Radiology & Nuclear Medicine, Erasmus MC, University Medical CenterDepartment of Ophthalmology, Erasmus MC, University Medical CenterAbstract Background Acute Retinal Pigment Epitheliitis (ARPE, Krill’s disease) is a rare inflammatory retinal disorder commonly affecting young adults. It often presents unilaterally with central vision disruption, and typically resolves with vision restoration within 6 to 12 weeks. The pathogenesis of ARPE remains a subject of ongoing debate. Adaptive Optics Flood Illumination Ophthalmoscopy (AO-FIO) imaging has emerged as a valuable tool capable of detecting early cone photoreceptor changes and recovery. This case study presents two patients with ARPE, with longitudinal follow-up using multimodal imaging, including optical coherence tomography (OCT) and AO-FIO. Case presentations A 30-year-old male presented with sudden vision loss in both eyes. The best corrected visual acuity (BCVA) was 20/33 and 20/40 Snellen in the right and left eye, respectively. OCT showed interruption of the ellipsoid zone (EZ) band and outer nuclear layer (ONL) in both eyes; AO-FIO imaging revealed a foveal lesion and diminished parafoveal cone density in both eyes compared to two age-matched controls. After 6 months, BCVA was restored to 20/20, and OCT showed recovery of the ONL and EZ. On AO-FIO, the foveal lesion was still present and the parafoveal cone density increased but remained reduced even up to 15 months after onset when compared to the controls. The second patient, a 30-year-old woman, presented with a unilateral drop in vision to 20/63 Snellen. OCT showed discontinuation of the EZ and hyperreflectivity within the ONL and retinal pigment epithelium in the affected eye. The unaffected eye showed no abnormalities. After 3 months, the BCVA improved to 20/16 Snellen and OCT showed recovery of the EZ. AO-FIO was conducted 9 months after onset and revealed reduced parafoveal cone density in the affected and non-affected eye compared to the controls while OCT still showed recovery of all retinal layers. Conclusions ARPE is a self-limiting disease with recovery of BCVA and OCT retinal layers within 6 months. However, our 2 cases showed that parafoveal cone density recovered during follow-up but did not reach levels observed in controls. AO-FIO is an imaging modality that enhances sensitivity in measurements and can therefore be used as a complementary tool for follow-up.https://doi.org/10.1186/s12886-024-03768-0Adaptive optics flood illumination ophthalmoscopyAcute retinal pigment epitheliitisKrill’s diseaseCone photoreceptors
spellingShingle P.A.T. Heutinck
S. Wooning
K. Liman
M. Durand
L. Sanchez Brea
C.C.W. Klaver
V.J.M. Verhoeven
D. Andrade De Jesus
A.A.H.J. Thiadens
Acute retinal pigment epitheliitis using adaptive optics imaging: a case report
BMC Ophthalmology
Adaptive optics flood illumination ophthalmoscopy
Acute retinal pigment epitheliitis
Krill’s disease
Cone photoreceptors
title Acute retinal pigment epitheliitis using adaptive optics imaging: a case report
title_full Acute retinal pigment epitheliitis using adaptive optics imaging: a case report
title_fullStr Acute retinal pigment epitheliitis using adaptive optics imaging: a case report
title_full_unstemmed Acute retinal pigment epitheliitis using adaptive optics imaging: a case report
title_short Acute retinal pigment epitheliitis using adaptive optics imaging: a case report
title_sort acute retinal pigment epitheliitis using adaptive optics imaging a case report
topic Adaptive optics flood illumination ophthalmoscopy
Acute retinal pigment epitheliitis
Krill’s disease
Cone photoreceptors
url https://doi.org/10.1186/s12886-024-03768-0
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