A morphometric analysis of the retinal arterioles with adaptive optics imaging in RPE65-associated retinal dystrophy after treatment with voretigene neparvovec.

التفاصيل البيبلوغرافية
العنوان:	A morphometric analysis of the retinal arterioles with adaptive optics imaging in RPE65-associated retinal dystrophy after treatment with voretigene neparvovec.
المؤلفون:	Kortuem FC; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany., Kempf M; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany.; Center for Rare Eye Diseases, University of Tübingen, Tübingen, Germany., Merle DA; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany.; Center for Ophthalmology, Institute for Ophthalmic Research, University of Tübingen, Tübingen, Germany.; Department of Ophthalmology, Medical University of Graz, Graz, Austria., Kuehlewein L; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany.; Center for Ophthalmology, Institute for Ophthalmic Research, University of Tübingen, Tübingen, Germany., Pohl L; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany., Reith M; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany., Jung R; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany., Ott S; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany., Stingl K; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany.; Center for Rare Eye Diseases, University of Tübingen, Tübingen, Germany., Stingl K; Center for Ophthalmology, University Eye Hospital, University of Tübingen, Tübingen, Germany.; Center for Rare Eye Diseases, University of Tübingen, Tübingen, Germany.
المصدر:	Acta ophthalmologica [Acta Ophthalmol] 2024 May; Vol. 102 (3), pp. e358-e366. Date of Electronic Publication: 2023 Sep 16.
نوع المنشور:	Journal Article
اللغة:	English
بيانات الدورية:	Publisher: Wiley-Blackwell Country of Publication: England NLM ID: 101468102 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1755-3768 (Electronic) Linking ISSN: 1755375X NLM ISO Abbreviation: Acta Ophthalmol Subsets: MEDLINE
أسماء مطبوعة:	Publication: Oxford, UK : Wiley-Blackwell Original Publication: Oxford, UK ; Malden, MA : Blackwell Munksgaard
مواضيع طبية MeSH:	Arterioles* , Retinal Dystrophies/diagnosis , Retinal Dystrophies/genetics , Retinal Vessels*, Humans ; Ophthalmoscopy
مستخلص:	Purpose: To investigate the changes in retinal arterial architecture after treatment with voretigene neparvovec in patients with retinal dystrophy caused by bi-allelic mutations in the RPE65 gene. Methods: Sixteen eyes treated with voretigene neparvovec at the University Eye Clinic in Tuebingen, Germany, underwent adaptive optics ophthalmoscopy (AO) imaging at baseline and 2 weeks, 1, 3, 6 and 12 months after treatment. Follow-up was performed in six eyes of four patients. For each eye, five different positions at arterial vessels were selected and the wall-to-lumen ratio (WLR), the lumen diameter (LD) and the wall cross-sectional area (WCSA) were measured by the manufacturer's software over the observational period. Results: Vast retinal atrophy dominated all gained AO images. WLR fluctuated in the observation period without statistically significant change. LD and WCSA changed significantly after 2 weeks from the baseline examination and returned to values similar to baseline thereafter. There were no signs of inflammation such as macrophages or perivascular accumulated fluid visible. Conclusion: AO imaging of the retinal vessels in RPE65-associated retinal dystrophies (IRD) is challenging. There was no change in the retinal arterial vasculature over the observation period of 12 months that would indicate inflammatory changes. Decrease of the LD and WCSA shortly after treatment might be caused by the perioperative prednisolone intake. AO of retinal vessels can be used as a diagnostic module to complement monitoring the disease and effects of genetic treatments if the acquisition is possible in selected cases. (© 2023 The Authors. Acta Ophthalmologica published by John Wiley & Sons Ltd on behalf of Acta Ophthalmologica Scandinavica Foundation.)
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معلومات مُعتمدة:	Egon Schumacher-Stiftung, Barnstorf, Germany; STI 727/1-1 German Research Council; 276693517 German Research Council
فهرسة مساهمة:	Keywords: LCA; RPE65; gene therapy; retinal inflammation; retinitis pigmentosa
تواريخ الأحداث:	Date Created: 20230916 Date Completed: 20240410 Latest Revision: 20240416
رمز التحديث:	20240416
DOI:	10.1111/aos.15765
PMID:	37715554
قاعدة البيانات:	MEDLINE

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تدمد:	1755-3768
DOI:	10.1111/aos.15765