Bilateral exudative retinal detachments after subretinal gene therapy with voretigene neparvovec-rzyl for RPE65 Leber Congenital Amaurosis
Purpose: To report panuveitis with exudative retinal detachments in a healthy 27-year-old woman with biallelic mutations in the RPE65 gene, who underwent bilateral sequential gene therapy with subretinal administration of voretigene neparvovec-rzyl. Observations: Visual acuity improved for 30 days a...
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| Format: | Article |
| Language: | English |
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Elsevier
2023-12-01
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| Series: | American Journal of Ophthalmology Case Reports |
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| Online Access: | http://www.sciencedirect.com/science/article/pii/S2451993623000877 |
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| author | Alcina K. Lidder Stephanie Choi Yasha S. Modi Scott E. Brodie Janet L. Davis Ninel Z. Gregori Byron L. Lam |
| author_facet | Alcina K. Lidder Stephanie Choi Yasha S. Modi Scott E. Brodie Janet L. Davis Ninel Z. Gregori Byron L. Lam |
| author_sort | Alcina K. Lidder |
| collection | DOAJ |
| description | Purpose: To report panuveitis with exudative retinal detachments in a healthy 27-year-old woman with biallelic mutations in the RPE65 gene, who underwent bilateral sequential gene therapy with subretinal administration of voretigene neparvovec-rzyl. Observations: Visual acuity improved for 30 days after surgery as oral corticosteroids were tapered. At postoperative week 6, vision declined due to sudden onset uveitis and exudative retinal detachments in both eyes. HLA Class II typing revealed the haplotype associated with sympathetic ophthalmia and Vogt-Koyanagi-Harada (VKH). The inflammation improved after corticosteroid, mycophenolate mofetil, and adalimumab therapy while vision remained poor. Conclusions and Importance: Surgically-induced sympathetic ophthalmia is a plausible explanation for the clinical findings; surgery of both eyes within one week would conceal the inciting eye. VKH or inflammation related to the gene therapy are other possible etiologies but severe bilateral panuveitis has not been reported with voretigene neparvovec-rzyl. Informed consent for gene therapy surgery should include a discussion of the rare complication of sympathetic ophthalmia following vitrectomy surgery. |
| first_indexed | 2024-03-09T01:33:51Z |
| format | Article |
| id | doaj.art-05675087d50e47d2afc8f087a3df93aa |
| institution | Directory Open Access Journal |
| issn | 2451-9936 |
| language | English |
| last_indexed | 2024-03-09T01:33:51Z |
| publishDate | 2023-12-01 |
| publisher | Elsevier |
| record_format | Article |
| series | American Journal of Ophthalmology Case Reports |
| spelling | doaj.art-05675087d50e47d2afc8f087a3df93aa2023-12-09T06:07:05ZengElsevierAmerican Journal of Ophthalmology Case Reports2451-99362023-12-0132101879Bilateral exudative retinal detachments after subretinal gene therapy with voretigene neparvovec-rzyl for RPE65 Leber Congenital AmaurosisAlcina K. Lidder0Stephanie Choi1Yasha S. Modi2Scott E. Brodie3Janet L. Davis4Ninel Z. Gregori5Byron L. Lam6Bascom Palmer Eye Institute, University of Miami Miller School of Medicine, Miami, FL, United StatesDepartment of Ophthalmology, New York University Grossman School of Medicine, New York, NY, United StatesDepartment of Ophthalmology, New York University Grossman School of Medicine, New York, NY, United StatesHarkness Eye Institute, Columbia University, New York, NY, United StatesBascom Palmer Eye Institute, University of Miami Miller School of Medicine, Miami, FL, United StatesBascom Palmer Eye Institute, University of Miami Miller School of Medicine, Miami, FL, United StatesBascom Palmer Eye Institute, University of Miami Miller School of Medicine, Miami, FL, United States; Corresponding author. Bascom Palmer Eye Institute, University of Miami Miller School of Medicine, 900 NW 17th Street, Miami, FL, 33136, United States.Purpose: To report panuveitis with exudative retinal detachments in a healthy 27-year-old woman with biallelic mutations in the RPE65 gene, who underwent bilateral sequential gene therapy with subretinal administration of voretigene neparvovec-rzyl. Observations: Visual acuity improved for 30 days after surgery as oral corticosteroids were tapered. At postoperative week 6, vision declined due to sudden onset uveitis and exudative retinal detachments in both eyes. HLA Class II typing revealed the haplotype associated with sympathetic ophthalmia and Vogt-Koyanagi-Harada (VKH). The inflammation improved after corticosteroid, mycophenolate mofetil, and adalimumab therapy while vision remained poor. Conclusions and Importance: Surgically-induced sympathetic ophthalmia is a plausible explanation for the clinical findings; surgery of both eyes within one week would conceal the inciting eye. VKH or inflammation related to the gene therapy are other possible etiologies but severe bilateral panuveitis has not been reported with voretigene neparvovec-rzyl. Informed consent for gene therapy surgery should include a discussion of the rare complication of sympathetic ophthalmia following vitrectomy surgery.http://www.sciencedirect.com/science/article/pii/S2451993623000877Subretinal gene therapyLeber congenital amaurosisVogt-koyanagi-harada diseaseSympathetic ophthalmiaRPE65-Associated inherited retinal diseasePanuveitis |
| spellingShingle | Alcina K. Lidder Stephanie Choi Yasha S. Modi Scott E. Brodie Janet L. Davis Ninel Z. Gregori Byron L. Lam Bilateral exudative retinal detachments after subretinal gene therapy with voretigene neparvovec-rzyl for RPE65 Leber Congenital Amaurosis American Journal of Ophthalmology Case Reports Subretinal gene therapy Leber congenital amaurosis Vogt-koyanagi-harada disease Sympathetic ophthalmia RPE65-Associated inherited retinal disease Panuveitis |
| title | Bilateral exudative retinal detachments after subretinal gene therapy with voretigene neparvovec-rzyl for RPE65 Leber Congenital Amaurosis |
| title_full | Bilateral exudative retinal detachments after subretinal gene therapy with voretigene neparvovec-rzyl for RPE65 Leber Congenital Amaurosis |
| title_fullStr | Bilateral exudative retinal detachments after subretinal gene therapy with voretigene neparvovec-rzyl for RPE65 Leber Congenital Amaurosis |
| title_full_unstemmed | Bilateral exudative retinal detachments after subretinal gene therapy with voretigene neparvovec-rzyl for RPE65 Leber Congenital Amaurosis |
| title_short | Bilateral exudative retinal detachments after subretinal gene therapy with voretigene neparvovec-rzyl for RPE65 Leber Congenital Amaurosis |
| title_sort | bilateral exudative retinal detachments after subretinal gene therapy with voretigene neparvovec rzyl for rpe65 leber congenital amaurosis |
| topic | Subretinal gene therapy Leber congenital amaurosis Vogt-koyanagi-harada disease Sympathetic ophthalmia RPE65-Associated inherited retinal disease Panuveitis |
| url | http://www.sciencedirect.com/science/article/pii/S2451993623000877 |
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