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Secondary glaucoma in CAPN5-associated neovascular inflammatory vitreoretinopathy.

Cham A, Bansal M, Banda HK, Kwon Y, Tlucek PS, Bassuk AG, Tsang SH, Sobol WM, Folk JC, Yeh S, Mahajan VB - Clin Ophthalmol (2016)

Bottom Line: Patient 3 did not develop steroid-response glaucoma in either eye.The FA implants have reduced uveitic and neovascular glaucoma.Nevertheless, IOP management remains complex due to steroid-response glaucoma, angle closure glaucoma, and hypotony.

View Article: PubMed Central - PubMed

Affiliation: Department of Ophthalmology and Visual Sciences; Omics Laboratory, University of Iowa, Iowa City, IA, USA.

ABSTRACT

Objective: The objective of this study was to review the treatment outcomes of patients with secondary glaucoma in cases of autosomal dominant neovascular inflammatory vitreoretinopathy (ADNIV), a hereditary autoimmune uveitis due to mutations in CAPN5.

Patients and methods: A retrospective, observational case series was assembled from ADNIV patients with secondary glaucoma. The main outcome measures were intraocular pressure (IOP), visual acuity, use of antiglaucoma medications, ocular surgeries, and adverse outcomes. Perimetry and optic disk optical coherence tomography (OCT) were also analyzed.

Results: Nine eyes of five ADNIV patients with secondary glaucoma were reviewed. Each received a fluocinolone acetonide (FA) implant for the management of posterior uveitis. Following implantation, no eyes developed neovascular glaucoma. Five eyes (in patients 1, 2, and 5) required Ahmed glaucoma valve surgery for the management of steroid-responsive glaucoma. Patient 2 also developed angle closure with iris bombe and underwent laser peripheral iridotomy. Patient 4 had both hypotony and elevated IOP that required periodic antiglaucoma medication in the FA-implanted eye. Patient 3 did not develop steroid-response glaucoma in either eye. Optic disk examinations were obscured by fibrosis and better assessed with OCT.

Conclusion: ADNIV patients show combined mechanism secondary glaucoma best assessed by OCT of the optic disk. The FA implants have reduced uveitic and neovascular glaucoma. Nevertheless, IOP management remains complex due to steroid-response glaucoma, angle closure glaucoma, and hypotony.

No MeSH data available.


Related in: MedlinePlus

Patient 1.Notes: (A) OD optic disk image shows vertically oval cup with INF thinning of the NRR. (B) OS optic disk image with media haze and diffuse disk pallor. (C) OD Goldmann perimetry with diffuse constriction, SUP > INF. (D) OS Goldmann perimetry shows remaining island of central and inferotemporal visual field. (E) Stratus OCT RNFL thickness analysis OD shows no RNFL loss. (F) OS RNFL quadrant and analysis show severe losses (<1% of normative data) in the NAS and TEMP quadrants. OS RNFL clock hour analysis corroborates RNFL thickness findings. OD RNFL quadrant and clock hour analyses were both normal.Abbreviations: INF, inferior; NRR, neuroretinal rim; SUP, superior; OCT, optical coherence tomography; RNFL, retinal nerve fiber layer; NAS, nasal; TEMP, temporal; NA, not applicable; S, superior; N, nasal; T, temporal; I, inferior.
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f1-opth-10-1187: Patient 1.Notes: (A) OD optic disk image shows vertically oval cup with INF thinning of the NRR. (B) OS optic disk image with media haze and diffuse disk pallor. (C) OD Goldmann perimetry with diffuse constriction, SUP > INF. (D) OS Goldmann perimetry shows remaining island of central and inferotemporal visual field. (E) Stratus OCT RNFL thickness analysis OD shows no RNFL loss. (F) OS RNFL quadrant and analysis show severe losses (<1% of normative data) in the NAS and TEMP quadrants. OS RNFL clock hour analysis corroborates RNFL thickness findings. OD RNFL quadrant and clock hour analyses were both normal.Abbreviations: INF, inferior; NRR, neuroretinal rim; SUP, superior; OCT, optical coherence tomography; RNFL, retinal nerve fiber layer; NAS, nasal; TEMP, temporal; NA, not applicable; S, superior; N, nasal; T, temporal; I, inferior.

Mentions: A 49-year-old man with ADNIV was evaluated of recurrent uveitis and elevated IOP. He carried the CAPN5 p.L244P mutation. His uveitis was managed with topical steroids and subtenon’s triamcinolone acetate injections for 20 years. On examination, his best-corrected visual acuity (BCVA) was 20/50 OD and 20/1,000 OS. He was functionally monocular. His eye pressure measured as high as 45 mmHg OD and 25 mmHg OS, and it was refractory to a maximum treatment of dorzolamide–timolol, brimonidine, pilocarpine, and latanoprost. He could not tolerate methazolamide (oral carbonic anhydrase inhibitor). On anterior segment examination, his corneal thickness was 556 µm OD and 592 µm OS, and there was 2+ flare OU. On right eye gonioscopy, the angle was open with visible scleral spur. The angular approach was 40°, and the iris was flat with 1+ pigmentation of the trabecular meshwork. In the left eye, both the scleral spur and ciliary body were visible and the angular approach was 35° with 1+ pigmentation of the trabecular meshwork. He was pseudophakic OU. The posterior segment exam showed 2+ vitreous cells OU, vitreous opacities, and a 3+ ERM OD and 1+ ERM OS. He also had significant cystoid macular edema (CME) and inferior thinning of the optic disk OD (Figure 1A). He had significant media opacity, due to intraocular fibrosis and tractional retinal detachment, which obscured view of his optic disk OS (Figure 1B). There was 360° of diffuse pigmentary clumping OU without any retinal tears or detachment OD. These findings were consistent with early stage III ADNIV OD and stage IV ADNIV OS.


Secondary glaucoma in CAPN5-associated neovascular inflammatory vitreoretinopathy.

Cham A, Bansal M, Banda HK, Kwon Y, Tlucek PS, Bassuk AG, Tsang SH, Sobol WM, Folk JC, Yeh S, Mahajan VB - Clin Ophthalmol (2016)

Patient 1.Notes: (A) OD optic disk image shows vertically oval cup with INF thinning of the NRR. (B) OS optic disk image with media haze and diffuse disk pallor. (C) OD Goldmann perimetry with diffuse constriction, SUP > INF. (D) OS Goldmann perimetry shows remaining island of central and inferotemporal visual field. (E) Stratus OCT RNFL thickness analysis OD shows no RNFL loss. (F) OS RNFL quadrant and analysis show severe losses (<1% of normative data) in the NAS and TEMP quadrants. OS RNFL clock hour analysis corroborates RNFL thickness findings. OD RNFL quadrant and clock hour analyses were both normal.Abbreviations: INF, inferior; NRR, neuroretinal rim; SUP, superior; OCT, optical coherence tomography; RNFL, retinal nerve fiber layer; NAS, nasal; TEMP, temporal; NA, not applicable; S, superior; N, nasal; T, temporal; I, inferior.
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Related In: Results  -  Collection

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f1-opth-10-1187: Patient 1.Notes: (A) OD optic disk image shows vertically oval cup with INF thinning of the NRR. (B) OS optic disk image with media haze and diffuse disk pallor. (C) OD Goldmann perimetry with diffuse constriction, SUP > INF. (D) OS Goldmann perimetry shows remaining island of central and inferotemporal visual field. (E) Stratus OCT RNFL thickness analysis OD shows no RNFL loss. (F) OS RNFL quadrant and analysis show severe losses (<1% of normative data) in the NAS and TEMP quadrants. OS RNFL clock hour analysis corroborates RNFL thickness findings. OD RNFL quadrant and clock hour analyses were both normal.Abbreviations: INF, inferior; NRR, neuroretinal rim; SUP, superior; OCT, optical coherence tomography; RNFL, retinal nerve fiber layer; NAS, nasal; TEMP, temporal; NA, not applicable; S, superior; N, nasal; T, temporal; I, inferior.
Mentions: A 49-year-old man with ADNIV was evaluated of recurrent uveitis and elevated IOP. He carried the CAPN5 p.L244P mutation. His uveitis was managed with topical steroids and subtenon’s triamcinolone acetate injections for 20 years. On examination, his best-corrected visual acuity (BCVA) was 20/50 OD and 20/1,000 OS. He was functionally monocular. His eye pressure measured as high as 45 mmHg OD and 25 mmHg OS, and it was refractory to a maximum treatment of dorzolamide–timolol, brimonidine, pilocarpine, and latanoprost. He could not tolerate methazolamide (oral carbonic anhydrase inhibitor). On anterior segment examination, his corneal thickness was 556 µm OD and 592 µm OS, and there was 2+ flare OU. On right eye gonioscopy, the angle was open with visible scleral spur. The angular approach was 40°, and the iris was flat with 1+ pigmentation of the trabecular meshwork. In the left eye, both the scleral spur and ciliary body were visible and the angular approach was 35° with 1+ pigmentation of the trabecular meshwork. He was pseudophakic OU. The posterior segment exam showed 2+ vitreous cells OU, vitreous opacities, and a 3+ ERM OD and 1+ ERM OS. He also had significant cystoid macular edema (CME) and inferior thinning of the optic disk OD (Figure 1A). He had significant media opacity, due to intraocular fibrosis and tractional retinal detachment, which obscured view of his optic disk OS (Figure 1B). There was 360° of diffuse pigmentary clumping OU without any retinal tears or detachment OD. These findings were consistent with early stage III ADNIV OD and stage IV ADNIV OS.

Bottom Line: Patient 3 did not develop steroid-response glaucoma in either eye.The FA implants have reduced uveitic and neovascular glaucoma.Nevertheless, IOP management remains complex due to steroid-response glaucoma, angle closure glaucoma, and hypotony.

View Article: PubMed Central - PubMed

Affiliation: Department of Ophthalmology and Visual Sciences; Omics Laboratory, University of Iowa, Iowa City, IA, USA.

ABSTRACT

Objective: The objective of this study was to review the treatment outcomes of patients with secondary glaucoma in cases of autosomal dominant neovascular inflammatory vitreoretinopathy (ADNIV), a hereditary autoimmune uveitis due to mutations in CAPN5.

Patients and methods: A retrospective, observational case series was assembled from ADNIV patients with secondary glaucoma. The main outcome measures were intraocular pressure (IOP), visual acuity, use of antiglaucoma medications, ocular surgeries, and adverse outcomes. Perimetry and optic disk optical coherence tomography (OCT) were also analyzed.

Results: Nine eyes of five ADNIV patients with secondary glaucoma were reviewed. Each received a fluocinolone acetonide (FA) implant for the management of posterior uveitis. Following implantation, no eyes developed neovascular glaucoma. Five eyes (in patients 1, 2, and 5) required Ahmed glaucoma valve surgery for the management of steroid-responsive glaucoma. Patient 2 also developed angle closure with iris bombe and underwent laser peripheral iridotomy. Patient 4 had both hypotony and elevated IOP that required periodic antiglaucoma medication in the FA-implanted eye. Patient 3 did not develop steroid-response glaucoma in either eye. Optic disk examinations were obscured by fibrosis and better assessed with OCT.

Conclusion: ADNIV patients show combined mechanism secondary glaucoma best assessed by OCT of the optic disk. The FA implants have reduced uveitic and neovascular glaucoma. Nevertheless, IOP management remains complex due to steroid-response glaucoma, angle closure glaucoma, and hypotony.

No MeSH data available.


Related in: MedlinePlus