Neovascular Proliferation over a Cosmetic Artificial Iris Implant Open Access

Iris implants were initially introduced to address patients with large acquired or congenital defects. The idea of using these devices to alter iris color has gained widespread acceptance, leading to the production of cosmetic iris implants for this purpose [1]. The first device, named NewColorIris (Kahn Medical Devices, Panama City, Panama), was developed in Panama in 2006, followed by a new generation, BrightOcular, created in 2012 by a US company (Stellar Devices, New York, USA). These devices are associated with numerous complications, some of which pose significant risks. Commonly reported consequences include the onset of glaucoma, endothelial dysfunction, cataract formation, and iris abnormalities such us iris defects, corectopia, anterior and posterior synechiae, often necessitating surgical intervention [2]. Here, we present a unique case involving neovascularization of the angle originating from the iris strands, resulting in neovascular glaucoma subsequent to a combined arterial and venous vascular occlusion. The CARE Checklist has been completed by the authors for this case report and attached as supplementary material (for all online suppl. material, see https://doi.org/10.1159/000546599).

A 28-year-old woman, who had undergone bilateral cosmetic iris implantation (BrightOcular, Stellar Devices, New York, USA) 6 years prior in Tunisia, presented as an emergency case to the Sant’Orsola-Malpighi University Hospital of Bologna (Italy), complaining of bilateral blurry vision and ocular pain, that had started few days earlier. Until that moment, she had not noticed any changes in her vision. During the initial examination, her best corrected visual acuity was hand motion in the right eye and 20/100 in the left eye. Intraocular pressure (IOP), measured with a Goldmann applanation tonometer, was 45 mm Hg in the right and 30 mm Hg in the left eye. Slit-lamp examination revealed pronounced conjunctival hyperemia, bilateral corneal edema, deep anterior chamber, artificial iris implants with natural iris strands in the pupil area, posterior subcapsular cataract in the right eye, and a clear lens in the left eye (Fig. 1).

Gonioscopy revealed iris root atrophy with remnants of the peripheral iris impinging on the trabecular meshwork and angular recession in both eyes. Fundus examination revealed advanced glaucomatous optic disc cupping in the right eye and moderate excavation in the left eye. Moreover, macular OCT showed widespread retinal atrophy in the right eye, complete disorganization of the retinal layers, and profound retinal vascular ischemia, suggestive of previous retinal vascular occlusion. In the left eye, the only atypical finding was the dilation and tortuosity of the temporal branches of the central retinal vein. We performed fluorescein angiography, which showed hypofluorescence of the optic disk in the right eye and mild hyperfluorescence in the left eye. However, due to the presence of the prosthesis, it was not possible to accurately evaluate peripheral retina. RNFL thickness evaluation on the OCT revealed diffuse reduction of the fibers in the right optic nerve, while in the left eye, fibers were well conserved. Finally, we performed specular microscopy, which revealed a reduced endothelial cell count (1,179 cells/mm³ and 1,213 cells/mm³ in right and left eye, respectively) in both eyes.

Since medical therapy with prostaglandin analogs, carbonic anhydrase inhibitors, and beta-blockers failed to control IOP, we opted for sequential bilateral Baerveldt (Abbott Laboratories, Abbott Park, IL, USA) glaucoma valve implantation, resulting in the normalization of IOP. One week post-surgery, a repeated macular OCT revealed the development of widespread macular and retinal edema in the right eye. In the left eye, diffuse hemorrhagic spots were observed in the posterior pole, along with intraretinal exudates, an initial epiretinal membrane, a small subfoveal neuroepithelial detachment, and vitritis. Slight chorioretinal folds were also visible, suggesting hypotensive maculopathy (Fig. 2), with an IOP of 6 mm Hg. Assuming possible transient vascular and inflammatory damage due to surgery, we prescribed topical and systemic anti-inflammatory therapy with dexamethasone 0.1% and tobramycin 0.3% eye drops six times daily in left eye and systemic indomethacin 100 mg BID. After 1 month, macular OCT revealed resolution of edema in the right eye, restoration of foveal profile, resolution of hemorrhages, exudates, and chorioretinal folds in the left eye. Visual acuity in the right eye remained unchanged, while in the left eye, it improved to 20/25.

Despite these improvements, 2 months after surgery, the patient presented with inferior angle neovascularization in the right eye, growing above the artificial implant and almost reaching the pupillary opening. Intraocular impairment was still well controlled; however, it was important to prevent IOP impairment considering the presence of diffuse neovessels of the angle. Therefore, three intravitreal injections of bevacizumab, administered at 1-month intervals, were administered to inhibit further growth of these pathologic vessels, which regressed almost completely after 3 months (Fig. 3). Six months after the last injection, IOP was maintained within normal limits in both eyes, endothelial cell density was still approximately 1,200 cells/mm² (1,168 cells/mm³ and 1,198 cells/mm³ in right and left eye, respectively), and best corrected visual acuity was hand motion and 20/25 in the left eye.

At present, we chose not to move forward with cataract surgery and PPV because, following the implantation of the Baerveldt valve and anti-VEGF therapy, we successfully controlled IOP, and the patient did not experience ocular pain. Proceeding with the surgery might have compromised IOP control without offering any improvement in visual acuity, especially considering the right eye’s limited visual potential. Furthermore, we had to consider the patient’s fragile psychological state, given her heightened sensitivity to the cosmetic aspect of the eye, which had already developed an outward deviation. She had undergone psychological therapy for approximately a year. Introducing an in-the-bag iris+IOL prosthesis could have further influenced the eye’s appearance, potentially adversely affecting her mental well-being.

Cosmetic iris implants were introduced in Panama in 2006. Complications associated with the use of first-generation cosmetic iris implants are well known, and extensive descriptions of them can be found in the literature [2‒7]. The second generation of these devices, marketed under the name of BrightOcular implants, aimed to eliminate the risks associated with their implantation by introducing innovations in implant design. Nevertheless, there is now a great deal of evidence that they are no safer than their predecessors.

Our patient presented with almost all the most common clinical issues associated with these implants, with a preponderance of secondary glaucoma manifestations. Several mechanisms may be involved in this increase in IOP. First, the presence of the iris implant causes direct angle obstruction, thus inducing chronic angle-closure glaucoma. Second, continuous rubbing of the implant on the natural iris induces pigment dispersion, responsible for trabecular meshwork obstruction, consequently creating an obstacle to aqueous humor outflow [8]. Moreover, gonioscopy revealed angle recession, known to be associated with IOP increase, most probably because of indirect trabecular damage [9].

In our patient, long-standing ocular hypertension was responsible for serious repercussions, mostly in the optic nerve and retinal vasculature. OCT provided an image of a deeply damaged retina in the right eye, diffuse atrophy and impairment of retinal layers, and profound retinal ischemia, suggestive of combined central retinal vein and artery occlusion. Advanced glaucoma is an important risk factor for venous thrombosis formation [10], eventually leading to arterial obstruction by back pressure and combined arterial and venous occlusion [11]. This condition is characterized by clinical findings typical of both central retinal artery occlusion and central retinal vein occlusion at presentation, such as optic disc edema and hyperemia, retinal hemorrhage and edema, and varying degrees of retinal ischemic whitening. During follow-up, most of these eyes developed a pale and atrophic optic disc, retinal ischemia, and alterations in retinal circulation [12], similar to what we found in our patient.

On the other hand, the left eye presented peculiarities of venous impairment, such as dilation and tortuosity, suggestive of a pre-vascular occlusion. However, the retinal layers were well represented, and the foveal anatomy was conserved. After Baerveldt tube implantation in the left eye, the patient developed slight optic disc swelling, diffuse hemorrhages at the posterior pole, retinal thickening, hyperreflectivity of the internal retinal layers, a small subfoveal serous neuroepithelial detachment, and retinal and choroidal folds radiating from the fovea. The clinical picture was suggestive of hypotony maculopathy, which could explain most of the findings. Moreover, previous ocular hypertension and the relatively young age of the patient have been found to be important prerequisites for the development of this condition due to the greater elasticity and flexibility of the sclera of younger patients compared to older patients [13]. Macular edema is an atypical finding, although it can rarely develop due to the high hydrostatic pressure gradient across the retinal vasculature that allows fluid to pass into extracellular spaces [14]. The condition usually resolves when the IOP returns to normal values, which occurred spontaneously in our patient.

We hypothesized that the ischemic insult to the retina triggered the production of angiogenic mediators, which induced the formation of new pathologic vessels in the anterior chamber, probably starting from the natural iris residual strands in the angle, similar to what occurs during neovascular glaucoma following central retinal vein occlusion [15]. Nevertheless, it was quite unpredictable whether they could grow and extend above an inert material, such as an iris prosthesis. Moreover, because of the presence of a small artificial pupil and dense posterior subcapsular cataract, it was impossible to perform panretinal photocoagulation, making it even more difficult to control the complications resulting from retinal impairment. Therefore, after considering the most likely pathogenesis, we decided on treatment with anti-angiogenic drugs in order to reduce the abnormal vascularization, thus trying to preserve the eye’s anatomy as much as possible and prevent the onset of phthisis bulbi.

To our knowledge, this is the first case describing neovascularization growing and extending above an inert material, in this case, an iris prosthesis. In conclusion, there are few reports of long-term follow-up of BrightOcular implants, although to date the evidence clearly demonstrates how dangerous they are. Therefore, it is essential to disseminate detailed and exhaustive information among the general population to apprise potential consumers of the risks associated with these implants.

This study was reviewed and approved by the Institutional Review Board of Area Vasta Emilia Centro (AVEC), approval No. 901/2022/Oss/AOUBo, and was performed in accordance with the tenets of the Declaration of Helsinki. Written informed consent was obtained from the patient for the publication of this case report and accompanying images.

The authors have no conflicts of interest to declare.

There was no funding for this article.

N.G. and M.R. wrote the full manuscript and edited the figure. A.M., D.I., and L.F. reviewed the manuscript.

All the data analyzed in this study are included in this article and its online supplementary material. Further inquiries can be directed to the corresponding authors.