Diagnosis, Workup and Treatment

Further investigation into the patient’s ocular history found that he had undergone the I-Brite eye-whitening procedure four years prior to presentation. Examination showed extensive scleral thinning in the left eye which did not have an overtly inflammatory appearance, however the presence of cell and flare in the anterior chamber suggested an active inflammatory process. The history and examination pointed to a diagnosis of scleromalacia with active inflammation following the I-Brite procedure. Additionally, the progression of scleral thinning with active inflammation showed an incomplete response to topical steroid therapy.

The patient’s topical steroid therapy was increased from four to six times per day in the left eye and aggressive lubrication was instituted in both eyes with artificial tears during the day and erythromycin ointment at night. Additionally, punctal plugs were placed into the lower eyelids bilaterally.

One region of sclera in the left eye had already been reinforced with a Tutoplast graft on presentation. The severe thinning of the temporal sclera made it likely that further grafting would be necessary if the patient continued to progress, however no further surgical intervention had been performed at the time of this publication.

In the presence of persistent intraocular inflammation and presumed active scleritis despite topical corticosteroid treatment, the patient was referred to the Uveitis Service at the Wills Eye Hospital for systemic workup of possible exacerbating conditions and consideration of systemic immunosuppression to further stabilize the eye.

Discussion

Eye-whitening procedures were pioneered in eastern Asia as a treatment for chronically hyperemic eyes, pingueculae and pigmentation. The basis of surgical eye-whitening technique involves extensive conjunctival resection (wide conjunctivectomy) and tenonectomy with topical mitomycin-C. Variations on the technique have included the use of bevacizumab injections.1 Domestically, the I-Brite eye-whitening system is a proprietary version of these techniques employing conjunctivectomy and MMC. The subject matter has been a topic of much debate due to the concern for severe complications. While some papers have reported high patient satisfaction rates with low complication rates,2 others have found complications in as many as 82.9 to 91.7 percent of patients, over half of which were considered to be “severe.”1,3,4 The most common complications include fibroproliferation (43.8 percent); dry eyes (32 percent); ocular hyperemia (30.9 percent); and intraocular pressure elevation (13 percent). Severe complications include scleral calcification (6.2 percent); scleromalacia (4.4 percent); diplopia (3.6 percent); strabismus (1.5 percent); and keratitis (1.2 percent), with scleral necrosis being present in 0.1 percent of cases.1 Soolienah Rhiu and colleagues looked at incidence of scleral thinning, which was found to be as high as 43.8 percent. Additionally, persistent conjunctival epithelial defects were particularly common, found to be present in 45.8 percent of patients.4

It is often a relatively young population that seeks out these services, with recent studies showing average age ranging from 35.9 to 39.2 years old, with nearly two-thirds being female.1,4 This young population is at particular risk as complications may be chronic and worsen with time, leading to a lifetime of treatment. It has been suggested that nearly one-third of patients require a reoperation due to complications of eye-whitening procedures, most commonly due to fibroproliferation following the initial surgery.1

Editor's Note
The published version of this case report and an earlier online version contained a reference to an ASCRS Cornea Clinical Committee Alert regarding the safety of eye-whitening, and a link to the alert. ASCRS indicates that this was a one-time communication with its members and the Alert is no longer available online.

The initial report also stated that the Korean Ministry of Health and Welfare had banned the eye-whitening procedure in 2011. We have learned that in 2013, the Seoul High Court overturned the Ministry’s ban.
Necrotizing scleritis without inflammation, also known as scleromalacia perforans, is most commonly associated with rheumatoid arthritis; however, there is a steady flow of cases showing scleromalacia following conjunctivectomy with MMC in the setting of pterygium surgery or eye-whitening procedures.5,6 Individuals with a history of these procedures may not need to undergo the usual extensive laboratory and imaging workup for those with scleritis unless there is a history of prior systemic symptoms or suggestion of underlying rheumatologic disease. A thorough history prior to pursuing these procedures may help to decrease the incidence of postoperative complications in certain patients. Poor wound healing due to the use of MMC has been reported in individuals with acne rosacea, atopic keratoconjunctivitis, keratoconjunctivitis sicca, Sjögren’s syndrome, blepharitis and herpes keratitis.7

Therapeutic options for necrotizing scleritis secondary to eye-whitening procedures or pterygium excisions with MMC focus on minimizing inflammation and severity of scleritis, while treating scleral thinning to minimize risk for globe perforation. Therapeutics for inflammation can include topical steroids but may also include systemic immunosuppressants such as oral corticosteroids, antimetabolites, immunomodulators or cytotoxic agents. Treatments for scleral thinning may include scleral patch graft, amniotic membrane graft, lamellar corneoscleral graft or conjunctival flaps to reinforce areas of reduced integrity.8-10

The high incidence of severe complications from cosmetic eye-whitening procedures suggests that patients should likely be counseled against pursuing this procedure. Scrutiny of the procedure led the American Society of Cataract and Refractive Surgery to release a clinical alert warning providers about the risks associated with eye-whitening. Additionally, due to the concerning outcomes of eye-whitening surgeries, the Korean Ministry of Health and Welfare banned the procedure in 2011. Despite these warnings the procedure continues to be advertised to patients domestically.  



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