Objectives: To describe the surgical management of a series of cases of gonococcal keratitis from the Indian subcontinent. This is a retrospective, observational case series undertaken in a national tertiary referral centre, Ispahani Islamia Eye Institute and Hospital (IIEIH), Dhaka, Bangladesh.

Methods: Three patients were treated for corneal perforation secondary to gonococcal keratitis over a period of seventeen months. We discuss our surgical management and final visual outcomes.

Results: Our cohort consisted of six eyes from three patients, five of which ultimately perforated after medical treatment. Surgical management consisted of large 12mm tectonic Penetrating Keratoplasty (PK) in one eye, conjunctival hood fashioning in one eye and tectonic grafts from residual sclerocorneal buttons in the remaining eyes. At final follow up, 60% of eyes achieved Corrected Distance Visual Acuity (CDVA) better than Logarithm of

the Minimum Angle of Resolution (logMAR) 0.8.

Conclusions: We present various surgical methods for the management of patients with severe gonococcal keratitis. With access to fresh donor corneal tissue the use of conjunctival hood and sclerocorneal buttons are invaluable adjuncts when managing cases of this potentially devastating condition.

Neisseria gonorrhoeae is a gram-negative diplococcus with the propensity to penetrate intact corneal epithelium, resulting in perforation and fulminant intraocular disease [1-5]. Visual morbidity results from corneal ulceration and/or perforation and scarring and endophthalmitis. Schwab et al., have suggested that, in Bangladesh, which is prone to flooding in monsoon season because of its sea level location, flood waters act as reservoirs for the transmission of Neisseria gonorrhoea. This has however not been proven to date. Prompt diagnosis and medical treatment in the early phase is critical to achieve resolution, however this is rarely achievable due to obstacles including patients withholding sexual histories and genito-urinary symptoms. Furthermore, even a correct diagnosis does not equate to cessation of disease propagation, due to the emergence of antibiotic resistant strains [6-11]. The Ophthalmologist should thus be prepared to encounter advanced disease and have at their disposal an arsenal of surgcal techniques to use as required.

In Bangladesh the availability of fresh donor corneal tissue is limited. Consequently, we have developed alternative techniques for both the emergent and definitive management of keratitis secondary to Neisseria gonorrhoeae. Here we report the surgical management of five such cases.

We describe three consecutive patients referred to the tertiary centre Ispahani Islamia Eye Institute and Hospital (IIEIH) with gonoccocal keratitis related corneal perforation, over a period of seventeen months. All our patients were male, between the ages of 18 and 21 years (mean age 20.0 years). All patients had been misdiagnosed with EKC in other hospitals and were prescribed topical antibiotics. Diagnosis in our institute was confirmed using culture after conjunctival swab and corneal scrape. All patients required surgical intervention and were followed up for fifteen months. Postoperatively antibiotic and anti-hypertensiveve drops were tapered according to clinical response. Furthermore, a topical steroid was given in every case to prevent further melt and graft rejection.

A 21-year old fisherman was referred to IIEIH with a tenday history of mucopurulent discharge and lid swelling. He had been commenced on topical ciprofloxacin 0.3% by his local ophthalmologist six days prior to presenting to us. His Corrected Distance Visual Acuity (CDVA) was right eye Counting Fingers (CF) and left eye Perception of Light (PL). Examination showed bilateral marked eyelid oedema, severe haemorhhagic chemosis and preauricular lymphadenopathy. The left eye showed a nasal corneal perforation secondary to a fulminant peripheral melt, with a completely flat Anterior Chamber (AC) and fibrinous reaction (Figure 1a). The right eye had a supero-temporal furrow, with copious amount of purulent discharge (Figure 1b). A gram stain of the eye exudate showed gram-negative diplococci and cultures yielded penicillin resistant Neisseria gonorrhoeae. The patient was immediately admitted and administered 1g intravenous ceftriaxone daily, oral doxycycline 100mg b.d, topical moxifloxacin 0.5% hourly, atropine 1% o.d. and normal saline qds. Day three post-admission, the left eye had proceeded to complete perforation and complete iris exposure (Figure 1c). The right eye had localised thinning supero-temporally, but the AC was formed (Figure 1d). Hence the decision was taken to perform a large 12mm tectonic PK in the left eye, with a temporary tarsorraphy for persistent epithelial defect, and conjunctival hood fashioning in the right eye (Figure 1e, 1f). In our institute, conjunctival hood formation is a common surgical procedure used when corneal thinning and non-responsive infective processes are apparent. We fashion a superior conjunctival flap, ensuring a meticulous tenectomy is performed. A limited inferior peritomomy is performed. The free end of the superior flap is brought over the area of the cornea intended to be covered and is sutured into place where the inferior peritomy was created. At the limbal area, we take great care to ensure episcleral bites are taken for added stability. All sutures used are 10.0 nylon. The patient responded very favourably to the surgery and we displaced the conjunctival hood after ten days, with the graft in the contralateral eye remaining clear (Figure 1g, 1h). At final follow-up of 15 months, his CDVA was logMAR 0.2 in the right eye and logMAR 0.8 in the left eye (Figure 1i, 1j).

An 18-year old rickshaw driver presented to us three days after being started on topical ciprofloxacin 0.3% by a different institute. Both eyes were reduced to CF CDVA, with copious discharge and extreme pain. A gram stain of the eye exudate showed gram-negative diplococci and cultures yielded penicillin-resistant gonococci. The patient was immediately admitted and administered 1g intravenous ceftriaxone daily, oral doxycycline 100 mg b.d, topical moxifloxacin 0.5% hourly, atropine 1% o.d. and normal saline qds. Unfortunately, bilateral perforation necessitated surgical intervention (Figure 2a, 2b). We performed a corneoscleral keratoplasty in both eyes (Figure 2c, 2d). At final follow-up of 12 months, his CDVA was logMAR 0.3 in the right eye and logMAR 1.0 in the left eye (Figure 2e, 2f).

The patient chose not to pursue regrafting in the left eye for visual rehabilitation.

A 21-year old student presented after increasing discharge and pain from both eyes. Three weeks previously, he had been treated with a course of oral ciprofloxacin 500mg b.d. for purulent urethral discharge, following previous unprotected sexual intercourse with a female. Two weeks following this, he presented to an ophthalmologist elsewhere with chemosis, purulent discharge and profuse matting of the lashes. Both eyes were reduced to CDVA CF (Figure 3a, 3b). A gram stain of the exudate showed gram-negative diplococci and cultures yielded penicillinresistant gonococci. The patient was immediately admitted and administered 1g intravenous ceftriaxone daily, oral doxycycline 100mg b.d, topical moxifloxacin 0.5% hourly, atropine 1% o.d. and normal saline qds. Five days after admission the situation had improved somewhat, but the right eye still required a patch graft (Figure 3c-3e). At final follow-up, the patient had CDVA logMAR 1.0 in both eyes (Figure 3f). Table 1 summarises patient demographics, surgical management, and visual outcomes.

The incidence of gonococcal infection is increasing worldwide [6-8,10,12-14]. The demographics of the patients in our study represent the patients affected globally in as much as NG associated keratoconjunctivitis is primarily a disease of young men. One should, however be suspicious of gonococcal infection in any sexually active individual [15]. What is for certain is that this is a potentially devastating condition whereby intact epithelium does not preclude to corneal perforation within 24 hours [16-18].

To the best of our knowledge, this is the largest reported, single centre consecutive case series of surgical management of gonococcal keratitis from the Indian subcontinent. We have described the importance of thorough sexual histories (no matter how reluctant the patient is to disclose this information due to cultural barriers), identifying the pathogen early, daily clinical assessment and prompt surgical intervention. Bangladesh is reliant on corneal grafts being delivered from overseas at infrequent intervals and as such the option of fresh corneas for both therapeutic and visual rehabilitative purposes is limited. Hence IIEIH has developed other techniques to manage the full range of perforations, from the conjunctival hood formation for micro-perforations to using preserved full thickness corneoscleral buttons for tectonic grafts. As we have shown, patients can have excellent outcomes following such surgeries (60% of patients had CDVA better than logMAR 0.8) and if required secondary procedures can be offered for visual purposes.

Adherence to local guidelines and strict liaison with microbiologists are key in the initial treatment of patients with gonococcal keratitis. Performing a scrape and ensuring sensitivities are sought is mandatory due to the emergence of multidrug resistant strains, specifically to fluoroquinolones, tetracyclines and penicillins [6-9,12,15]. All three patients in our series were scraped and found to be sensitive to ciprofloxacin and ceftriaxone. However, due to the late presentation, despite prompt admission and strict administration of the correct antibiotic, progression to perforation still occurred. It is imperative to do surgery immediately in order to control residual infection and restore the integrity of the globe. Various options exist, depending on the type of perforations encountered, clinical expertise, availability of human tissue and equipment. Lamellar tectonic keratoplasties are an excellent option in the event of severe thinning, very localised perforation or descemetocele formation. The rationale for using lamellar keratoplasty over penetrating is to reduce the risk of rejection, endophthalmitis, and secondary glaucoma. Kestelyn et al., described a series of six patients over a period of fourteen months in a Rwandan population [5]. One patient had a delay in initiating antibiotics and went on to have a descemetocele with subsequent lamellar corneal graft within three days [5]. At final follow-up, CDVA was logMAR 0.3 and the eye was devoid of inflammation [5]. Tong et al., described deep anterior lamellar keratoplasty as a technique to treat a severe but non-perforated corneal melt in a 25-year old Malay man with gonococcal keratitis [19]. At five months, CDVA was logMAR 0.1.Day et al., reported a large limbal-lamellar tectonic patch graft in a HIV positive patient with superior corneal melt secondary to gonococcal keratitis [20]. At 2 months, the CDVA was logMAR 1.0 with a clear graft [20]. Tipple et al., presented a 25 year-old homosexual male presenting with gonococcal keratitis as the first presentation of HIV infection [21]. Seven days after intense medical treatment, the patient underwent a lamellar tectonic graft, resulting in CDVA logMAR 0.5 at final follow-up [21]. Kawashima et al. described five consecutive cases in Japanese patients of corneal perforations secondary to gonococcal keratitis all being managed by therapeutic keratoplasties, either lamellar or penetrating using cryopreserved grafts [22]. Mean visual outcome at thirty-five months was logMAR 0.0, with no major complications such as graft failure [22]. Whilst lamellar keratoplasties are important to bear in mind, all of our patients presented with advanced perforations, hence there was no role for the use of this technique. 

A more plausible and practical option for Countries with scant availability to cornea donors is corneoscleral patch grafts from preserved buttons. In a landmark paper, Schwab et al., reported a seasonal outbreak of gonococcal keratitis in thirty-two eyes corresponding to the heavy rainy season in Malawi [2]. Ten of these eyes proceeded to perforation, necessitating scleral patch grafts in six [2]. The post-operative visual outcomes in these six patients ranged from logMAR 0.6 to 1.5 [2]. Guerrero et al. describe a thirty-three-year-old homosexual man presenting with a 360-degree peripheral melt, involving 90% of corneal thickness [15]. Despite a degree of stabilisation with conservative management including a bandage contact lens, the patient underwent a twelve mm diameter full thickness penetrating keratoplasty one-week later [15]. At one-month follow-up, his visual acuity was logMAR 0.5 [15]. To our knowledge, no direct reports in the literature exist discussing the management of micro-perforations secondary to gonococcal keratitis with conjunctival hood procedures, although as shown in this case series a role for this technique certainly exists. 

To summarise, always be suspicious and fearful of gonococcal keratitis. Explore the sexual history in detail and obtain conjunctival swabs and corneal scrapes as necessary. Prompt diagnosis and treatment is the only way to ensure optimal ocular recovery, with many surgical options available in advanced cases.

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Figure 1a: Anterior segment photograph of the left eye of case 1, showing a nasal corneal perforation secondary to a fulminant peripheral melt.

Figure 1b: Anterior segment photograph of the right eye of case 1, showing a supero-temporal ring furrow and copious purulent discharge.

Figure 1c: Day 3 post admission and treatment with intra-venous ceftriaxone. Complete iris exposure can be seen.

Figure 1d: Day 3 post admission and treatment with intra-venous ceftriaxone. There is localised thinning supero-temporally, but the AC is formed.

Figure 1f: Day 1 post conjunctival hood formation: note the hinge of the flap was created supero-nasally and brought across for fixation infero-temporally.

Figure 1g: Day 10 post- tectonic PK surgery. Inflammatory reaction settling and graft are clear.

Figure 1h: Day 10 post conjunctival hood formation: this has now been displaced as the ulcer has healed.

Figure 2a: Anterior segment photograph of the left eye of case 2, showing complete corneal perforation.

Figure 2b: Anterior segment photograph of the right eye of case 2, showing superior corneal perforation.

Figure 3a: Anterior segment photograph of the right eye of case 3, showing copious purulent discharge obscuring the underlying corneal perforation.