Indolent corneal ulcers in a patient with congenital insensitivity to pain with anhidrosis: a case report and literature review

PURPOSE: To report a case of bilateral corneal neurotrophic ulcer in patient with congenital insensitivity to pain with anhidrosis (CIPA) and review the literature. CASE REPORT: A 6 year-old boy presented with bilateral central corneal sterile ulcer, decreased corneal sensitivity, moderately altered corneal reflex and normal tearing response. History taken, systemic evaluation and medical chart review were undertaken. DISCUSSION: Fifty-two cases of CIPA have been reported worldwide. Fourteen cases had corneal involvement. The clinical picture of our patient is characteristic of CIPA. CONCLUSIONS: Congenital insensitivity to pain with anhidrosis may present as neurotrophic corneal ulcer. We report herewith, this vision threatening corneal congenital abnormality. Early diagnosis and prompt treatment are mandatory to prevent corneal complications such as scarring and perforation.     

Systemic tetracyclines in the treatment of noninfected corneal ulcers: a case report and proposed new mechanism of action

Corneal melting refers to a condition in patients whose corneas undergo progressive necrosis of stromal tissue despite being culture negative. Recent evidence has shown that systemic tetracyclines demonstrate anti-collagenolytic activity that may have clinical effectiveness in disease treatment. The evidence led us to try this modality as adjunct therapy in the treatment of an 80-year-old white man with a noninfected corneal ulcer. One gram of tetracycline in four divided doses led to a dramatic resolution of the corneal ulcer. This positive result supports the new concept that tetracycline therapy inhibits tissue collagenolytic enzyme activity by a mechanism unrelated to its antibacterial efficacy and may prove useful in the treatment of nonspecific corneal ulceration.     

Huge corneal dermoid in a well-formed eye: a case report and review of the literature

A 25-day-old boy presented with a left corneal mass and left nasal obstruction. The mass involved the entire cornea with a skin-like surface and protruded outside the palpebral fissure. CT of the orbits disclosed a large cyst coating the entire left cornea, in an eye with a well-formed anterior chamber and a clearly evident lens. CT also revealed left nasal meningo-encephalocele. The eye with the mass was excised. The histopathologic report confirmed the diagnosis of corneal dermoid in an otherwise normally developed eye. This report of a huge dermoid involving the entire corneal diameter and extending into the sclera without ocular alteration posterior to Descemet's membrane is the first such report in the literature. The literature on corneal dermoids is also reviewed.     

Ethmoid sinus osteoma with orbital invasion: report of three cases and review of the literature

We report three rare cases of ethmoid osteoma extending to the medial quadrant of the orbit that had singular particularities, such as occurring in women, at an unusual age group, and complaint of epiphora. The radiographic images were typical of this condition. Patients were submitted to surgery with complete resolution of the disease.     

Efficacy of topical and systemic itraconazole as a broad-spectrum antifungal agent in mycotic corneal ulcer. A preliminary study

PURPOSE: To evaluate the efficacy of topical (1%) and systemic itraconazole against common fungi such as Aspergillus and other filamentous fungi that cause mycotic corneal ulcer. METHODS: A prospective randomised, controlled study was done in 54 clinically suspected cases of fungal keratitis of which 44 were culture proven. Half the cases (n=27) with superficial involvement were treated with only topical itraconazole (1%) and the other half were treated with both topical and systemic itraconazole. RESULTS: Aspergillus, Penicillium and Fusarium were the most common fungi isolated. The ulcer resolved in 42 eyes (77%) and 12 eyes (23%) did not respond well to treatment. Four of 12 non-responding eyes were caused by Fusarium species. CONCLUSION: Itraconazole, given either topically or systemically, is effective in treating mycotic corneal ulcers.     

Myxomatous corneal degeneration: a clinicopathological study of six cases and a review of the literature

Thirteen cases with myxomatous changes of the corneal stroma have been reported to date. We report six additional cases with clinical, histopathological, and immunohistochemical data. The clinical appearance is most often a gelatinous, whitish elevation with insidious onset. Histopathologically, there are inconspicuous spindle- and stellate-shaped cells in a loose, myxoid matrix. The typical location is in the anterior cornea beneath the epithelium, with varying degrees of extension into the stroma. Vimentin and smooth-muscle actin immunohistochemical stains are characteristically positive, and staining occasionally may be seen with muscle-specific actin, whereas CD34 staining usually is negative. In most cases, myxomatous changes are a degenerative process involving transformation of stromal keratocytes into cells with prominent secretory activity and myofibroblastic differentiation. Most occur in corneas with a history of ocular disease or trauma that disrupts Bowman's layer. We suggest labelling lesions with these features as "myxomatous corneal degeneration." So-called "primary corneal myxomas" also exist where there is no significant history. It remains unclear whether the myxomatous changes in such lesions are neoplastic or degenerative. Myxomatous corneal changes are likely under-recognized and under-diagnosed.     

Presumed Candida endogenous fungal endophthalmitis: a case report and literature review.

BACKGROUND: Endogenous fungal endophthalmitis (EFE) is a rare intraocular infection that has recently increased in incidence over the last few decades. The most common causative organism of endogenous endophthalmitis is Candida albicans. Treatment generally is initiated with an antifungal drug systemically and, depending on the ocular findings, may also include a vitrectomy with the injection of an intraocular antifungal agent. Specific predisposing factors may make a patient more at risk for development of an ocular infection. CASE REPORT: We present a case of presumed Candida endogenous endophthalmitis in an 83-year-old white man with a Candida albicans urinary tract infection and describe the general characteristics, treatment, and management of this condition. CONCLUSION: Endogenous fungal endophthalmitis is a potentially blinding condition that can signal an underlying systemic infection. Without a dilated fundus examination in the presence of ocular inflammation, the condition easily can be misdiagnosed. Fungal infection may lead to retinal and vitreal lesions that can be visually debilitating if left untreated. Prognosis for visual outcome is dependent on timely diagnosis and initiation of treatment.     

Amniotic membrane transplantation in refractory neurotrophic corneal ulcers: a randomized, controlled clinical trial

PURPOSE: This study was designed to compare and evaluate the efficacy of amniotic membrane transplantation with the conventional management (tarsorrhaphy and bandage contact lens) in eyes with refractory neurotrophic corneal ulcers. METHODS: Thirty eyes of 30 patients (14 females and 16 males) with neurotrophic corneal ulcers refractory to medical management were included and divided randomly into group 1 (n = 15), who received conventional management with a tarsorrhaphy (n = 11) or bandage contact lens (n = 4), and group 2 (n = 15), who underwent Amniotic Membrane Transplantation. The outcome parameters evaluated were epithelialization time, duration of healing of corneal ulcers, and improvement in best corrected visual acuity. RESULTS: The mean age in our study was 37 +/- 14.71 years. At the end of 3 months follow-up, 10 of 15 patients (66.67%) in group 1 showed complete epithelialization and subsequent healing and 11 of 15 patients (73.33%) in group 2 showed complete epithelialization and healing (P > 0.05). The median time for complete epithelialization was 21 days in both groups. Both groups showed an improvement in the best-corrected visual acuity. CONCLUSIONS: Both amniotic membrane transplantation and conventional management (tarsorrhaphy or bandage contact lens) are effective treatment modalities for refractory neurotrophic corneal ulcers.     

Conjunctival lymphangiectasia: a report of 11 cases and review of literature

Conjunctival lymphangiectasia is an uncommon clinical condition in which there is dilatation of lymphatic channels in the bulbar conjunctiva. Conjunctival lymphangiectasia is a rarely appreciated ocular surface disorder that typically occurs as a secondary phenomenon in response to local lymphatic scarring or distal obstruction. Conjunctival lymphangiectasia can either be unilateral or bilateral with focal or diffuse bulbar chemosis. We present 11 cases of biopsy-proven conjunctival lymphangiectasia. Of the 11 cases, 3 presented with bilateral diffuse bulbar chemosis, 1 had diffuse unilateral chemosis, and the remaining 7 presented with focal (<90°) bulbar chemosis. Three of these cases had co-existing pterygium, and one case presented with focal bulbar chemosis and a conjunctival keratin horn. All underwent surgical excision of the involved conjunctiva, either with no graft (n = 6), combined with amniotic membrane transplant (n = 3), or combined with conjunctival autograft (n = 2).     

Amniotic membrane transplantation and fibrin glue in the management of corneal ulcers and perforations: a review of 33 cases

PURPOSE: To evaluate the efficacy of amniotic membrane in corneal ulcers refractive to conventional treatment and amniotic membrane with fibrin glue in corneal perforations. METHODS: Amniotic membrane transplantation (AMT) was performed in 33 eyes from 32 patients for corneal ulcers refractive to conventional treatment. Fourteen ulcers were perforated and received fibrin glue and amniotic membrane. Ulcers were divided into 3 groups: neurotrophic or exposure, autoimmune, and other etiology. RESULTS: Overall success was observed in 80% (27/33 eyes) of the cases, with success rates of 87.5% (14/16 eyes), 70% (7/10 eyes), 85.7% (6/7 eyes) in groups 1, 2, and 3, respectively. The ulcers healed in a mean time of 3.6 +/- 1.6 weeks and the follow-up was 14.8 +/- 9.9 months. Failure was noted in 6 eyes with severe neurotrophic keratitis, Stevens-Johnson syndrome, ocular cicatricial pemphigoid, and Acanthamoeba keratitis. Grafts with fibrin sealant showed a success rate of 92.9 % (13/14 eyes) compared to 73.7% (14/19 eyes) for amniotic grafts alone. In patients with severe limbal damage, a success rate of only 20% (1/5) was observed. CONCLUSIONS: AMT is a viable option in the treatment of nonhealing corneal ulcers of various depth and etiologies. Perforations up to 3 mm can be safely managed by fibrin glue and AMT. These techniques lead to rapid reconstruction of the corneal surface and can give a good final functional result or allow keratoplasty to be done in more favorable conditions.