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ABSTRACT: Purpose: Timolol is an effective and safe medication that is widely used in glaucoma treatment. Although it is known that it is quickly taken up by the cornea following topical administration and that the cornea exhibits β-adrenergic receptors, there are few studies available on the clinical impact of timolol on central corneal thickness (CCT).
Methods: Twenty healthy subjects were tested in a double-blind, prospective, and randomized study. Intraocular pressure (IOP) and CCT were measured before and during administration of timolol 0.5% eyedrops over 28 days.
Results: Administration of timolol 0.5% resulted in a reduction of IOP from an initial value of 16 ± 2 mm Hg to 13 ± 0 mm Hg (p<0.001, R2 = 0.7033) as well as an increase in CCT from 555 ± 11 μm from the time of the baseline examination to 567 ± 9 μm (p = 0.005, R2 = 0.8754), an increase of epithelial thickness from 53 ± 2 μm to 59 ± 3 μm (p<0.001, R2 = 0.5063), and an increase of stromal thickness from
494 ± 4 μm to 498 ± 9 μm (p = 0.045, R2 = 0.4352) after 9 days each. From day 10 on, a decrease in CCT (R2 = 0.6164), epithelial thickness (R2 = 0.2216), and stromal thickness (R2 = 0.2092) was observed. At the end, the values had returned toward the initial values measured (CCT 553 ± 8 μm, p = 0.391; epithelial thickness, 50 ± 2 μm, p = 0.214; and stromal thickness, 493 ± 8 μm, p = 0.483). In contrast, endothelial thickness did not vary following administration of timolol 0.5% (p = 0.727, R2 = 0.009).
Conclusions: Topical administration of timolol 0.5% results in a reversible increase in CCT. These modest changes are unlikely to influence tonometry or clinical decision-making.
European journal of ophthalmology 05/2013; · 0.96 Impact Factor
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ABSTRACT: 5-Hydroxytryptamine (5-HT; serotonin) is a major neurotransmitter, and its receptors are found throughout the whole body. The 5-HT7 receptor subtype was detected in human corneal epithelial and endothelial cells and found to be functionally active in a corneal epithelial cell line. The aim of the present study was to demonstrate that native bovine corneal epithelial and endothelial cells express a functional 5-HT7 receptor positively coupled to adenylyl cyclase and protein kinase A (PKA) formation.
5-HT7 receptors were studied using polyclonal antibodies. cAMP concentration after 5-HT7 receptor stimulation with 5-carboxamidotryptamine (5-CT, a 5-HT7 agonist) was tested by enzyme immunoassay, PKA activity was estimated by kinase consumption of ATP.
Immunocytochemistry and immunofluorescence revealed the presence of 5-HT7 receptors in corneal epithelial and endothelial cells. Stimulation of corneal 5-HT7 receptors with 5-CT revealed a dose-dependent increase in intracellular cAMP concentration in corneal epithelium (0.01-0.34 pmol/ml) and endothelium (0.01-0.19 pmol/ml) between 10(-10) and 10(-7) mg/ml 5-CT (p = 0.001) with maximal stimulation from 10(-7) to 10(-3) mg/ml 5-CT (0.30 ± 0.03 and 0.18 ± 0.01 pmol/ml, respectively). Incubation with 10(-6) mg/ml SB269970 (a selective 5-HT7 antagonist) blocked 5-CT-induced cAMP increase in corneal epithelial (0.03 pmol/ml) and endothelial cells (0.02 pmol/ml; p = 0.001). Stimulation of corneal 5-HT7 receptors with 5-CT revealed a dose-dependent increase in PKA activity between 10(-10) and 10(-8) mg/ml 5-CT in corneal epithelium and endothelium (<1 to >99%; p = 0.013 and p = 0.017, respectively) with maximal stimulation from 10(-8) to 10(-4) mg/ml (>99%) 5-CT.
Our data demonstrate that native corneal epithelial and endothelial cells express a functional 5-HT7 receptor positively coupled to adenylyl cyclase and PKA formation. However, at the present time, the physiological role of 5-HT receptors and the cAMP-PKA pathway in the cornea remains a matter of speculation.
Ophthalmic Research 01/2012; 48(1):22-7. · 1.56 Impact Factor
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ABSTRACT: Brimonidine, an alpha-2 adrenoceptor agonist, is an effective and safe medication that is widely used in glaucoma treatment. Although it is known that it is quickly taken up by the cornea following topical administration and that the cornea has alpha-2 adrenoceptors, there are only few studies available on the impact brimonidine has on the cornea.
Twenty healthy test persons (12 female and 8 male subjects)-mean age about 33 years (22 to 38 years)-were tested in a double-blind, prospective, randomized study. Intraocular pressure as well as epithelial, stromal, and endothelial thickness was measured before, at 25 days while, and at 5 days after administration of brimonidine 0.1% eye drops twice daily. To check the impact of this medication, placebo (proper solution of preservative) eye drops were administered to the other eye twice daily.
Administration of brimonidine 0.1% resulted in a reduction of intraocular pressure from an initial value of 14 to 9 mmHg after 5 days (P=0.001) as well as an increase in total corneal thickness from 556 μm from the time of the baseline examination to 578 μm (P=0.001), an increase of epithelial thickness from 58 to 66 μm (P<0.001), and stromal thickness from 488 to 502 μm (P=0.008) after 2 days each. Another 2 days later, total corneal thickness was 559 μm (P=0.276), epithelial thickness 56 μm (P=0.561), and stromal thickness 493 μm (P=0.315), which means that the values had returned more or less toward the initial values measured. In contrast, endothelial thickness did not vary following administration of brimonidine 0.1% (P=0.965). With treatment with brimonidine 0.1%, mean intraocular pressure in thin corneas (<556 μm) was lower than in the thick corneas (>556 μm, P=0.018).
Topical administration of brimonidine 0.1% results in a reversible increase in corneal thickness. The question whether this increase is of clinical significance and whether it is the result of epithelial and/or endothelial receptor stimulation cannot be finally answered at the present time.
Journal of ocular pharmacology and therapeutics: the official journal of the Association for Ocular Pharmacology and Therapeutics 07/2011; 27(5):503-9. · 1.46 Impact Factor
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ABSTRACT: To evaluate the expression of melanoma cell adhesion molecule (MCAM) as prognostic factor in uveal melanoma patients.
Paraffin sections from 35 primary uveal melanomas were analyzed immunhistochemically for the expression of MCAM. Further known prognostic factors such as cell type, ciliary body invasion, tumor stage, and local extension were evaluated. In 16 cases, patients had developed metastatic disease. The patients without metastasis (n = 19) had a mean follow-up of 10.6 (9-13) years.
All tumors were positive for MCAM (mean: 57.05%). In the group without metastasis, an average MCAM expression of 35.37% (SD 19.66%) compared to 82.78% (SD 11.73%) in the metastasis group was found. The Cox regression analysis showed a significant association of an enhanced MCAM expression and death due to metastasis (HR: 365.48, 95% CI: 18.51, 7217.85).
MCAM expression of primary uveal melanomas can serve as a possible prognostic parameter for metastasis. If further experiments approve the cellular adhesion molecules to account for early micrometastasis of tumor cells, a possible target might be available for future treatment.
Current eye research 11/2009; 34(11):1004-9. · 1.51 Impact Factor
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ABSTRACT: We present 3 cases of opacification in the Biocomfold 92S intraocular lens (IOL), with a documented increase in the opacification over time in 1 case. Histopathological analysis revealed the opacification was caused by calcification in the IOL's optic material. The pattern of calcium deposition, however, was different in each IOL. The causative mechanisms for this complication are unclear. Further research is warranted.
Journal of cataract and refractive surgery 04/2009; 35(3):593-7. · 2.75 Impact Factor
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ABSTRACT: The G-protein-coupled receptor/cyclic adenosine monophosphate (cAMP)/protein kinase A (PKA) pathway is one of the most common and versatile signal pathways in eukaryotic cells. The aim of this study was to characterize subtypes of adrenergic G-protein-coupled receptors and their influence on cAMP concentration and PKA activity in bovine corneal epithelial and endothelial cells.
Adrenergic receptors and PKA were studied using polyclonal antibodies. cAMP concentration was determined with an enzyme immunoassay, and PKA activity was estimated by the kinases consumption of adenosine triphosphate.
In bovine corneal epithelial and endothelial cells, immunocytochemistry and Western blot were positive for alpha(1)-, alpha(2A)-, beta(1)- and beta(2)-adrenergic receptors. Stimulation of corneal epithelial and endothelial beta-adrenoceptors with isoprenaline led to a dose-dependent increase in cAMP concentration and activation of PKA. Stimulation of corneal alpha(2A)-adrenoceptors with brimonidine resulted in a dose-dependent decrease in cAMP concentration and the inhibition of PKA activity.
In corneal epithelial and endothelial cells, beta-adrenergic stimulation leads to activation of PKA via stimulation of adenylyl cyclase, and alpha(2A)-adrenoceptor stimulation inhibits PKA activity via inhibition of adenylyl cyclase. Stimulation and inhibition of the corneal cAMP-PKA pathway may play a role in important corneal functions such as wound healing or homeostasis. Long-term therapy with alpha(2A)-agonists or beta-antagonists may influence these functions in a currently unknown way.
Ophthalmic Research 09/2008; 40(6):322-8. · 1.56 Impact Factor
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ABSTRACT: Isolated vitreous metastases are extremely rare and the pathogenesis of metastasis is still unclear. Here we present the detailed description of the disease progression in a 68-year-old patient with vitreous seeding of a metastatic cutaneous melanoma beginning at a very early stage.
Interventional case report and review of the literature.
The initial retrohyaloidal metastatic lesion was identified adjacent to a small epiretinal hemorrhage. As the disease progressed golden brown spherules appeared in the posterior vitreous emanating from the area of the lesion. Further progression led to a dense metastatic infiltration of the entire vitreous cavity and a decline of the visual acuity to 20/1200. Diagnostic and therapeutic pars plana vitrectomy was performed to confirm the diagnosis and preserve the eye and useful vision.
For the first time the formation of vitreous metastases derived from cutaneous melanoma was carefully studied beginning at a very early stage. This made it possible to analyze the rare mechanism of vitreous metastasis, which has not been conclusively known till now. The features of metastatic cutaneous melanoma to the vitreous are discussed in context of a review of the literature that resulted from the study of 17 patients with 22 affected eyes.
Albrecht von Graæes Archiv für Ophthalmologie 06/2007; 245(5):733-40. · 2.17 Impact Factor
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ABSTRACT: Muscarinic cholinoceptors are distributed widely in both the central and peripheral nervous system. The presence of muscarinic cholinoceptors in corneal tissue is well established. Previous reports have shown that corneal muscarinic cholinoceptors are of the m2 or m4 subtype. However, recent studies have indicated the presence of the m5 muscarinic cholinoceptor subtype in human corneal epithelium and endothelium. The aim of the study was to confirm the presence of the m5 cholinoceptor subtype in bovine corneal epithelium and endothelium and the activation of phosphatidyl inositol pathway by its stimulation.
Muscarinic m5 cholinoceptor sites, phosphatidyl inositol 4,5-biphosphate, inositol 1,4,5-triphosphate and protein kinase C, were studied using immunocytochemistry and immunofluorescence. Activation of protein kinase C after stimulation of the m5 muscarinic cholinoceptor subtype was measured using the HTS protein kinase C assay kit.
Immunocytochemistry/immunofluorescence revealed the presence of the m5 muscarinic cholinoceptor subtype, phosphatidyl inositol 4,5-biphosphate and protein kinase C in bovine corneal epithelial and endothelial cells. In bovine corneal epithelium and endothelium, protein kinase C activity was stimulated by acetylcholine in a dose-dependent manner (P<0.0001).
Our findings indicate that acetylcholine-induced stimulation of muscarinic m5 cholinoceptors activates the phosphatidyl inositol pathway in corneal epithelial and endothelial cells, resulting in increased protein kinase C activity. Further work will be needed to clear the physiologic role of this signaling pathway in corneal epithelium and endothelium.
Albrecht von Graæes Archiv für Ophthalmologie 04/2007; 245(4):595-9. · 2.17 Impact Factor
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ABSTRACT: Candida glabrata is a rare cause of endophthalmitis after penetrating keratoplasty. Adequate therapy is still under discussion. With respect to severe complications and side effects of antifungal therapy, a substantial knowledge of sensitivity and resistance of the organism is necessary.
We report on a 26-year-old man with a hyperacute onset of the infection only 10 hours after surgery. A combined therapy for fluconazole and steroids administered over 3 months had shown no effect on intraocular infection.
After topical and intracameral application of amphotericin B in combination with topical prednisolone 3 months after the onset of the endophthalmitis, the infection disappeared within 14 days, and the graft remained clear for 2 months. No toxic effects were noticed.
In the case presented here, topical and intracameral application of amphotericin B was sufficient and safe in the therapy for C. glabrata endophthalmitis after penetrating keratoplasty. Although typically the intraocular infection is first noticed within the first 2 weeks, a hyperacute onset has to be considered.
Cornea 01/2007; 25(10):1243-4. · 1.73 Impact Factor
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ABSTRACT: Transscleral diode laser cyclophotocoagulation (TDLC) has been used successfully in the treatment of refractive glaucoma. However, little is known about TDLC as a primary or secondary surgical procedure in primary open-angle and pseudoexfoliative glaucoma.
In a retrospective chart review we analyzed 90 eyes (48 OD/42 OS) in 90 patients (mean age 75 years, range 60-92 years; 51 men/39 woman) with primary open-angle glaucoma (n=66) and pseudoexfoliative glaucoma (n=24) who consecutively underwent TDLC with a minimum follow-up of 24 months. In 24 eyes (26.7%) TDLC was the primary surgical treatment. Mean energy was 80 J (60-92 J). Success was defined as a final intraocular pressure (IOP) between 4 and 18 mmHg, a minimum IOP reduction of 20% and the absence of major complications.
The overall success rate was 36.7% after 24 months. A mean of 1.3 procedures was performed per patient. No correlation between age and success rate (p=0.887) or gender and success rate (p=0.895) was seen. There was no significant reduction in antiglaucomatous medication (p=0.208), no significant loss of visual acuity (p=0.324) nor a significant relationship between loss of visual acuity and failure of treatment (p=0.201). In patients with primary open-angle glaucoma the success rate was 40.9% after 24 months; in patients with pseudoexfoliative glaucoma it was 25.0% after 24 months. There was no significant difference between primary open-angle glaucoma and pseudoexfoliative glaucoma (p=0.684). Previous ocular surgery decreased the success probability from 45.8 to 33.3% (p=0.001). Prolonged hypotonia occurred in 1 patient. No phthisis bulbi developed.
Transscleral diode laser cyclophotocoagulation is an effective and safe method not only in the treatment of refractive glaucoma, but also as a primary surgical procedure in primary open-angle and pseudoexfoliative glaucoma. TDLC may be used more widely in glaucoma therapy, although further long-term studies have to confirm these findings.
Albrecht von Graæes Archiv für Ophthalmologie 11/2006; 244(10):1293-9. · 2.17 Impact Factor
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ABSTRACT: Muscarinic acetylcholine receptors are located throughout the body. The demonstration of muscarinic receptors in corneal tissue has been inconsistent. Using freshly fixed human corneal tissue, we show a complete profile of muscarinic receptor subtypes in human corneal epithelium and endothelium.
Muscarinic receptor sites were studied using immunocytochemistry, immunofluorescence and immunoblotting.
Antibodies to M2, M4 and M5 muscarinic receptor subtypes bound in human corneal epithelium and endothelium. No binding was found for antibodies to M1 and M3 muscarinic receptor subtypes.
Our studies indicate the presence of M2, M4 and M5 muscarinic acetylcholine receptor subtypes in human corneal epithelium and endothelium. These receptors may play a role in the regulation of corneal homeostasis, other functions, like wound healing, or the pathogenesis of corneal diseases.
Albrecht von Graæes Archiv für Ophthalmologie 10/2006; 244(9):1191-5. · 2.17 Impact Factor
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ABSTRACT: Endostatin is an endogenous angiogenesis inhibitor which requires E-selectin for its antiangiogenic activity. The aim of this study was to investigate the expression of endostatin in human choroidal neovascular membranes (CNV) secondary to age-related macular degeneration (AMD) with regard to vascularization and proliferative activity. An interventional case series of 36 patients who underwent removal of CNV were retrospectively investigated. Thirty-six CNV were analyzed by light microscopic immunohistochemistry for the expression of CD34 (endothelial cells, EC), CD105 (activated EC), Ki-67 (cell proliferation), Cytokeratin 18 (epithelial cells), VEGF (vascular endothelial growth factor), E-selectin and endostatin. Donor eyes (n=7) including one with AMD were used as controls. Endostatin immunoreactivity was present in choroidal vessels of five as well as in the retinal pigment epithelium (RPE)-Bruch's membrane complex of two donor eyes without AMD. In one eye with AMD, endostatin was detected in RPE, Bruch's membrane and choroidal vessels. Ninety-two percent (33/36) of CNV disclosed endostatin staining. RPE-Bruch's membrane complex, choroidal vessels and stroma were positive in 50% (18/36), 72% (26/36), and 78% (28/36) of the membranes, respectively. Both control eyes and CNV expressed all the investigated markers except E-selectin being positive only in membranes. Endostatin, an endogenous angiogenesis inhibitor, is expressed in CNV and its therapeutic up-regulation may be a new strategy in the treatment of neovascular AMD.
Experimental Eye Research 09/2006; 83(2):329-38. · 3.26 Impact Factor
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ABSTRACT: Monoamine receptors are found throughout the body. Reports about the presence of monoamine receptors in the human cornea are inconsistent.
Immunohistochemistry, immunofluorescence and immunoblotting were used to localize monoamine receptor sites on human corneal epithelium and endothelium.
Antibodies to alpha-1, beta-1 and beta-2 adrenergic receptors and to D1-like and 5HT-7 receptors were bound in corneal epithelium. Antibodies to alpha-1, alpha-2A, beta-1 and beta-2 adrenergic receptors and to 5HT-7 receptors were bound in corneal endothelium.
Our data demonstrate the presence of several monoamine receptors in the human cornea. These receptors may play a role in the regulation of fluid transport or corneal homeostasis.
Acta Ophthalmologica Scandinavica 03/2006; 84(1):110-5. · 1.85 Impact Factor
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Acta Oncologica 02/2006; 45(2):218-9. · 3.33 Impact Factor
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ABSTRACT: Massive retinal gliosis is a rare, benign, intraocular tumor. It develops mostly in long-standing ocular disease such as chronic inflammation, vascular disorders, glaucoma, trauma, or congenital abnormalities. We report on a case of massive retinal gliosis, which developed 17 years following retinal detachment surgery.
Case report.
A 64-year-old woman developed a peripheral fundus tumor 17 years after successful retinal detachment surgery. Follow-up examination demonstrated growth of the mass. The tumor was treated by pars plana vitrectomy and local endoresection. Histopathological study revealed massive reactive gliosis of the retina.
To our knowledge, this is the first histopathologically proven case of a massive retinal gliosis, which developed after a non-drainage retinal detachment surgery. Endoresection is a therapeutic option in suspected massive retinal gliosis. It provides material for histological diagnosis which obviates unnecessary intervention in a functioning eye.
Albrecht von Graæes Archiv für Ophthalmologie 04/2004; 242(3):255-8. · 2.17 Impact Factor
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Jens Martin Rohrbach
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ABSTRACT: As financial resources run short there are considerations to integrate the ophthalmopathological laboratories of the eye hospitals into the institutes for pathology. The term "return" which is used sometimes in the discussion is historically incorrect as the pathology of the eye--similar to dermatopathology in the clinics for dermatology--was developed quite independently from general pathology in the eye hospitals over the last 150 years. Due to the work of outstanding ophthalmologists like Otto Becker, Theodor Leber, Julius Michel, and Ernst Fuchs ophthalmopathology augmented the ophthalmological knowledge much more than any other subdiscipline in the late 19th and the early 20th century. At any time, the very close link of the clinical picture and the morphology proved to be very advantageous for the understanding of the diseases. As a consequence of new diagnostic tools and progress in therapy the pathology of the eye is always changing and it is a still very important motor of modern ophthalmology. Whether in the clinical or in the experimental field: classical ophthalmopathology serves to characterise the phenotype, and nothing indicates that genomics or proteomics will make it unnecessary within the foreseeable future. Pure diagnostic pathology of the eye does not cover the costs. However, there are enough good reasons to keep the ophthalmopathological laboratories as parts of the eye hospitals.
Klinische Monatsblätter für Augenheilkunde 03/2004; 221(2):87-91. · 0.51 Impact Factor
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ABSTRACT: Posttransplantation lymphoproliferative disorder (PTLD) of the iris is a rare entity with only ten cases having been published as yet. Its clinical aspect is typical. Therapy is multimodal and affords an interdisciplinary approach.
A 7-year-old boy developed a lymphoproliferative mass of the iris with uveitis 4 years after heart transplantation and immunosuppression. A progressive, flesh-colored thickening of the iris with secondary angle closure glaucoma necessitated a diagnostic and therapeutic iridectomy. Morphological investigation of the iris specimen disclosed a polymorphic posttransplantation lymphoproliferative disorder (PTLD) and the presence of Epstein-Barr virus (EBV) within the tissue. The EBV load in peripheral blood monocytes was massively elevated, thus indicating a chronic EBV infection. After conservative treatment and radiation therapy, the iris mass quickly resolved. There was no evidence of systemic PTLD.
PTLD is a well-known, EBV-induced entity that rarely affects the eye. EBV is principally detectable in specimens of iris PTLD. If conservative, antiviral treatment fails, the iris lesions can be treated by local radiation therapy with very good success. In the near future, patients with PTLD of the eye may benefit from immunologic treatment with ex vivo generation of virus-specific T-lymphocytes.
Albrecht von Graæes Archiv für Ophthalmologie 02/2004; 242(1):44-50. · 2.17 Impact Factor
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ABSTRACT: To report on a case of bifocal rupture of the limbus that developed in a young male with secondary angle closure glaucoma 7 months after penetrating eye injury.
A 20-year old male suffered from severe polytrauma due to a car accident. Examination revealed an open globe injury of the left eye due to corneal penetration by a foreign body (glass). After primary wound closure a pars plana vitrectomy with lens extraction and removal of the foreign body was performed. Five months later IOP increased markedly and could neither be controlled by antiglaucomatous medication nor by cyclophotocoagulation. Seven months after the injury a bifocal, closed rupture of the upper nasal and temporal corneoscleral limbus occurred. IOP of the eye was elevated despite the rupture. The limbal dehiscence was readapted and IOP increased again. A new limbal rupture occurred and a tectonic keratoplasty was performed. Because a marked thinning of the transplanted cornea occurred accompanied by strong evidence of advanced epithelial ingrowth the eye was enucleated. Histologic examination of the excised tissue and enucleated eye showed diffuse epithelial ingrowth.
This is first reported case of delayed, spontaneous, bifocal rupture of the corneoscleral limbus after primary open globe injury. It may be speculated that severe contusion of the eye with structural damage of the corneoscleral limbus preceded the penetrating injury and that the later limbal rupture was caused by a marked elevated IOP due to epithelial ingrowth.
Klinische Monatsblätter für Augenheilkunde 12/2003; 220(11):787-90. · 0.51 Impact Factor
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ABSTRACT: The Melkersson-Rosenthal syndrome complex is characterised by asymmetric orofacial oedema, facial palsy, furrowed tongue (lingua plicata) and additional neurological symptoms.
A 21-year old man presented with a bilateral asymmetric oedema of the eyelids which increased during the follow-up period of 12 years. Intermittent swelling of the auditory channel occurred and resulted in hearing loss. Furthermore, the patient complained unspecific neurological symptoms as headache, tinnitus and subjective visual defects. The lid oedema did not only result in cosmetic problems but seriously restricted visual fields.
Our patient showed the typical symptoms of the Melkersson-Rosenthal complex, lacking however facial palsy which is often caused by mechanical compression along the course of the facial nerve. The differential diagnosis of recurrent swelling of the lids was discussed on an interdisciplinary plane. The inflammatory activity could be reduced and the frequency of swelling episodes could be diminished by a treatment with hydroxychloroquine. The initially relapsing, then persistent swelling had led to severe restriction of the visual field and resulted in a severe social stigmatisation and psychic stress for the patient. The surgical resection of the granulomatous tissue was carried out under steroid treatment to prevent recurrence. Histochemical analysis showed inflammation of the lymphoid plasmacellular type with mucipolysaccharidoid deposition typical of Melkersson-Rosenthal syndrome. Blepharoplasty resulted in an improved cosmetic appearance. The patient remained symptom-free with an excellent functional result.
After persistence and constancy of the findings of a granulomatous blepharitis, surgical correction is a good opportunity to minimise the subjective complaints and neurological symptoms.
Klinische Monatsblätter für Augenheilkunde 06/2003; 220(5):352-6. · 0.51 Impact Factor
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ABSTRACT: The keratitis-ichthyosis-deafness (KID) syndrome (Senter Syndrome) and its major criteria erythrokeratodermia, neuro-sensoric deafness and vascularising keratitis were defined in 1981. Several cases have been described since 1915, but up to now no causal therapy of this disease with sporadic occurrence has been found. Clinical experience using systemic Cyclosporin A (CsA) in the dermatological therapy of the KID syndrome differs. Up to now there has been no report on the use of topical CsA eye drops in the therapy of vascularising keratitis.
We report on a case of keratitis, ichthyosis and deafness (KID syndrome) in a 36-year old male. Both eyes were suffering from recurrent corneal ulcers (ulcus serpens corneae). No improvement had been observed on topical or systemic medication (including lubricants, antibiotics, steroids, etc.) during a history of more than two years.
Topical CsA 2 % (eye drops, 3 times daily) was administered in addition to lubricants. After 3 months the deep corneal neovascularisations were significantly reduced. The corneal ulcer was closed and visual acuity increased from 20/400 (OU) to 20/100 (OD) and 20/200 (OS), respectively.
A dose-dependent keratinocyte growth inhibition was found in vitro for cyclosporin A using ichthyosis skin samples. The reduction of activated T-lymphocytes and the resulting reduced HLA-Class-2 expression of conjunctiva and cornea as well as the inhibition of activated T-Cells in the lacrimal gland might be responsible. Nevertheless, the effect of topical CsA on the vascularizing keratitis in the KID syndrome is unknown.
Klinische Monatsblätter für Augenheilkunde 06/2002; 219(5):383-6. · 0.51 Impact Factor
