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SHORT COMMUNICATION ARCH SOC ESP OFTALMOL 2006; 81: 661-664 TOPICAL CYCLOSPORINE IN THE TREATMENT OF OCULAR ACTINIC PRURIGO CICLOSPORINA TÓPICA COMO TRATAMIENTO DEL PRÚRIGO ACTÍNICO OCULAR ORTIZ-CASTILLO JV1, BOTO-DE-LOS-BUEIS A2, DE-LUCAS-LAGUNA R2, PASTOR-NIETO B1, PELÁEZ-RESTREPO N1, FONSECA-SANDOMINGO A1 ABSTRACT RESUMEN Case report: A 14-year-old girl from Peru suffered severe limbitis and conjunctivitis. She also presented with clinical skin features diagnosed as actinic prurigo (AP). Her symptoms were successfully controlled with sustained topical therapy of 2% Cyclosporine A. Discussion: AP is an idiopathic photodermatosis that affects mainly the hispanic population of Latin America. There are ocular signs of severe limbitis and conjunctivitis (like atopic keratoconjunctivitis) in 45% of cases. Literature on the subject is very limited and currently topical cyclosporine seems to be the best therapy available (Arch Soc Esp Oftalmol 2006; 81: 661-664). Caso clínico: Presentamos el caso de una mujer de 14 años latinoamericana, con una limbo-conjuntivitis severa. Habia sido diagnosticada de prúrigo actínico (PA) por el dermatólogo. Con el diagnóstico de PA ocular se inició tratamiento con colirio de ciclosporina A 2% con buena respuesta. Discusión: El PA es una fotodermatosis idiopática que afecta principalmente a la población mestiza de Latino América. Los ojos se ven afectados en el 45% de los casos, como una limbo-conjuntivitis severa semejante a la atópica. A pesar de la escasa bibliografía existente, la ciclosporina tópica representa la mejor alternativa en el tratamiento del PA ocular. Key words: Actinic prurigo, topical cyclosporine, conjunctivitis, limbitis, photodermatosis. Palabras clave: Prúrigo actínico, ciclosporina tópica, conjuntivitis, limbitis, fotodermatosis. Received: 27/3/06. Accepted: 20/11/06. Ophthalmology Service. La Paz Hospital Madrid. Spain 1 Graduate in Medicine. 2 Ph.D. in Medicine. Communication presented at the LXXX Congress of SEO (Córdoba 2004). The authors state they have no commercial interest and have not received financial support. Correspondence: Ana Boto de los Bueis C/. O'Donnell 32, 1.º B 28009 Madrid Spain E-mail: [email protected] ORTIZ-CASTILLO JV, et al. INTRODUCTION Actinic prurigo (AP) is a photodermatosis typical of Latin American countries. The ocular expressions of this dermatosis can simulate an atopical conjunctivitis (1). This communication presents the case of a girl with severe limbus conjunctivitis due to AP. CASE REPORT A 14 year-old female from Perú who in the last 6 months exhibited severe episodes of reddening and photophobia. She was not responsive to dexametasone (Colircusí Dexametasona® 0,1 %, Alcon Cusi, Spain) and levocabastine (Bilina®, Esteve, Barcelona, Spain). She referred suffering atopical conjunctivitis and dermatitis since age 7 as well as a herpetic keratitis outbreak in the left eye (LE) 4 years ago and a corneal ulcer in the right eye (RE) two months ago. The patient exhibited erithematous and flaking skin lesions in the malar, frontal and perilabial areas as well as the sides of the nose and hands. There was not palpebral involvement (fig. 1). The ophthalmological exploration evidenced a corrected visual acuity of CD at 2 mt in RE and 2/3 in LE, while biomicroscopy showed that in both eyes (BE) the patient had a papillar reaction in the superior tarsal conjunctiva (fig. 2), gelatinous limbar hypertrophy, bulbar conjunctival hyperpigmentation spots (fig. 3) and superficial dotted keratitis. The RE exhibited pannus and a central leucoma, and the LE a small paracentral leucoma. She did not refer familial atopy history and did not show other atopy systemic expressions. The allergy study discarded sensitivity to common allergens. The serum IgE was normal as was the tear IgE (Lacrytest, Adiatec lab, France). Fig. 2: Papillar hypertrophy in superior RE tarsum. Fig. 1: Erithematous and flaking facial lesions due to actinic prurigo. 662 Fig. 3: RE limbus conjunctivits due to AP, and vascularized central corneal leucoma secondary to previous infectious ulcer. ARCH SOC ESP OFTALMOL 2006; 81: 661-664 Topical cyclosporine for treatment of ocular actinic prurigo The dermatology service was consulted. They referred substituting the atopic dermatitis diagnosis established two years ago by AP after the results of triple cutaneous biopsy. Topical treatment was initiated with tacrolimus ointment 0.03% continued to this date, which led to a stabilization of the skin condition. With the ocular AP diagnostic, in Sept. 2004 we began treatment with cyclosporine A eye drops 2%, prepared in polyvinyl alcohol by the pharmacy service, with expiry date at 28 days. The initial dosage was 1 drop/8 hours in BE, leading to an improvement two weeks later. In April 2005 and while in treatment with 1% cyclosporine every 12 hours, the patient exhibited a reactivation of the condition in BE, which was controlled increasing the eye drops to cyclosporine 2%/8h. One week later she exhibited a central herpetic dendrite in the LE which was resolved with acyclovir cream (Zovirax ophthalmic cream®, Glaxo-Wellcome, Tres Cantos, Madrid, Spain) 5 times a day for 21 days. Since then, she continues treatment with 1% cyclosporine eye drops every 12 hours and valaciclovir 500 mg/day (Valtrex®, GlaxoSmithKline, USA). In Feb. 2006 the patient exhibited a corrected VA in RE of 1/8 and 1/2 in LE, with the only inflammatory finding of slight temporal-inferior limbitis in BE. DISCUSSION This communication presents the case of a young Latin American female with a severe bilateral limbal conjunctivitis condition, initially attributed to atopia and associated to chronic dermatitis. The final diagnostic was AP, a recurring chronic idiopathic photodermatosis caused by an immune reaction by the skin and mucous to UVA and UVB radiation. This condition occurs almost exclusively in Latin American countries where the population lives at a height exceeding 1,000 m above sea level and it usually expresses between the ages of 6 and 8. The skin is affected in areas exposed to light such as the nose, malar regions and lips (2). The eyes are involved in 45% of cases, primarily affecting the conjunctiva and the limbus, which exhibited diffuse hyperemia, aqueous secretion, hyperpig- mented bulbar conjunctival areas, hypertrophic tarsal papillae, follicles, focal limbitis and pseudopterigium. The AP diagnostic is based on the clinical presentation and the anatomic-pathological study of mucose of the lips, the conjunctiva or the skin. The tarsal conjunctival histology typically exhibits epithelium thinning and atrophy, vacuolization of basal cells with lymphocyte infiltrates organized in sub-mucous follicles, conjunctival pigmentation and eosinophiles, while the most characteristic histopathological finding of AP, in mucous and in conjunctiva, is the presence of lymphoid follicles in the laminae itself (2). The tacrolimus ointment is the best option for dermatosis. Eye lesions call for a different therapeutic approach. At present and notwithstanding the scarce amount of literature on the subject, studies point to topical cyclosporine as the best option for treating ocular actinic prurigo (3). In our case, the patient improved with this treatment in about 2 weeks but it was necessary to continue it to avoid recurrences. Although there is no experience in the utilization of topical tactrolimus in ocular AP, recently this collyrium has proved to be efficient in chronic blepharoconjunctivitis (4) and severe atopical conjunctivitis (5). In the future, this power immunosuppressant macrolide could be a therapeutic alternative for ocular AP cases resistant to treatment with cyclosporine. REFERENCES 1. Magana M, Mendez Y, Rodriguez A, Mascott M. The conjunctivitis of solar (actinic) prurigo. Pediatr Dermatol 2000; 17: 432-435. 2. Hojyo-Tomoka T, Vega-Memije E, Granados J, Flores O, Cortes-Franco R, Teixeira F, et al. Actinic prurigo: an update. Int J Dermatol 1995; 34: 380-384. 3. McCoombes JA, Hirst LW, Green WR. Use of topical cyclosporin for conjunctival manifestations of actinic prurigo. Am J Ophthalmol 2000; 130: 830-831. 4. Joseph MA, Kaufman HE, Insler M. Topical tacrolimus ointment for treatment of refractory anterior segment inflammatory disorders. Cornea 2005; 24: 417-420. 5. Rikkers SM, Holland GN, Drayton GE, Michel FK, Torres MF, Takahashi S. Topical tacrolimus treatment of atopic eyelid disease. Am J Ophthalmol 2003; 135: 297-302. ARCH SOC ESP OFTALMOL 2006; 81: 661-664 663