Unilateral acute posterior multifocal placoid pigment epitheliopathy in a patient with a strongly positive purified protein derivative test / Epiteliopatia pigmentar placóide multifocal posterior aguda unilateral na presença de reação forte positiva para o teste tuberculínico

ABSTRACT A 27-year-old man presented with a complaint of decreased visual acuity in the right eye. Best-corrected visual acuity was 20/60 in the right eye and 20/20 in the left eye. Right eye fundoscopy revealed mild vitritis, multifocal yellowish lesions, and macular serous retinal detachment. Left eye evaluation was normal. Acute posterior multifocal placoid pigment epitheliopathy was diagnosed in the right eye. Complementary exams revealed a strong reaction to the Mycobacterium tuberculosis purified protein derivative test, thus treatment for tuberculosis was initiated. Baseline fluorescein angiography of the right eye revealed early hypofluorescence and late staining of the lesions. Optical coherence tomography of the right eye demonstrated the accumulation of subretinal and intraretinal fluid associated with cystoid macular edema. During follow-up, the retinal fluid and cysts disappeared, which was followed by disorganization of foveal interdigitation and ellipsoid zones. This is the second described case of unilateral acute posterior multifocal placoid pigment epitheliopathy in a patient with a strong positive result to the M. tuberculosis purified protein derivative test.

RESUMO Um homem de 27 anos apresentou uma queixa de diminuição da acuidade visual no olho direito. A acuidade visual melhor corrigida foi 20/60 no olho direito e 20/20 no olho esquerdo. A fundoscopia do olho direito revelou vitreíte leve, lesões amareladas multifocais e descolamento seroso da retina em região macular. A avaliação do olho esquerdo foi normal. Epiteliopatia pigmentar placóide multifocal posterior aguda foi diagnosticada no olho direito. Os exames complementares revelaram forte reação ao teste do derivado proteico purificado Mycobacterium tuberculosis, iniciando terapia antibiótica contra tuberculose. A angiografia fluoresceínica basal do olho direito revelou hipofluorescência precoce e tardia das lesões. A tomografia de coerência óptica do olho direito demonstrou fluído sub- e intrarretiniano associado a edema macular cistóide. Durante o seguimento, os fluídos e cistos retinianos desapareceram, seguido da desorganização das zonas de interdigitação e elipsóide em região foveal. Este é o segundo caso descrito de epiteliopatia pigmentar placóide multifocal posterior aguda unilateral em um paciente com um forte resultado positivo para o teste tuberculínico.

Unilateral acute posterior multifocal placoid pigment epitheliopathy in a patient with a strongly positive purified protein derivative test.

A 27-year-old man presented with a complaint of decreased visual acuity in the right eye. Best-corrected visual acuity was 20/60 in the right eye and 20/20 in the left eye. Right eye fundoscopy revealed mild vitritis, multifocal yellowish lesions, and macular serous retinal detachment. Left eye evaluation was normal. Acute posterior multifocal placoid pigment epitheliopathy was diagnosed in the right eye. Complementary exams revealed a strong reaction to the Mycobacterium tuberculosis purified protein derivative test, thus treatment for tuberculosis was initiated. Baseline fluorescein angiography of the right eye revealed early hypofluorescence and late staining of the lesions. Optical coherence tomography of the right eye demonstrated the accumulation of subretinal and intraretinal fluid associated with cystoid macular edema. During follow-up, the retinal fluid and cysts disappeared, which was followed by disorganization of foveal interdigitation and ellipsoid zones. This is the second described case of unilateral acute posterior multifocal placoid pigment epitheliopathy in a patient with a strong positive result to the M. tuberculosis purified protein derivative test.

[Congenital Simple Hamartoma of the Retinal Pigment Epithelium]. / Kongenitales einfaches Hamartom des retinalen Pigmentepithels.

BACKGROUND: Congenital simple hamartoma of the retinal pigment epithelium (CSHRPE) is an uncommon benign lesion with characteristic clinical features. Ophthalmoscopically it appears as a small localized, well circumscribed, pigmented tumor in the foveal region. In contrast to the more common flat congenital hypertrophy of the RPE the CSHRPE has an elevated nodular appearance. PATIENTS AND METHODS: Retrospective case series of three patients with CSHRPE. Clinical morphological features using different imaging techniques are presented. RESULTS: A typical dark lesion was incidentally noted in the macula of two patients. Optical coherence tomography (OCT) demonstrated a nodular preretinal hyperreflectivity with shadowing of deeper structures. In one patient the CSHRPE was hypofluorescent throughout the angiogram. The third patient presented with a reduced visual acuity of 0.3. A characteristic lesion was found at the foveal center. OCT revealed a hyperreflective preretinal lesion with associated moderate disruption of the foveal architecture. Amblyopia treatment slightly improved visual acuity in this case. The lesions remained stationary in two patients (follow-up 8 - 14 months). CONCLUSIONS: CSHRPE are usually detected as an incidental finding. Given its benign character and typically asymptomatic presentation an observational treatment approach is generally recommended. The lesions generally remain stationary and are not known to grow. In cases with visual impairment due to foveal involvement amblyopia treatment should be initiated.

Secondary glaucoma induced by bilateral acute depigmentation of the iris / Glaucoma secundário à despigmentação bilateral aguda da íris

Abstract We report a case of a middle-aged woman who developed acute, bilateral, symmetrical, slightly transilluminating depigmentation of the iris and pigment discharge into the anterior chamber following the use of oral moxifloxacin for bacterial sinusitis. She had been misdiagnosed as having autoimmune uveitis, treated with steroids and tropicamide, and underwent severe ocular hypertension and glaucoma despite posterior correct diagnosis.

Resumo Relato de um caso de uma paciente do sexo feminino de meia idade que desenvolveu despigmentação bilateral simultânea aguda com dispersão de pigmentos na câmara anterior e discreta transiluminação após o uso de moxifloxacino oral para tratamento de sinusite bacteriana. Ela Havia sido diagnosticada com uveite autoimune e tratada com corticosteroide tópico e tropicamida e evoluiu com hipertensão ocular grave e glaucoma apesar de ,posteriormente, o diagnóstico ter sido correto.

Detection of an Iris Pigmented Epithelium Cyst in a Child by High Frequency Ultrasonography.

An 18-month-old boy presented with an iris pigmented epithelium cyst. The diagnosis was confirmed using high frequency ultrasonography. The cyst was determined to be in an intermediate location, thus needing no treatment provided that any potential complications were noted. However, continued monitoring is required to detect any changes to adapt management. [J Pediatr Ophthalmol Strabismus. 2017;54:e31-e33.].

Clinical and histopathological features of adenomas of the ciliary pigment epithelium.

PURPOSE: Adenomas of the ciliary pigment epithelium (CPE) are rare benign tumours which have mainly to be differentiated from malignant ciliary body melanomas. Here we report on a consecutive series of patients with CPE adenomas and describe their characteristics. METHODS: The retrospective hospital-based case series study included all patients who were consecutively operated for CPE adenomas. RESULTS: Of the 110 patients treated for ciliary body tumours, five patients (4.5%) had a CPE adenoma. Mean age was 59.0 ± 9.9 years (range: 46-72 years). Mean tumour apical thickness was 6.6 ± 1.7 mm. Tumour colour was mostly homogenously brown to black, and the tumour surface was smooth. The tumour masses pushed the iris tissue forward without infiltrating iris or anterior chamber angle. Sonography revealed an irregular echogram with sharp lesion borders and signs of blood flow in Color Doppler flow imaging. Ultrasonographic biomicroscopy demonstrated medium-low internal reflectivity and acoustic attenuation. In magnetic resonance imaging (MRI), the tumours as compared to brain were hyperintense on T1-weighted images and hypointense on T2-weighted images. Tumour tissue consisted of cords and nests of pigment epithelium cells separated by septa of vascularized fibrous connective tissue, leading to a pseudo-glandular appearance. The melanin granules in the cytoplasm were large and mostly spherical in shape. In four patients, the tumours were hyperpigmented. Tumour cells were large with round or oval nuclei and clearly detectable nucleoli. CONCLUSIONS: These clinical characteristics of CPE adenomas, such as homogenous dark brown colour, smooth surface, iris dislocation and anterior chamber angle narrowing but no iris infiltration, segmental cataract, pigment dispersion, and, as compared to brain tissue, hypointensity and, as compared to extraocular muscles or lacrimal gland, hyperintensity on T2-weighted MRI images, may be helpful for the differentiation from ciliary body malignant melanomas.

Primary adenocarcinoma of pigmented ciliary epithelium in a phthisical eye.

We report a case of adenocarcinoma of the pigmented ciliary epithelium arising in a phthisical eye. A 92-year-old man who initially presented with severe ocular pain had calcification extending from the posterior pole to ciliary body on B-scan ultrasonography to a degree not previously reported. We highlight the importance of screening for intraocular neoplasms in adults with a long-standing phthisical eye.

[A case of familial pupillary iris pigment epithelium cysts]. / Przypadek rodzinnego wystepowania zrenicznych cyst nablonka barwnikowego teczówki.

Iris cysts are rare lesions. In some cases, they may mimic melanoma and cause diagnostic difficulties. Through imaging tests such as ultrasound biomicroscopy we can evaluate the structure changes and determine the location for easy diagnosis. Iris cysts often require only observation. In some cases specialist treatment is necessary. One of the methods is transpupillary cystotomy perform using the Nd:YAG laser. The paper presents the case of familial pupillary cysts of the iris pigment epithelium in a father and a daughter.

Amblyopia secondary to iris cyst.

CLINICAL CASE: A 5 year-old child diagnosed with moderate anisometropic amblyopia secondary to primary cyst of iris pigment epithelium. He was evaluated with ultrasound biomicroscopy (BMU) and optical coherence tomography (OCT) of anterior segment. DISCUSSION: The OCT, although with some limitations, is a useful tool to study the anterior segment. It is probably more recommendable than BMU in the childhood.

[Partially pigmented vitreous cyst]. / Kyste vitréen partiellement pigmenté.

Free-floating iris pigment epithelial cysts in the vitreous are very rare. We report the case of a partially pigmented cyst found on routine examination in the vitreous of a 51-year-old asymptomatic patient. Usually, non-pigmented cysts are derived from the hyaloid system. Pigmented cysts, however, are derived from the iris pigment epithelium. When they are not dislodged, these cysts can be confused with iris melanoma or primary stromal cysts. Treatment is usually unnecessary, unless they become symptomatic.

Treatment of peripheral pigment epithelium iris cyst with SLT laser.

PURPOSE: To report a case of a peripheral pigment epithelium iris cyst treated with selective laser trabeculoplasty laser. CASE REPORT: A 15-year-old girl applied to our clinic with the complaint of painless blurred vision in the right eye. Gonioscopy through a widely dilated pupil revealed a clear, oval, pale brown, semitransparent cyst that was located in the inferotemporal aspect of the iris. The cyst was touching the lens, and the lens was pushed superonasally by the cyst. The iris cyst was treated with selective laser trabeculoplasty laser. A small iris notch emerged at the 8-o'clock position of the pupillary margin. Only mild flare and minimal pigment dispersion were documented postoperatively, which resolved in a few days with a topical corticosteroid. CONCLUSIONS: Selective laser trabeculoplasty laser may be considered as a potentially useful and relatively less invasive technique in the treatment of peripheral pigment epithelium iris cysts.

Effect of primary iris and ciliary body cyst on anterior chamber angle in patients with shallow anterior chamber.

OBJECTIVE: To evaluate the prevalence of primary iris and/or ciliary body cysts in eyes with shallow anterior chamber and their effect on the narrowing of the anterior chamber angle. METHODS: Among the general physical check-up population, subjects with shallow anterior chambers, as judged by van Herick technique, were recruited for further investigation. Ultrasound biomicroscope (UBM) was used to detect and measure the cysts located in the iris and/or ciliary body, the anterior chamber depth (ACD), the angle opening distance at 500 µm (AOD500), and the trabecular-iris angle (TIA). A-scan ultrasonography was used to measure the ocular biometry, including lens thickness, axial length, lens/axial length factor (LAF), and relative lens position (RLP). The effect of the cyst on narrowing the corresponding anterior chamber angle and the entire angle was evaluated by the UBM images, ocular biometry, and gonioscopic grading. The eye with unilateral cyst was compared with the eye without the cyst for further analysis. RESULTS: Among the 727 subjects with shallow anterior chamber, primary iris and ciliary body cysts were detected in 250 (34.4%) patients; among them 96 (38.4%) patients showed unilateral single cyst, 21 (8.4%) patients had unilateral double cysts, and 42 (16.8%) patients manifested unilateral multiple and multi-quadrants cysts. Plateau iris configuration was found in 140 of 361 (38.8%) eyes with cysts. The mean size of total cysts was (0.6547 ± 0.2319) mm. In evaluation of the effect of the cyst size and location on narrowing the corresponding angle to their position, the proportion of the cysts causing corresponding angle narrowing or closure among the cysts larger than 0.8 mm (113/121, 93.4%) was found to be significantly higher than that of the cysts smaller than 0.8 mm (373/801, 46.6%), and a significant higher proportion was also found in the cysts located at iridociliary sulcus (354/437, 81.0%) than in that at the pars plicata (131/484, 27.1%). In evaluating the effect of the cyst on the entire anterior chamber angle, the eyes with multiple and multi-quadrants cysts manifested significant narrowing of the entire anterior chamber angle as compared with the eyes without cysts, based on the data analysis in comparison of TIA, AOD500, and gonioscopic grading evaluation. The unilateral single or double cysts in the eyes had no significant effect on narrowing of anterior chamber angle as compared with eyes without cysts. The iris and/or ciliary body cysts did not seem to affect the axial length, ACD, lens thickness, RLP, LAF. CONCLUSIONS: The prevalence of primary iris and ciliary body cyst was 34.4% in the subjects with shallow anterior chamber. The cysts larger than 0.8 mm, locating at iridociliary sulcus, or multiple and extensive cysts were inclined to cause the angle narrowing or closure.

Complete ring cyst of iris pigment epithelium documented by ultrasound biomicroscopy.

The authors report a complete ring cyst of the iris pigment epithelium confirmed by ultrasound biomicroscopy. In this case, ultrasound biomicroscopy effectively excluded the differential diagnoses of ring melanoma of the iris and multiple separate cysts of the iris pigment epithelium.

In-vivo PET imaging of implanted human retinal pigment epithelium cells in a Parkinson's disease rat model.

BACKGROUND AND AIM: Researchers find that monitoring the differentiation of implanted cells in vivo is difficult. This study was designed to show that it is possible to track the efficacy of transplanted human retinal pigment epithelial cells (RPE cells) in a rat model of Parkinson's disease by using positron emission tomography (PET). METHODS: RPE cells or normal saline were injected into striatum of the injured side of the rat model in treated and control groups, respectively. PET imaging of both groups was undertaken before transplantation and at intervals afterwards, using C-raclopride and C-beta-CFT as the markers. Observation of the rats' behaviour and immunofluorescence confocal microscopy were also used to prove the PET results. RESULTS: PET studies showed increased accumulation of C-raclopride and decreased C-beta-CFT in the injured side of striatum in both groups. C-raclopride decreased along with a concomitant increase of C-beta-CFT after transplantation in the treated group. The changes shown by the PET studies paralleled the behavioural states and confocal microscopy observations in the treated animals. CONCLUSION: These results suggest that even a clinical PET scanner could, to a certain extent, provide some information on the existence and in-vivo differentiation of RPE cells in a rat model of Parkinson's disease.