Background: Corticosteroids are widely used in medicine. Few cases of central serous chorioretinopathy (CSC) have been reported following topical corticosteroid administration. We describe the first case of pediatric CSC related to topical corticosteroid administration. Case presentation: A 14-year-old boy presented with decreased vision, pigment epithelial detachments, and serous retinal detachments in the right eye after starting treatment for atopic dermatitis with Betamethasone Valerate 0.1% topical ointment. His condition resolved 2 weeks after discontinuing the steroid and administering Bromfenac 0.9 mg/ml eyedrops. Conclusions: Although the pathogenesis of CSC is poorly understood, ophthalmologists should be informed about the potential link between CSC and topical corticosteroid treatment, and they should be aware that CSC might, albeit infrequently, affect children

Donati, Simone

Fontanel, Liviana

Nucci, Paolo

Radice, Paolo

Ranno, Stefano

Ruggiero, Edoardo

English

Archivio istituzionale della ricerca - Università dell'Insubria

Ranno et al. Italian Journal of Pediatrics            (2023) 49:1  https://doi.org/10.1186/s13052-022-01386-4CASE REPORT© The Author(s) 2023. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http:// creat iveco mmons. org/ licen ses/ by/4. 0/. The Creative Commons Public Domain Dedication waiver (http:// creat iveco mmons. org/ publi cdoma in/ zero/1. 0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.Open AccessCentral Serous Chorioretinopathy in a 14-year-old atopic boy: a case reportStefano Ranno1, Liviana Fontanel2*  , Edoardo Ruggiero3, Paolo Nucci4, Paolo Radice1 and Simone Donati1,2 Abstract Background: Corticosteroids are widely used in medicine. Few cases of central serous chorioretinopathy (CSC) have been reported following topical corticosteroid administration. We describe the first case of pediatric CSC related to topical corticosteroid administration.Case presentation: A 14-year-old boy presented with decreased vision, pigment epithelial detachments, and serous retinal detachments in the right eye after starting treatment for atopic dermatitis with Betamethasone Valerate 0.1% topical ointment. His condition resolved 2 weeks after discontinuing the steroid and administering Bromfenac 0.9 mg/ml eyedrops.Conclusions: Although the pathogenesis of CSC is poorly understood, ophthalmologists should be informed about the potential link between CSC and topical corticosteroid treatment, and they should be aware that CSC might, albeit infrequently, affect children.Keywords: Central serous Chorioretinopathy case report, Corticosteroid adverse effects, Corticosteroids, Pediatric ophthalmologyBackgroundCentral serous chorioretinopathy (CSC) is an ocular disease that causes an idiopathic serous detachment of the retina and a fluid leakage from the choroid through a defect in the retinal pigment epithelium (RPE) outer blood-retina barrier [1]. CSC is a common cause of vis-ual impairment in the working-age population and has been estimated as the fourth most frequent non-surgical retinopathy after age-related macular degeneration, dia-betic retinopathy, and retinal vein occlusion [2]. Patients often experience loss of central vision, central scotoma, micropsia or metamorphopsia. Visual acuity may only be moderately decreased, and there may be a hyper-opic shift [2]. Although many cases resolve spontane-ously with minimal sequelae, 30-45% of patients have recurrence and a poorer visual prognosis [3]. CSC typi-cally affects men aged 25 to 50 years, but it may occur in women or older individuals [3]. Although the patho-genesis remains unknown, risk factors include type A personality, Cushing disease, pregnancy, and corticoster-oid use [1]. Actually, there are only five reported pedi-atric CSC cases [4–8]. To the best of our knowledge, we report the first pediatric CSC case associated with topi-cal corticosteroid use.Case presentationA 14-year-old boy presented with a 2-week history of decreased visual acuity in the right eye.The boy suffered from atopic dermatitis (AD) with facial and scalp involvement and had been treated by a dermatologist since he was 5 years old. The disease had been well controlled for several years by skin hydration with soaking baths and use of topical ointments.However, skin lesions had worsened in the past 2 years and the patient was prescribed Betamethasone Valerate *Correspondence:  simone.donati@uninsubria.it2 University of Insubria, Department of Medicine and Surgery, Via Guicciardini 9, 21100 Varese, ItalyFull list of author information is available at the end of the articlePage 2 of 4Ranno et al. Italian Journal of Pediatrics            (2023) 49:1 0.1% topical ointment 3 times a day. Although the patient was instructed to stop the therapy after 5 weeks, he continued using the ointment 3 times a day for about 6 months, after noticing significant improvement.He presented at our attention complaining decreased visual acuity in the right eye. On ocular examination, his best corrected visual acuity measured 20/25 in his right eye and 20/20 in his left eye. Intraocular pressure was normal and anterior segment examination in each eye was unremarkable. Posterior segment examination of the right eye showed a large serous retinal detachment involving the posterior pole. Posterior segment examina-tion of the left eye was normal.Therefore, we decided to perform a macular optical coherence tomography (OCT) examination (Spectralis HRA; Heidelberg Engineering, Heidelberg, Germany). OCT examination in the right eye (Fig.  1) revealed the presence of a detachment of the neurosensory retina in macular region, associated with elongated photoreceptor segments and irregular RPE. Multiple associated pigment epithelial detachments were located inferonasally and inferotemporally to the fovea. In the left eye OCT exami-nation showed normal finds.Lastly, the blood pressure values were within normal ranges and the patient claimed to have a peaceful mind.Structural retinal findings associated to clinical signs and history addressed the diagnosis to the CSC, probably linked to the persistent topical steroid treatment.We decided to treat the patients with Bromfenac 0.9 mg/ml eye drops 3 times a day and to discontinue Betamethasone Valerate ointment. The therapy was well-tolerated by the patient and no side effects were reported.At 2 weeks follow-up, OCT examination showed a complete resolution of neuroepithelium detachment (Fig.  2), associated to a restoration of normal visual acuity.Discussion and conclusionsThis atypical case of CSC in a pediatric patient resolved promptly on cessation of the topical steroid and adminis-tration of Bromfenac eye drops.The pathophysiology of CSC is poorly understood. Guyer and colleagues suggested that the pathogenesis of CSC may be choroidal vascular hyperpermeability. They noted diffuse hyperpermeability around active leak-age sites seen with indocyanine green videoangiogra-phy (ICG-V) but not with fluorescein angiography (FA). Therefore, they concluded that hyperpermeability was at the level of the choroid rather than the RPE [9]. An alter-native theory suggests that CSC results from dysfunction of the RPE which causes a reverse in fluid movement in a chorioretinal direction [10].CSC induced by the systemic use of steroids was first reported in 1984 in two patients that developed serous macular detachment upon initiation of systemic Beta-methasone therapy for retrobulbar neuritis [11]. How-ever, the best evidence for an association between corticosteroid use and CSC comes from two large, retro-spective case-control studies [12, 13]. Tittl and colleagues conducted the first of these, studying systemic factors associated with CSC [12]. This study included a total of 230 CSC cases and 230 age and sex-matched controls. They found that 21 CSC subjects were using corticoster-oid medications, whereas 7 control subjects were using Fig. 1 Right eye, Baseline. Infrared funduscopic image (left) and macular OCT B-scan (right). Presence of subretinal fluid causing a circumscribed area of serous retinal detachment in the macular region (white arrow), associated with elongated photoreceptor segments and irregular retinal pigment epithelium layerPage 3 of 4Ranno et al. Italian Journal of Pediatrics            (2023) 49:1  corticosteroids. This difference yielded an odds ratio of 3.2 (95% CI 1.3 to 7.70, P = 0.0063). Carvalho-Recchia et  al. published the first report of a consecutive series of patients with acute CSC studied prospectively for an association with corticosteroids [13]. They found a sta-tistically significant difference in corticosteroid exposure between study patients and controls.The mainstay of Atopic Dermatitis treatment is repre-sented by topical corticosteroids, while several routes of steroid administration have been discussed in the litera-ture. These medications reduce inflammation and pru-ritus primarily by inhibiting the transcriptional activity of various proinflammatory genes. Topical corticoster-oids are available in a wide range of potencies, from the least potent Group 1 preparations (e.g. Hydrocortisone 1% ointment), to the most potent Group 7 preparations (e.g. Clobetasol Propionate 0.05% ointment). The greater the potency of topical corticosteroid used, the greater the risk of systemic and local side effects [14]. Betametha-sone Valerate 0.1% is in group 3 (high potency topical corticosteroids).Despite that, it was suggested that steroid-induced CSC may be related to an idiosyncratic response in selected vulnerable individuals rather than to a dose-dependent effect, since very low doses can induce CSC episodes [15].CSC has been associated with topical steroid use in several case reports. In 2004, Karadimas and colleagues reported two cases of presumed topical steroid-associ-ated CSC [16]. Fernandez and colleagues reported a sus-pected case of topical steroid-associated CSC, in which a 43-year-old female developed CSC after 1 month of topical steroid use for lichen planus [17]. Ezra and col-leagues also reported a case of a 25-year-old male with psoriasis and 15 years of topical steroid use. He experi-enced a single episode of CSC with resolution upon ster-oid cessation [18].While CSC has previously been linked to sys-temic corticosteroid use, it is rarely linked to topical administration.Idiopathic CSC in children has been reported in a few case reports [5–8], however this condition has never been linked to topical corticosteroid therapy in young patients.In light of this, we assume that this may be the first pediatric case of CSC related to transdermal steroid treatment.Poor imaging is a limitation of this case report, because CSC diagnosis is typically based on a serous retinal detachment described on OCT examination and con-firmed by FA, which reveals early-phase localized dye leakage and late-phase dye pooling under the sensory retina. However, we chose not to perform FA on our young patient because OCT scans clearly revealed the diagnosis.In conclusion, it should be kept in mind that CSC can affect children as well as adults, and this case outlines the importance for ophthalmologists to carefully assess CSC patients for local and systemic corticosteroid use.AbbreviationsAD: Atopic dermatitis; CSC: Central serous chorioretinopathy; FA: Fluorescein angiography; ICG-V: Indocyanine green videoangiography; OCT: Optical coherence tomography; RPE: Retinal pigment epithelium.Fig. 2 Right eye, Follow up at 2 weeks after stopping corticosteroid ointment. Infrared funduscopic image (left) and macular OCT B-scan (right). Complete remission of neuroepithelium detachment (white arrow), with resolution of subretinal fluid and photoreceptors - retinal pigment epithelium irregularityPage 4 of 4Ranno et al. Italian Journal of Pediatrics            (2023) 49:1 • fast, convenient online submission •  thorough peer review by experienced researchers in your field•  rapid publication on acceptance•  support for research data, including large and complex data types•  gold Open Access which fosters wider collaboration and increased citations  maximum visibility for your research: over 100M website views per year •  At BMC, research is always in progress.Learn more biomedcentral.com/submissionsReady to submit your research ?  Choose BMC and benefit from: AcknowledgementsNot applicable.Authors’ contributionsSR and ER collected data; SR, LF and ER drafted the work; PN and SD substan-tively revised it; PN, SD and PR supervised the redaction and the discussion of the clinical case. All authors read and approved the final manuscript. All authors agreed both to be personally accountable for the author’s own contributions and to ensure that questions related to the accuracy or integrity of any part of the work, even ones in which the author was not personally involved, are appropriately investigated, resolved, and the resolution docu-mented in the literature.FundingThis manuscript was supported by University of Insubria Funds for research.Availability of data and materialsThe datasets used and analyzed during the current study are available from the corresponding author on reasonable request.DeclarationsEthics approval and consent to participateAll methods were performed in accordance with the ethical standards as laid down in the Declaration of Helsinki and its later amendments or comparable ethical standards. Written informed consent to discuss and publish this case was obtained from the parents of the patient.Authors confirm they have obtained ethical approval and consent for this case report.This case report has been granted an exemption from requiring ethics approval from the Ethics Committee of Insubria University, Varese.Consent for publicationWritten informed consent for publication of clinical details and clinical images was obtained from the parents of the patient. A copy of the consent form is available for review by the Editor of this journal.Competing interestsThe authors declare that they have no competing interests.Author details1 Ophthalmology Department, Circolo and Fondazione Macchi Hospital, ASST Sette Laghi, Varese, Italy. 2 University of Insubria, Department of Medicine and Surgery, Via Guicciardini 9, 21100 Varese, Italy. 3 Eye Vision Ophthalmology Center, Como, Italy. 4 Department of Clinical Sciences and Community Health, University of Milan, Milan, Italy. 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Central Serous Chorioretinopathy in a 14-year-old atopic boy: a case report

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Central Serous Chorioretinopathy in a 14-year-old atopic boy: a case report

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