Spontaneous Resolution of Central Serous Chorioretinopathy in a Patient With Congenital Retinal Macrovessel
2011; Lippincott Williams & Wilkins; Volume: 124; Issue: 25 Linguagem: Inglês
10.1161/circulationaha.111.029835
ISSN1524-4539
Autores Tópico(s)Vascular Malformations Diagnosis and Treatment
ResumoHomeCirculationVol. 124, No. 25Spontaneous Resolution of Central Serous Chorioretinopathy in a Patient With Congenital Retinal Macrovessel Free AccessBrief ReportPDF/EPUBAboutView PDFView EPUBSections ToolsAdd to favoritesDownload citationsTrack citationsPermissions ShareShare onFacebookTwitterLinked InMendeleyReddit Jump toFree AccessBrief ReportPDF/EPUBSpontaneous Resolution of Central Serous Chorioretinopathy in a Patient With Congenital Retinal Macrovessel Francisco J. Ascaso, MD, PhD Francisco J. AscasoFrancisco J. Ascaso From the Department of Ophthalmology, Lozano Blesa University Clinic Hospital and Aragon Health Sciences Institute, Zaragoza, Spain. Originally published20 Dec 2011https://doi.org/10.1161/CIRCULATIONAHA.111.029835Circulation. 2011;124:e904–e905An otherwise healthy 46-year-old man complained with sudden visual loss and a black spot in his left eye. The patient referred a recent stressful episode and was not using corticosteroids. Best-corrected visual acuity was 20/20 and 20/40 in the right eye and left eye, respectively. Applanation tonometry and anterior segment examination of both eyes were unremarkable.Fundus examination of the left eye revealed a serous elevation of the retina in the macular area, with loss of foveal reflex. There was also a peculiar, large aberrant superotemporal venule branching inferiorly and crossing the horizontal raphe with several tributaries, two of which passed through the macula, but not involving the fovea. The right fundus was normal. Fundus fluorescein angiography (FFA) of the left eye showed a few microaneurysms in the capillary network of the perifoveal area and an ink blot pattern of leak in the temporal juxtafoveal area with diffuse spreading of the stain accumulating under the retina at the late stage with the typical aspect of central serous chorioretinopathy (CSCR) (Figure, part A). Optical coherence tomography (OCT) revealed a serous retinal detachment (RD) of the macular area without vitreous traction (Figure, part B). The patient showed elevated 24-hour urine cortisol, although 24-hour urine catecholamines were normal. A diagnosis of acute CSCR with congenital retinal macrovessel was made. In the present case, the decreased visual acuity was secondary to the serous RD that occurred suddenly at 46 years of age, despite the congenital nature of the arteriovenous abnormality. One month later, the visual acuity in the patient's left eye improved to 20/20. At that time, no dye leakage was detectable on FFA (Figure, part C), and the serous RD resolved spontaneously (Figure, part D).Download figureDownload PowerPointFigure. A, Fundus fluorescein angiography (FFA) of the left eye showing a large aberrant superotemporal venule crossing the macula (large arrow), a few microaneurysms in the capillary network of the perifoveal area (small arrows), and an ink blot pattern of leak in the temporal juxtafoveal area (arrowhead). B, Optical coherence tomography (OCT) demonstrated a serous macular detachment due to intraretinal fluid accumulation (arrowheads). C, One month later, the visual acuity improved to 20/20, and no dye leakage was detectable on FFA. D, OCT revealed spontaneous resolution of intraretinal fluid with restoration of the foveal depression (arrowhead).Although the first description of an isolated enlarged vessel traversing through the macula was made by Mauthner in 1869, the term "congenital retinal macrovessel" (CRM) was coined in 1982 by Brown et al. CRM is a rare and typically unilateral abnormality of the retinal circulation, generally a single venule, which, in an aberrant manner, crosses the posterior pole or the fovea itself. It was classified in group 1 of congenital arteriovenous communications in the retina.1 Prevalence of CRMs is approximately 1/200 000. Although the underlying cause is unknown, it is believed these anomalies occur around the 15th to 16th week of gestation, during the differentiation and canalization of cords of mesenchymal cells invading the retinal nerve fiber layer.2 They are typically benign and are often detected in a routine exploration. Visual disturbance in the involved eye is infrequent and has been attributed to hemorrhage, to foveal cyst, or to the mere presence of the aberrant vessel in the foveal area.In the present case, the decreased visual acuity was secondary to the serous RD that occurred suddenly at 46 years of age, even though the arteriovenous abnormality seemed to be congenital. A differential diagnosis must be made with other vascular entities such as vascular loops, congenital venous tortuosity or secondary to venous obstruction, arteriovenous communications, and even with tumors such as branch-shaped angioma, retinal capillary hemangioma, retinoblastoma, and choroidal melanoma.2 FFA proved to be valuable in establishing a definitive diagnosis. The clinical findings of unilateral neurosensory RD in the macular region and a leakage point of fluorescein that appeared early and spread over time diffusely, under the macula, are characteristic of CSCR.Development of serous RDs and decreased visual acuity have been associated with CRMs after rapid changes in gravitational forces (eg, bungee cord jumping and roller coaster rides).3 This combined presentation of serous RD with CRM was also attributed to vitreous traction in a case that resolved spontaneously following posterior hyaloids detachment.1 Recently, Kumar et al described the only case of CSCR with CRM; however, that patient was lost to follow-up.4 Ours is the first well-documented case report of a spontaneously resolved CSCR associated with CRM. There was no trauma, vitreous traction, or arteriovenous communication that could account for the occurrence of CSCR.The etiologic relationship between CSCR and CRM remains to be determined. Although they could be coincidental findings, microalterations of the blood-retinal barrier might take place around the aberrant macrovessel and be associated with the anomalies of the perifoveal capillary bed and the serum levels of stress hormones such as cortisol and catecholamines typically increased in patients with CSCR.DisclosuresNone.FootnotesCorrespondence to Francisco J. Ascaso, MD, Department of Ophthalmology, University Clinic Hospital, San Juan Bosco 15, ES-50009 Zaragoza, Spain. E-mail [email protected]comReferences1. Arai J, Kasuga Y, Koketsu M, Yoshimura N. Development and spontaneous resolution of serous retinal detachment in a patient with a congenital retinal macrovessel. Retina. 2000; 20:674–676.CrossrefMedlineGoogle Scholar2. Gurwood SA, Bailey JT, Pelino JC. Congenital retinal macrovessel: a case report. Optometry. 2001; 72:597–602.MedlineGoogle Scholar3. Beatty S, Goodall K, Radford R, Lavin MJ. Decompensation of a congenital retinal macrovessel with arteriovenous communications induced by repetitive roller coaster rides. Am J Ophthalmol. 2000; 130:527–528.CrossrefMedlineGoogle Scholar4. Kumar V, Ghosh B, Raina U, Goel N. Central serous chorioretinopathy in a patient with congenital retinal macrovessel. Can J Ophthalmol. 2009; 44:e57.CrossrefMedlineGoogle Scholar Previous Back to top Next FiguresReferencesRelatedDetailsCited By Savastano A, Tartaro R, Savastano M and Rizzo S (2019) MACULAR MACROVESSELS ASSOCIATED WITH SELF-LIMITING LEAKAGE IN A YOUNG PATIENT, RETINAL Cases & Brief Reports, 10.1097/ICB.0000000000000572, 13:3, (248-250), . Pichi F, Freund K, Ciardella A, Morara M, Abboud E, Ghazi N, Dackiw C, Choudhry N, Souza E, Cunha L, Arevalo J, Liu T, Wenick A, He L, Villarreal G, Neri P and Sarraf D (2018) Congenital Retinal Macrovessel and the Association of Retinal Venous Malformations With Venous Malformations of the Brain, JAMA Ophthalmology, 10.1001/jamaophthalmol.2018.0150, 136:4, (372), Online publication date: 1-Apr-2018. Medina-Tapia A, Molina-Sócola F, Llerena-Manzorro L, López-Herrero F, Castilla-Martino M, Martínez-Borrego A and Sánchez-Vicente J (2017) Macrovaso retiniano asociado a telangiectasias periféricas retinianas e isquemia retiniana, Archivos de la Sociedad Española de Oftalmología, 10.1016/j.oftal.2016.10.012, 92:7, (338-342), Online publication date: 1-Jul-2017. Medina-Tapia A, Molina-Sócola F, Llerena-Manzorro L, López-Herrero F, Castilla-Martino M, Martínez-Borrego A and Sánchez-Vicente J (2017) Congenital retinal macrovessel associated with retinal peripheral telangiectasia and retinal ischaemia, Archivos de la Sociedad Española de Oftalmología (English Edition), 10.1016/j.oftale.2017.03.009, 92:7, (338-342), Online publication date: 1-Jul-2017. Lee K, Oh J, Kim S and Huh K (2013) Central Serous Chorioretinopathy in a Patient with Retinal Macrovessel, Journal of the Korean Ophthalmological Society, 10.3341/jkos.2013.54.7.1139, 54:7, (1139), . December 1, 2011Vol 124, Issue 25 Advertisement Article InformationMetrics © 2011 American Heart Association, Inc.https://doi.org/10.1161/CIRCULATIONAHA.111.029835PMID: 22184046 Originally publishedDecember 20, 2011 PDF download Advertisement SubjectsDiagnostic TestingImaging
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