Central retinal artery occlusion (CRAO) is a rare but blinding disorder. We present a case of a 81-year-old woman with multiple cardiovascular comorbidities admitted to the emergency department due to sudden, painless vision loss on left eye (oculus sinister (OS)) on awakening. The patient also reported long standing fatigue associated with effort that started 4 months before admission. She presented best corrected visual acuity of counting fingers OS. Funduscopy OS revealed macular oedema with cherry red spot pattern. Blood cultures came positive for Streptococcus gallolyticus in the context of a bacteremia and native mitral valve vegetation identified on transoesophageal echocardiography. CRAO of embolic origin was admitted in the context of an infective endocarditis. CRAO can be the first manifestation of a potentially fatal systemic condition and thus multidisciplinary approach is warranted with close collaboration between ophthalmologists and internists in order to provide proper management and the best possible treatment.info:eu-repo/semantics/publishedVersio

Azevedo, L

Hipólito-Fernandes, D

Serras-Pereira, R

Vieira, L

English

PubMed

Repositório do Centro Hospitalar de Lisboa Central, EPE

1Serras- Pereira R, et al. BMJ Case Rep 2020;13:e235763. doi:10.1136/bcr-2020-235763Case reportCentral retinal artery occlusion from Streptococcus gallolyticus endocarditisRita Serras- Pereira,1 Diogo Hipolito- Fernandes   ,1 Luísa Azevedo,2 Luísa Vieira1 Reminder of important clinical lessonTo cite: Serras- Pereira R, Hipolito- Fernandes D, Azevedo L, et al. BMJ Case Rep 2020;13:e235763. doi:10.1136/bcr-2020-2357631Department of Ophthalmology, Centro Hospitalar de Lisboa Central, EPE, Lisboa, Portugal2Department of Internal Medicine, Centro Hospitalar de Lisboa Central, EPE, Lisboa, PortugalCorrespondence toDr Diogo Hipolito- Fernandes;   cdiogo777@ gmail. comAccepted 3 August 2020© BMJ Publishing Group Limited 2020. No commercial re- use. See rights and permissions. Published by BMJ.SUMMARYCentral retinal artery occlusion (CRAO) is a rare but blinding disorder. We present a case of a 81- year- old woman with multiple cardiovascular comorbidities admitted to the emergency department due to sudden, painless vision loss on left eye (oculus sinister (OS)) on awakening. The patient also reported long standing fatigue associated with effort that started 4 months before admission. She presented best corrected visual acuity of counting fingers OS. Funduscopy OS revealed macular oedema with cherry red spot pattern. Blood cultures came positive for Streptococcus gallolyticus in the context of a bacteremia and native mitral valve vegetation identified on transoesophageal echocardiography. CRAO of embolic origin was admitted in the context of an infective endocarditis. CRAO can be the first manifestation of a potentially fatal systemic condition and thus multidisciplinary approach is warranted with close collaboration between ophthalmologists and internists in order to provide proper management and the best possible treatment.BACKGROUNDCentral retinal artery occlusion (CRAO) is a rare but blinding disorder characterised by an obstruc-tion of retinal blood flow that may be due to an embolus, thrombus, vasculitis, traumatic vessel wall damage or spasm. The major risk factors can be divided in arteritic and non- arteritic in nature, the latter representing more than 90% of cases, and CRAO can be further classified as permanent, transient and with cilioretinal artery sparing, an artery present in 15%–30% of the population.1 2 Incidence of non- arteritic CRAO is approximately 1–2 in 100 000.3 Embolism from an atherosclerotic plaque is the most frequent cause and risk factors are similar to those for cardiovascular and cerebro-vascular events: hypertension, diabetes mellitus, tobacco use, carotid and coronary artery disease.1 In the European Assessment Group for Lysis in the Eye trial, 73% of patients with diagnosis of CRAO had hypertension, 40% had at least 70% of carotid stenosis, 22% had coronary artery disease, 20% atrial fibrillation and 17% valvular heart disease and in patients with no prior history of cardio-vascular risk factors, at least one was identified in 78% of patients.3 CRAO usually presents with acute and painless loss of monocular vision with 80% of patients having a final visual acuity (VA) of counting fingers or worse.1 2 Classical fundo-scopic findings include retinal oedema, cherry red spot, retinal arteriolar attenuation and a normal optic disc.3 Occasionally, emboli may be seen in the central retinal artery (CRA) or its branches.3 Visual outcomes in CRAO are typically poor, although with some variability, depending on factors such as duration of CRAO, the presence of cilioretinal artery and aetiology of the emboli.3 Management of CRAO is independent from its cause and can be divided in acute, with attempts to restore ocular perfusion; subacute, with prevention of secondary ocular complications such as neovascularisation and glaucoma; and long term, with prevention of other end- organ ischaemic events.1Infective endocarditis (IE) is likewise a rare condi-tion, potentially life- threatening, with mortality rates as high as 40% annually.4 Systemic emboli-sation can occur in up to 80% of patients with IE and the risk seems to be greater with mitral valve involvement.1 4 5 Valvular vegetations can release emboli and lead to amaurosis fugax or retinal artery occlusion.4 CRAO is a very rare complication of IE. Streptococcus gallolyticus, a gram- positive, non- enterococcal, group D Streptococcus from the S. bovis group, is an opportunistic pathogen asymp-tomatically found in the gastrointestinal tract of humans and an increasing cause of bacteremia and IE in the elderly.5 6 It is estimated to be the aetio-logic agent of IE in 11%–14% of cases.5To the best of our knowledge, this is the first reported case of CRAO due to S. gallolyticus IE and highlights the importance of multidisciplinary approach.CASE PRESENTATIONA 81- year- old woman with prior history of valvular cardiomyopathy (moderate mitral and aortic valve stenosis), paroxysmal atrial fibrillation under oral anticoagulation, implanted pacemaker, type II diabetes mellitus, hypertension, dyslipidaemia, obesity and peripheral venous insufficiency was admitted to the emergency department of a tertiary care hospital due to sudden, painless loss of VA on the left eye (OS) on awakening. On ophthalmolog-ical examination, she presented best corrected VA (BCVA) of 20/25 right eye (oculus dextrus (OD)) and counting fingers OS. Pupils were isochoric and isoreactive, no restrictions on extraocular movements. On biomicroscopy, bilateral pseu-dophakia was identified. Funduscopy OS revealed macular oedema with cherry red spot pattern. The clinical diagnosis of CRAO OS was made and the patient was immediately treated with systemic Protected by copyright. on October 12, 2020 at Hospitais da Universidade Coimbra.http://casereports.bmj.com/BMJ Case Rep: first published as 10.1136/bcr-2020-235763 on 2 September 2020. Downloaded from 2 Serras- Pereira R, et al. BMJ Case Rep 2020;13:e235763. doi:10.1136/bcr-2020-235763Reminder of important clinical lessonacetazolamide and isosorbide dinitrate, ocular massage and topical ocular hypotensives.On further questioning, the patient also reported long standing fatigue associated with effort that started 4 months before admission and worsened the previous week, when she started feeling febrile. On physical examination, the patient was alert, attentive and oriented; febrile (temperature 38°C); haemo-dynamically stable; oxygen saturation of 97% (at room air) and on auscultation, cardiac sounds were rhythmic and a systolic murmur was evident.On complementary examination, blood tests revealed normo-cytic normochromic anaemia and increased inflammatory markers (table 1). ECG showed ventricular pacing rhythm and chest radiography showed an increased cardiothoracic ratio. Cranial CT did not reveal any acute parenchymatous brain lesions.The patient was admitted to the internal medicine department for further aetiologic investigation. A few days after admission, blood cultures came positive for S. gallolyticus spp gallolyticus sensitive to penicillin and a native mitral valve vegetation was identified on transoesophageal echocardiography. CRAO of embolic origin was admitted in the context of an IE. Optical coherence tomography confirmed an ischaemic and oedematous internal retina compatible with CRAO diagnosis (figure 1)TREATMENTThe patient was treated with intravenous ceftriaxone 2 g/day for 28 days and was discharged after full clinical stabilisation, 33 days after admission.OUTCOME AND FOLLOW-UPThe ophthalmology department collaborated closely with the internal medicine department. The patient refused to be submitted to any further ophthalmologic examinations or treat-ments but maintained periodic follow- up and was stable since discharge. At the time of the last visit, BCVA OS was counting fingers, anterior segment without significant changes, intraoc-ular pressure (IOP) within normal limits and retinal atrophy with optic disc pallor on funduscopy, without signs of iris, optic disc or retinal neovascularisation.DISCUSSIONCRAO due to IE is a rare event with few cases reported in the literature. The authors report the first case of CRAO due to S. gallolyticus endocarditis. CRAO is an ophthalmologic emer-gency and can be the first manifestation of a potentially fatal systemic condition, such as IE, with overlapping risk factors for major cardiovascular and cerebrovascular events.3Table 1 Laboratory tests on admission and on dischargeLaboratory tests on admissionLaboratory tests on dischargeErythrocytes 3.03×1012/L↓ 3.62 1012/L↓Haemoglobin 8.5×10 g/L↓ 10.4×10 g/L↓Hematocrit 26.3%↓ 32.5%↓Mean cell volume 86.8 fL 89.8 fLMean cell haemoglobin 28.1 pg 28.7 pgRed cell distribution width 13.4% 15.1%Leucocytes 13.24×109/L↑ 5.79×109/LNeutrophils 11.25×109/L↑ 3.75×109/LEosinophils 0.2×109/L 0.12×109/LBasophils 0.4×109/L 0.9×109/LLymphocytes 0.97×109/L↑ 1.57×109/LMonocytes 0.96×109/L 0.30×109/LPlatelets 249×109/L 228×109/LC- reactive protein 152.5 mg/L↑ 0.9 mg/LProthrombin time 25.7 seg↑ 14.1 segActivated partial prothrombin time 31.5 seg↑ 35.1 segUrea 90 mg/dL↑ 52 mg/dL↑Creatinine 1.62 mg/dL↑ 1.18 mg/dL↑Estimated glomerular filtration rate 30 mL/min/1.73↓ 44 mL/min/1.73↓Lactic dehydrogenase 339 U/L↑ 227 U/L↑Sodium 134 mEq/L 142 mEq/LPotassium 4.6 mEq/L 4.7 mEq/LChloride 107 mEq/L 106 mEq/LCalcium 8.1 mg/dL 9.5 mg/dLMagnesium 1.89 mg/dL 1.82 mg/dLFigure 1 Optical coherence tomography imaging showing hyperreflective and thickened inner retinal layers that correspond to retinal oedema left eye. Oculus dextrus, OD; oculus sinister, OS.Protected by copyright. on October 12, 2020 at Hospitais da Universidade Coimbra.http://casereports.bmj.com/BMJ Case Rep: first published as 10.1136/bcr-2020-235763 on 2 September 2020. Downloaded from 3Serras- Pereira R, et al. BMJ Case Rep 2020;13:e235763. doi:10.1136/bcr-2020-235763Reminder of important clinical lessonAny process that leads to the obstruction of blood flow to the CRA can be a cause of CRAO.3 Embolism from an athero-sclerotic plaque is the leading cause of CRAO, most frequently in the internal carotid artery followed by the aortic arch and heart.1 3 Less commonly, CRAO can be caused by a thrombotic event in the setting of vasculitis, especially giant cell arteritis, or hypercoagulable state such as hyperhomocysteinaemia, factor V Leiden, protein C and S deficiencies, sickle cell disease, parane-oplasic syndromes, among others.1–3 7 Certain ocular conditions can also lead to CRAO by decreasing ocular perfusion across the optic nerve head: acutely raised IOP, optic nerve head drusen and preretinal arterial loops.3 These less frequent causes should always be considered in patients without classic cardiovascular risk factors. As far as we know, CRAO due to an embolus from a cardiac vegetation is a very rare complication of IE with no estimated prevalence in the current literature.IE should be suspected in patients with risk factors for the disease and an acute or subacute illness.8 Damaged endothelium in the setting of valvular stenosis can lead to the formation of a platelet and fibrin thrombus with risk of bacterial colonisation, which in turn may lead to further endothelial injury and culmi-nate in the formation of an infected vegetation.9 Eighty to ninety per cent of cases involve gram- positive cocci, with S. aureus identified as the most frequent causative pathogen in devel-oped nations and S. viridans group in developing nations.9 IE has a wide clinical presentation, often non- specific, and embolic events may prompt patients to seek medical attention in the first place.The documentation by an ophthalmologist of an embolic event may be central to diagnostic guidance in the emergency room, as this case highlights. An IE should always be suspected in a patient with a heart murmur and documented cardiac pathology, particularly with positive inflammation markers and bacteremia. In the setting of degenerative valvular stenosis, non- specific presentation of long standing fatigue and an embolic event (including CRAO), a diagnostic hypothesis of IE should not be dismissed even if concurrent cardiovascular risk factors are present. In the present case report, a medical history and general examination pointed towards a septic embolus due to IE even in the presence of multiple cardiovascular risk factors.IE is a potentially life- threatening condition with high mortality rates and treatment should be started as soon as the diagnosis is made. Likewise, there is a high degree of morbidity and mortality associated with CRAO, not only due to severe vision compromise and decreased quality of life, but also due to immediate and long- term ocular and systemic events.2 There is an increased risk of stroke, acute myocardial infarction and death in patients with CRAO admitted to hospitals as CRAO represents end- organ ischaemia and potentially underlying atherosclerotic disease that puts patients at risk of future ischaemic events.1 2 Lavin et al reported an incidence of 36.7% of critical carotid artery disease and 20% of major echocardiographic abnormal-ities, such as myocardial infarction, heart failure and critical valvular disease, in CRAO patients.10A multidisciplinary approach is warranted with close collab-oration between ophthalmologists and other specialists in order to provide proper management and the best possible treatment to these patients.Learning points ► Central retinal artery occlusion (CRAO) can be the first manifestation of a potentially fatal systemic condition, such as IE, with overlapping risk factors for major cardiovascular and cerebrovascular events. ► CRAO is a very rare complication in the setting of an infective endocarditis (IE). ► IE should be suspected in patients with risk factors for the disease and an acute or subacute illness. ► The documentation by an ophthalmologist of an embolic event may be central to diagnostic guidance in the emergency room. ► Close collaboration between specialists from different areas is fundamental in order to provide optimal management to our patients.Contributors RS- P: data acquisition, data analysis and paper conception. DH- F: data analysis and paper conception. LA and LV: data aquisition, data analysis and paper revision.Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not- for- profit sectors.Competing interests None declared.Patient consent for publication Not required.Provenance and peer review Not commissioned; externally peer reviewed.ORCID iDDiogo Hipolito- Fernandes http:// orcid. org/ 0000- 0002- 5972- 4068REFERENCES 1 Varma DD, Cugati S, Lee AW, et al. A review of central retinal artery occlusion: clinical presentation and management. Eye 2013;27:688–97. 2 Dattilo M, Newman NJ, Biousse V. Acute retinal arterial ischemia. Ann Eye Sci 2018;3:28–3. 3 Dattilo M, Biousse V, Newman NJ. Update on the management of central retinal artery occlusion. Neurol Clin 2017;35:83–100. 4 Mohananey D, Mohadjer A, Pettersson G, et al. Association of vegetation size with embolic risk in patients with infective endocarditis: a systematic review and meta- analysis. JAMA Intern Med 2018;178:502–10. 5 Ziakas NG, Kotsidis S, Ziakas A. Central retinal artery occlusion due to infective endocarditis. Int Ophthalmol 2014;34:315–9. 6 Isenring J, Köhler J, Nakata M, et al. Streptococcus gallolyticus subsp. gallolyticus endocarditis isolate interferes with coagulation and activates the contact system. Virulence 2018;9:248–61. 7 Wathek C, Kharrat O, Maalej A, et al. Ophthalmic artery occlusion as a complication of infectious endocarditis. J Fr Ophtalmol 2014;37:e161–3. 8 Wang A, Gaca JG, Chu VH. Management considerations in infective endocarditis: a review. JAMA 2018;320:72–83. 9 Cahill TJ, Prendergast BD. Infective endocarditis. The Lancet 2016;387:882–93. 10 Lavin P, Patrylo M, Hollar M, et al. Stroke risk and risk factors in patients with central retinal artery occlusion. Am J Ophthalmol 2018;196:96–100.Protected by copyright. on October 12, 2020 at Hospitais da Universidade Coimbra.http://casereports.bmj.com/BMJ Case Rep: first published as 10.1136/bcr-2020-235763 on 2 September 2020. Downloaded from 4 Serras- Pereira R, et al. BMJ Case Rep 2020;13:e235763. doi:10.1136/bcr-2020-235763Reminder of important clinical lessonCopyright 2020 BMJ Publishing Group. All rights reserved. For permission to reuse any of this content visithttps://www.bmj.com/company/products-services/rights-and-licensing/permissions/BMJ Case Report Fellows may re-use this article for personal use and teaching without any further permission.Become a Fellow of BMJ Case Reports today and you can: ► Submit as many cases as you like ► Enjoy fast sympathetic peer review and rapid publication of accepted articles ► Access all the published articles ► Re-use any of the published material for personal use and teaching without further permissionCustomer ServiceIf you have any further queries about your subscription, please contact our customer services team on +44 (0) 207111 1105 or via email at support@bmj.com.Visit casereports.bmj.com for more articles like this and to become a FellowProtected by copyright. on October 12, 2020 at Hospitais da Universidade Coimbra.http://casereports.bmj.com/BMJ Case Rep: first published as 10.1136/bcr-2020-235763 on 2 September 2020. Downloaded from 

Central Retinal Artery Occlusion from Streptococcus Gallolyticus Endocarditis

Central retinal artery occlusion (CRAO) is a rare but blinding disorder. We present a case of a 81-year-old woman with multiple cardiovascular comorbidities admitted to the emergency department due to sudden, painless vision loss on left eye (oculus sinister (OS)) on awakening. The patient also reported long standing fatigue associated with effort that started 4 months before admission. She presented best corrected visual acuity of counting fingers OS. Funduscopy OS revealed macular oedema with cherry red spot pattern. Blood cultures came positive for Streptococcus gallolyticus in the context of a bacteremia and native mitral valve vegetation identified on transoesophageal echocardiography. CRAO of embolic origin was admitted in the context of an infective endocarditis. CRAO can be the first manifestation of a potentially fatal systemic condition and thus multidisciplinary approach is warranted with close collaboration between ophthalmologists and internists in order to provide proper management and the best possible treatment

Serras-Pereira, Rita

Hipolito-Fernandes, Diogo

Azevedo, Luísa

Vieira, Luísa

PubMed Central

Central retinal artery occlusion from Streptococcus gallolyticus endocarditis

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Central Retinal Artery Occlusion from Streptococcus Gallolyticus Endocarditis

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Repositório do Centro Hospitalar de Lisboa Central, EPE

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