Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis
Autor(a) principal: | |
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Data de Publicação: | 2019 |
Outros Autores: | , , |
Tipo de documento: | Artigo |
Idioma: | eng |
Título da fonte: | Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) |
Texto Completo: | https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541 |
Resumo: | Susac syndrome is a rare, probably immune-mediated endotheliopathy presenting with encephalopathy, sensorineural hearing loss and retinal arterial occlusions. A 33-year-old female with Susac syndrome was worsening despite high-dose steroids so a brain biopsy was performed which suggested a possible fungal infection. Treatment with amphotericin B resulted in prompt reversal of symptoms and radiological findings, and no further symptoms occurred during 8 years of follow-up. A diagnosis of fungal infection was not confirmed. The etiology of Susac syndrome is unknown and this anecdotal observation suggests that an infectious agent susceptible to amphotericin might have caused or triggered Susac syndrome in this patient. |
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Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the PathogenesisUm Caso de Provável Síndrome de Susac com Resposta Anedótica à Anfotericina: Implicações na PatogéneseAmphotericin BMeningoencephalitisSusac SyndromeAnfotericina BMeningoencefaliteSíndrome de SusacSusac syndrome is a rare, probably immune-mediated endotheliopathy presenting with encephalopathy, sensorineural hearing loss and retinal arterial occlusions. A 33-year-old female with Susac syndrome was worsening despite high-dose steroids so a brain biopsy was performed which suggested a possible fungal infection. Treatment with amphotericin B resulted in prompt reversal of symptoms and radiological findings, and no further symptoms occurred during 8 years of follow-up. A diagnosis of fungal infection was not confirmed. The etiology of Susac syndrome is unknown and this anecdotal observation suggests that an infectious agent susceptible to amphotericin might have caused or triggered Susac syndrome in this patient.A síndrome de Susac é uma endoteliopatia rara, provavelmente imunomediada que se apresenta com a tríade sindromática de encefalopatia, surdez neurosensorial e oclusões de ramos arteriais da retina. Apresenta-se o caso de uma mulher de 33 anos com o diagnóstico de síndrome de Susac que agravou sob terapêutica imunossupressora, pelo que se decidiu pela realização de uma biópsia cerebral, que levantou a suspeita de uma possível infeção fúngica. O tratamento com anfotericina B resultou numa recuperação clínica e imagiológica rápida e não se verificou recorrência de sintomas num período de seguimento de oito anos. O diagnóstico de meningoencefalite fúngica não foi confirmado. A etiologia da síndrome de Susac é desconhecida. A observação fortuita da resposta terapêutica deste caso sugere que um agente infecioso suscetível à anfotericina B possa ter causado ou desencadeado esta síndrome, nesta doente.Ordem dos Médicos2019-11-04info:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/articleapplication/pdfapplication/pdfimage/tiffimage/tiffimage/tiffapplication/pdfinode/x-emptyimage/jpeghttps://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541oai:ojs.www.actamedicaportuguesa.com:article/10541Acta Médica Portuguesa; Vol. 32 No. 11 (2019): November; 727-730Acta Médica Portuguesa; Vol. 32 N.º 11 (2019): Novembro; 727-7301646-07580870-399Xreponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãoinstacron:RCAAPenghttps://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/5795https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10186https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10187https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10188https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10189https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10428https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10429https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10809Direitos de Autor (c) 2019 Acta Médica Portuguesainfo:eu-repo/semantics/openAccessGil-Gouveia, RaquelMarto, NatáliaVilela, PedroCatarino, Ana2022-12-20T11:06:00Zoai:ojs.www.actamedicaportuguesa.com:article/10541Portal AgregadorONGhttps://www.rcaap.pt/oai/openaireopendoar:71602024-03-19T16:19:54.131143Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãofalse |
dc.title.none.fl_str_mv |
Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis Um Caso de Provável Síndrome de Susac com Resposta Anedótica à Anfotericina: Implicações na Patogénese |
title |
Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis |
spellingShingle |
Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis Gil-Gouveia, Raquel Amphotericin B Meningoencephalitis Susac Syndrome Anfotericina B Meningoencefalite Síndrome de Susac |
title_short |
Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis |
title_full |
Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis |
title_fullStr |
Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis |
title_full_unstemmed |
Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis |
title_sort |
Anecdotal Response to Amphotericin in a Patient with Probable Susac Syndrome: Implications in the Pathogenesis |
author |
Gil-Gouveia, Raquel |
author_facet |
Gil-Gouveia, Raquel Marto, Natália Vilela, Pedro Catarino, Ana |
author_role |
author |
author2 |
Marto, Natália Vilela, Pedro Catarino, Ana |
author2_role |
author author author |
dc.contributor.author.fl_str_mv |
Gil-Gouveia, Raquel Marto, Natália Vilela, Pedro Catarino, Ana |
dc.subject.por.fl_str_mv |
Amphotericin B Meningoencephalitis Susac Syndrome Anfotericina B Meningoencefalite Síndrome de Susac |
topic |
Amphotericin B Meningoencephalitis Susac Syndrome Anfotericina B Meningoencefalite Síndrome de Susac |
description |
Susac syndrome is a rare, probably immune-mediated endotheliopathy presenting with encephalopathy, sensorineural hearing loss and retinal arterial occlusions. A 33-year-old female with Susac syndrome was worsening despite high-dose steroids so a brain biopsy was performed which suggested a possible fungal infection. Treatment with amphotericin B resulted in prompt reversal of symptoms and radiological findings, and no further symptoms occurred during 8 years of follow-up. A diagnosis of fungal infection was not confirmed. The etiology of Susac syndrome is unknown and this anecdotal observation suggests that an infectious agent susceptible to amphotericin might have caused or triggered Susac syndrome in this patient. |
publishDate |
2019 |
dc.date.none.fl_str_mv |
2019-11-04 |
dc.type.status.fl_str_mv |
info:eu-repo/semantics/publishedVersion |
dc.type.driver.fl_str_mv |
info:eu-repo/semantics/article |
format |
article |
status_str |
publishedVersion |
dc.identifier.uri.fl_str_mv |
https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541 oai:ojs.www.actamedicaportuguesa.com:article/10541 |
url |
https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541 |
identifier_str_mv |
oai:ojs.www.actamedicaportuguesa.com:article/10541 |
dc.language.iso.fl_str_mv |
eng |
language |
eng |
dc.relation.none.fl_str_mv |
https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541 https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/5795 https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10186 https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10187 https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10188 https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10189 https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10428 https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10429 https://www.actamedicaportuguesa.com/revista/index.php/amp/article/view/10541/10809 |
dc.rights.driver.fl_str_mv |
Direitos de Autor (c) 2019 Acta Médica Portuguesa info:eu-repo/semantics/openAccess |
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Direitos de Autor (c) 2019 Acta Médica Portuguesa |
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openAccess |
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application/pdf application/pdf image/tiff image/tiff image/tiff application/pdf inode/x-empty image/jpeg |
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Ordem dos Médicos |
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Ordem dos Médicos |
dc.source.none.fl_str_mv |
Acta Médica Portuguesa; Vol. 32 No. 11 (2019): November; 727-730 Acta Médica Portuguesa; Vol. 32 N.º 11 (2019): Novembro; 727-730 1646-0758 0870-399X reponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação instacron:RCAAP |
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Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação |
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RCAAP |
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Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) |
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Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação |
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