Hypersomnia in Whipple disease: case report.
Autor(a) principal: | |
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Data de Publicação: | 2006 |
Outros Autores: | , , , , , |
Tipo de documento: | Artigo |
Idioma: | eng |
Título da fonte: | Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) |
Texto Completo: | http://hdl.handle.net/10400.16/839 |
Resumo: | Arq Neuropsiquiatr. 2006 Sep;64(3B):865-8. Hypersomnia in Whipple disease: case report. Maia LF, Marta M, Lopes V, Rocha N, Lopes C, Martins-da-Silva A, Monteiro L. SourceDepartment of Neurological Disordes and Senses, Hospital Geral de Santo António, Largo Prof. Abel Salazar, 4099-001 Porto, Portugal. Abstract Whipple disease (WD) is a rare systemic infection caused by Tropheryma whippelii. Neurological involvement has been recognised in 40% of patients, either as initial manifestations or during the course of the disease. We report on a 45 years-old man with WD with initial, persistent and irresistible episodes of daytime somnolence. The patient was HLA-DQB1*0602 positive (genetic marker for narcolepsy). WD diagnosis was suspected on clinical and MRI basis and confirmed by histological and immunohistochemical study of duodenal biopsy. Forty months later all clinical features improved, narcoleptic-like episodes disappeared and cerebral MRI and CSF normalised. Longitudinal neurophysiological studies revealed persistent sleep pattern abnormalities with sleep fragmentation, paucity of slow wave and of REM sleep. The disruption of the hypocretin circuitry in the hypothalamic - diencephalic region triggered by the infection was the probable cause of the hypersomnia and narcopleptic symptoms. WD should be added to the list of causes of secondary hypersomnia. |
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Hypersomnia in Whipple disease: case report.Arq Neuropsiquiatr. 2006 Sep;64(3B):865-8. Hypersomnia in Whipple disease: case report. Maia LF, Marta M, Lopes V, Rocha N, Lopes C, Martins-da-Silva A, Monteiro L. SourceDepartment of Neurological Disordes and Senses, Hospital Geral de Santo António, Largo Prof. Abel Salazar, 4099-001 Porto, Portugal. Abstract Whipple disease (WD) is a rare systemic infection caused by Tropheryma whippelii. Neurological involvement has been recognised in 40% of patients, either as initial manifestations or during the course of the disease. We report on a 45 years-old man with WD with initial, persistent and irresistible episodes of daytime somnolence. The patient was HLA-DQB1*0602 positive (genetic marker for narcolepsy). WD diagnosis was suspected on clinical and MRI basis and confirmed by histological and immunohistochemical study of duodenal biopsy. Forty months later all clinical features improved, narcoleptic-like episodes disappeared and cerebral MRI and CSF normalised. Longitudinal neurophysiological studies revealed persistent sleep pattern abnormalities with sleep fragmentation, paucity of slow wave and of REM sleep. The disruption of the hypocretin circuitry in the hypothalamic - diencephalic region triggered by the infection was the probable cause of the hypersomnia and narcopleptic symptoms. WD should be added to the list of causes of secondary hypersomnia.Academia Brasileira de Neurologia - ABNEURORepositório Científico do Centro Hospitalar Universitário de Santo AntónioMAIA, L.MARTA, M.LOPES, V.ROCHA, N.LOPES, C.MARTINS‐DA‐SILVA, A.MONTEIRO, L.2011-09-28T11:07:39Z2006-092006-09-01T00:00:00Zinfo:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/articleapplication/pdfhttp://hdl.handle.net/10400.16/839engArq Neuropsiquiatr. 2006 Sep;64(3B):865-8.0004-282Xinfo:eu-repo/semantics/openAccessreponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãoinstacron:RCAAP2023-10-20T10:54:03Zoai:repositorio.chporto.pt:10400.16/839Portal AgregadorONGhttps://www.rcaap.pt/oai/openaireopendoar:71602024-03-19T20:37:21.952145Repositório 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 |
Hypersomnia in Whipple disease: case report. |
title |
Hypersomnia in Whipple disease: case report. |
spellingShingle |
Hypersomnia in Whipple disease: case report. MAIA, L. |
title_short |
Hypersomnia in Whipple disease: case report. |
title_full |
Hypersomnia in Whipple disease: case report. |
title_fullStr |
Hypersomnia in Whipple disease: case report. |
title_full_unstemmed |
Hypersomnia in Whipple disease: case report. |
title_sort |
Hypersomnia in Whipple disease: case report. |
author |
MAIA, L. |
author_facet |
MAIA, L. MARTA, M. LOPES, V. ROCHA, N. LOPES, C. MARTINS‐DA‐SILVA, A. MONTEIRO, L. |
author_role |
author |
author2 |
MARTA, M. LOPES, V. ROCHA, N. LOPES, C. MARTINS‐DA‐SILVA, A. MONTEIRO, L. |
author2_role |
author author author author author author |
dc.contributor.none.fl_str_mv |
Repositório Científico do Centro Hospitalar Universitário de Santo António |
dc.contributor.author.fl_str_mv |
MAIA, L. MARTA, M. LOPES, V. ROCHA, N. LOPES, C. MARTINS‐DA‐SILVA, A. MONTEIRO, L. |
description |
Arq Neuropsiquiatr. 2006 Sep;64(3B):865-8. Hypersomnia in Whipple disease: case report. Maia LF, Marta M, Lopes V, Rocha N, Lopes C, Martins-da-Silva A, Monteiro L. SourceDepartment of Neurological Disordes and Senses, Hospital Geral de Santo António, Largo Prof. Abel Salazar, 4099-001 Porto, Portugal. Abstract Whipple disease (WD) is a rare systemic infection caused by Tropheryma whippelii. Neurological involvement has been recognised in 40% of patients, either as initial manifestations or during the course of the disease. We report on a 45 years-old man with WD with initial, persistent and irresistible episodes of daytime somnolence. The patient was HLA-DQB1*0602 positive (genetic marker for narcolepsy). WD diagnosis was suspected on clinical and MRI basis and confirmed by histological and immunohistochemical study of duodenal biopsy. Forty months later all clinical features improved, narcoleptic-like episodes disappeared and cerebral MRI and CSF normalised. Longitudinal neurophysiological studies revealed persistent sleep pattern abnormalities with sleep fragmentation, paucity of slow wave and of REM sleep. The disruption of the hypocretin circuitry in the hypothalamic - diencephalic region triggered by the infection was the probable cause of the hypersomnia and narcopleptic symptoms. WD should be added to the list of causes of secondary hypersomnia. |
publishDate |
2006 |
dc.date.none.fl_str_mv |
2006-09 2006-09-01T00:00:00Z 2011-09-28T11:07:39Z |
dc.type.status.fl_str_mv |
info:eu-repo/semantics/publishedVersion |
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info:eu-repo/semantics/article |
format |
article |
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publishedVersion |
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http://hdl.handle.net/10400.16/839 |
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http://hdl.handle.net/10400.16/839 |
dc.language.iso.fl_str_mv |
eng |
language |
eng |
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Arq Neuropsiquiatr. 2006 Sep;64(3B):865-8. 0004-282X |
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info:eu-repo/semantics/openAccess |
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openAccess |
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application/pdf |
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Academia Brasileira de Neurologia - ABNEURO |
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Academia Brasileira de Neurologia - ABNEURO |
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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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