Primary Whipple disease of the brain: case report with long-term clinical and MRI follow-up

Jan Peregrin,1 Hana Malikova2,3 1Department of Neurology, 2Department of Radiology, Na Homolce Hospital, 3Second Faculty of Medicine, Institute of Anatomy, Charles University, Prague, Czech Republic Abstract: Whipple disease (WD) is a rare systemic disorder caused by the bacteria Tropherym...

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Autores principales:	Peregrin J, Malikova H
Formato:	article
Lenguaje:	EN
Publicado:	Dove Medical Press 2015
Materias:	Neurosciences. Biological psychiatry. Neuropsychiatry RC321-571 Neurology. Diseases of the nervous system RC346-429
Acceso en línea:	https://doaj.org/article/6b7bc547993c4e6a9b5e8146ec0b1298
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spelling	oai:doaj.org-article:6b7bc547993c4e6a9b5e8146ec0b12982021-12-02T03:07:36ZPrimary Whipple disease of the brain: case report with long-term clinical and MRI follow-up1178-2021https://doaj.org/article/6b7bc547993c4e6a9b5e8146ec0b12982015-09-01T00:00:00Zhttps://www.dovepress.com/primary-whipple-disease-of-the-brain-case-report-with-long-term-clinic-peer-reviewed-article-NDThttps://doaj.org/toc/1178-2021Jan Peregrin,1 Hana Malikova2,3 1Department of Neurology, 2Department of Radiology, Na Homolce Hospital, 3Second Faculty of Medicine, Institute of Anatomy, Charles University, Prague, Czech Republic Abstract: Whipple disease (WD) is a rare systemic disorder caused by the bacteria Tropheryma whipplei. In its classic form, it manifests with gastrointestinal problems including diarrhea, abdominal pain, and weight loss. However, various other systems can be affected, including the central nervous system (CNS). Even more rarely, the CNS is primarily affected without gastrointestinal symptoms and with a negative small bowel biopsy. The incidence of primary CNS WD is unknown. We report the case of a young female with the primary CNS form of WD. In this report, we highlight the main clinical features and diagnostic procedures that lead to the diagnosis and comment on the treatment and clinical response. We stress the importance of neuroimaging and brain biopsy. A unique feature of this case is that the patient has been followed up for 12 years. At the time of diagnosis, no neurological manifestations were detected, although a tumor-like lesion in the right temporal lobe and hypothalamic infiltration were present on magnetic resonance imaging (MRI). The first neurological manifestations developed 2 years later despite recommended antibiotic treatment, with cognitive impairment developing more than 10 years later. According to the MRI findings and clinical course, the disease was active for several years when multiple lesions on MRI appeared despite antibiotic therapy. In the discussion, we compare the present case with similar cases previously reported and we elaborate on the similarities and discrepancies in clinical features, diagnostic procedures, results, and treatment options. Keywords: central nervous system, brain biopsy, antibiotic treatmentPeregrin JMalikova HDove Medical PressarticleNeurosciences. Biological psychiatry. NeuropsychiatryRC321-571Neurology. Diseases of the nervous systemRC346-429ENNeuropsychiatric Disease and Treatment, Vol 2015, Iss default, Pp 2461-2469 (2015)
institution	DOAJ
collection	DOAJ
language	EN
topic	Neurosciences. Biological psychiatry. Neuropsychiatry RC321-571 Neurology. Diseases of the nervous system RC346-429
spellingShingle	Neurosciences. Biological psychiatry. Neuropsychiatry RC321-571 Neurology. Diseases of the nervous system RC346-429 Peregrin J Malikova H Primary Whipple disease of the brain: case report with long-term clinical and MRI follow-up
description	Jan Peregrin,1 Hana Malikova2,3 1Department of Neurology, 2Department of Radiology, Na Homolce Hospital, 3Second Faculty of Medicine, Institute of Anatomy, Charles University, Prague, Czech Republic Abstract: Whipple disease (WD) is a rare systemic disorder caused by the bacteria Tropheryma whipplei. In its classic form, it manifests with gastrointestinal problems including diarrhea, abdominal pain, and weight loss. However, various other systems can be affected, including the central nervous system (CNS). Even more rarely, the CNS is primarily affected without gastrointestinal symptoms and with a negative small bowel biopsy. The incidence of primary CNS WD is unknown. We report the case of a young female with the primary CNS form of WD. In this report, we highlight the main clinical features and diagnostic procedures that lead to the diagnosis and comment on the treatment and clinical response. We stress the importance of neuroimaging and brain biopsy. A unique feature of this case is that the patient has been followed up for 12 years. At the time of diagnosis, no neurological manifestations were detected, although a tumor-like lesion in the right temporal lobe and hypothalamic infiltration were present on magnetic resonance imaging (MRI). The first neurological manifestations developed 2 years later despite recommended antibiotic treatment, with cognitive impairment developing more than 10 years later. According to the MRI findings and clinical course, the disease was active for several years when multiple lesions on MRI appeared despite antibiotic therapy. In the discussion, we compare the present case with similar cases previously reported and we elaborate on the similarities and discrepancies in clinical features, diagnostic procedures, results, and treatment options. Keywords: central nervous system, brain biopsy, antibiotic treatment
format	article
author	Peregrin J Malikova H
author_facet	Peregrin J Malikova H
author_sort	Peregrin J
title	Primary Whipple disease of the brain: case report with long-term clinical and MRI follow-up
title_short	Primary Whipple disease of the brain: case report with long-term clinical and MRI follow-up
title_full	Primary Whipple disease of the brain: case report with long-term clinical and MRI follow-up
title_fullStr	Primary Whipple disease of the brain: case report with long-term clinical and MRI follow-up
title_full_unstemmed	Primary Whipple disease of the brain: case report with long-term clinical and MRI follow-up
title_sort	primary whipple disease of the brain: case report with long-term clinical and mri follow-up
publisher	Dove Medical Press
publishDate	2015
url	https://doaj.org/article/6b7bc547993c4e6a9b5e8146ec0b1298
work_keys_str_mv	AT peregrinj primarywhipplediseaseofthebraincasereportwithlongtermclinicalandmrifollowup AT malikovah primarywhipplediseaseofthebraincasereportwithlongtermclinicalandmrifollowup
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Primary Whipple disease of the brain: case report with long-term clinical and MRI follow-up

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