Neurosarcoidosis-related intracranial haemorrhage: three new cases and a systematic review of the literature.

Hdl Handle:
http://hdl.handle.net/10147/254547
Title:
Neurosarcoidosis-related intracranial haemorrhage: three new cases and a systematic review of the literature.
Authors:
O'Dwyer, J P; Al-Moyeed, B A; Farrell, M A; Pidgeon, C N; Collins, D R; Fahy, A; Gibney, J; Swan, N; Dempsey, O J; Kidd, D P; Reid, J M; Smyth, S; McCabe, D J H
Affiliation:
Department of Neurology, The Adelaide and Meath Hospital, Dublin, incorporating the National Children's Hospital, Trinity College, Dublin, Ireland.
Citation:
Neurosarcoidosis-related intracranial haemorrhage: three new cases and a systematic review of the literature. 2012:notEur J Neurol
Journal:
European journal of neurology : the official journal of the European Federation of Neurological Societies
Issue Date:
9-Jun-2012
URI:
http://hdl.handle.net/10147/254547
DOI:
10.1111/j.1468-1331.2012.03783.x
PubMed ID:
22681045
Abstract:
BACKGROUND AND PURPOSE: Intracranial haemorrhage in neurosarcoidosis (NS-ICH) is rare, poorly understood and the diagnosis of NS may not be immediately apparent. METHODS: The clinical features of three new NS-ICH cases are described including new neuropathological findings and collated with cases from a systematic literature review. RESULTS: Cases: (i) A 41-year-old man with headaches, hypoandrogenism and encephalopathy developed a cerebellar haemorrhage. He had neuropathological confirmation of NS with biopsy-proven angiocentric granulomata and venous disruption. He responded to immunosuppressive therapy. (ii) A 41-year-old man with no history of hypertension was found unconscious. A subsequently fatal pontine haemorrhage was diagnosed. Liver biopsy revealed sarcoid granulomas. (iii) A 36-year-old man with raised intracranial pressure headaches presented with a seizure and a frontal haemorrhage. Hilar lymph node biopsy confirmed sarcoidosis, and he was treated successfully. Systematic Review: Twelve other published cases were identified and collated with our cases. Average age was 36 years and M:F = 2.3:1; 46% presented with neurological symptoms and 31% had CNS-isolated disease. Immediate symptoms of ICH were acute/worsening headache or seizures (60%). ICH was supratentorial (62%), infratentorial (31%) or subarachnoid (7%). 40% had definite NS, 53% probable NS and 7% possible NS (Zajicek criteria). Antigranulomatous/immunosuppressive therapy regimens varied and 31% died. CONCLUSIONS: This series expands our knowledge of the pathology of NS-ICH, which may be of arterial or venous origin. One-third have isolated NS. Clinicians should consider NS in young-onset ICH because early aggressive antigranulomatous therapy may improve outcome.
Item Type:
Article
Language:
en
ISSN:
1468-1331

Full metadata record

DC FieldValue Language
dc.contributor.authorO'Dwyer, J Pen_GB
dc.contributor.authorAl-Moyeed, B Aen_GB
dc.contributor.authorFarrell, M Aen_GB
dc.contributor.authorPidgeon, C Nen_GB
dc.contributor.authorCollins, D Ren_GB
dc.contributor.authorFahy, Aen_GB
dc.contributor.authorGibney, Jen_GB
dc.contributor.authorSwan, Nen_GB
dc.contributor.authorDempsey, O Jen_GB
dc.contributor.authorKidd, D Pen_GB
dc.contributor.authorReid, J Men_GB
dc.contributor.authorSmyth, Sen_GB
dc.contributor.authorMcCabe, D J Hen_GB
dc.date.accessioned2012-12-05T12:36:26Z-
dc.date.available2012-12-05T12:36:26Z-
dc.date.issued2012-06-09-
dc.identifier.citationNeurosarcoidosis-related intracranial haemorrhage: three new cases and a systematic review of the literature. 2012:notEur J Neurolen_GB
dc.identifier.issn1468-1331-
dc.identifier.pmid22681045-
dc.identifier.doi10.1111/j.1468-1331.2012.03783.x-
dc.identifier.urihttp://hdl.handle.net/10147/254547-
dc.description.abstractBACKGROUND AND PURPOSE: Intracranial haemorrhage in neurosarcoidosis (NS-ICH) is rare, poorly understood and the diagnosis of NS may not be immediately apparent. METHODS: The clinical features of three new NS-ICH cases are described including new neuropathological findings and collated with cases from a systematic literature review. RESULTS: Cases: (i) A 41-year-old man with headaches, hypoandrogenism and encephalopathy developed a cerebellar haemorrhage. He had neuropathological confirmation of NS with biopsy-proven angiocentric granulomata and venous disruption. He responded to immunosuppressive therapy. (ii) A 41-year-old man with no history of hypertension was found unconscious. A subsequently fatal pontine haemorrhage was diagnosed. Liver biopsy revealed sarcoid granulomas. (iii) A 36-year-old man with raised intracranial pressure headaches presented with a seizure and a frontal haemorrhage. Hilar lymph node biopsy confirmed sarcoidosis, and he was treated successfully. Systematic Review: Twelve other published cases were identified and collated with our cases. Average age was 36 years and M:F = 2.3:1; 46% presented with neurological symptoms and 31% had CNS-isolated disease. Immediate symptoms of ICH were acute/worsening headache or seizures (60%). ICH was supratentorial (62%), infratentorial (31%) or subarachnoid (7%). 40% had definite NS, 53% probable NS and 7% possible NS (Zajicek criteria). Antigranulomatous/immunosuppressive therapy regimens varied and 31% died. CONCLUSIONS: This series expands our knowledge of the pathology of NS-ICH, which may be of arterial or venous origin. One-third have isolated NS. Clinicians should consider NS in young-onset ICH because early aggressive antigranulomatous therapy may improve outcome.en_GB
dc.languageENG-
dc.language.isoenen
dc.rightsArchived with thanks to European journal of neurology : the official journal of the European Federation of Neurological Societiesen_GB
dc.titleNeurosarcoidosis-related intracranial haemorrhage: three new cases and a systematic review of the literature.en_GB
dc.typeArticleen
dc.contributor.departmentDepartment of Neurology, The Adelaide and Meath Hospital, Dublin, incorporating the National Children's Hospital, Trinity College, Dublin, Ireland.en_GB
dc.identifier.journalEuropean journal of neurology : the official journal of the European Federation of Neurological Societiesen_GB
dc.description.provinceLeinsteren

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