Cargando…

Neurofibromatosis type 1 is not associated with subarachnoid haemorrhage

BACKGROUND: The prevalence of intracranial aneurysms (IAs) has been proposed to be elevated in the patients with neurofibromatosis type 1 (NF1). Our aims were to determine the prevalence of NF1 in a large Finnish population based cohort of IA patients and, on the other hand, the occurrences of subar...

Descripción completa

Detalles Bibliográficos
Autores principales: Kurtelius, Arttu, Kallionpää, Roope A., Huttunen, Jukka, Huttunen, Terhi J., Helin, Katariina, Koivisto, Timo, Frösen, Juhana, von und zu Fraunberg, Mikael, Peltonen, Sirkku, Peltonen, Juha, Jääskeläinen, Juha E., Lindgren, Antti E.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Public Library of Science 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5456355/
https://www.ncbi.nlm.nih.gov/pubmed/28575128
http://dx.doi.org/10.1371/journal.pone.0178711
_version_ 1783241236263469056
author Kurtelius, Arttu
Kallionpää, Roope A.
Huttunen, Jukka
Huttunen, Terhi J.
Helin, Katariina
Koivisto, Timo
Frösen, Juhana
von und zu Fraunberg, Mikael
Peltonen, Sirkku
Peltonen, Juha
Jääskeläinen, Juha E.
Lindgren, Antti E.
author_facet Kurtelius, Arttu
Kallionpää, Roope A.
Huttunen, Jukka
Huttunen, Terhi J.
Helin, Katariina
Koivisto, Timo
Frösen, Juhana
von und zu Fraunberg, Mikael
Peltonen, Sirkku
Peltonen, Juha
Jääskeläinen, Juha E.
Lindgren, Antti E.
author_sort Kurtelius, Arttu
collection PubMed
description BACKGROUND: The prevalence of intracranial aneurysms (IAs) has been proposed to be elevated in the patients with neurofibromatosis type 1 (NF1). Our aims were to determine the prevalence of NF1 in a large Finnish population based cohort of IA patients and, on the other hand, the occurrences of subarachnoid haemorrhage and unruptured intracranial aneurysms in a nationwide population-based cohort of NF1 patients and its matched ten-fold control cohort. METHODS: The Kuopio IA Database (www.kuopioneurosurgery.fi) includes all ruptured and unruptured IA cases admitted to the Kuopio University Hospital (KUH) from its defined Eastern Finnish catchment population since 1980. In this registry-based study, we cross-linked the Kuopio IA database with the Finnish national registry covering all hospital diagnoses. The NF1 diagnoses of the 4543 patients with either saccular of fusiform IA were identified from 1969 to 2015 and verified from patient records. Our second approach was to analyze the occurrence of aneurysmal subarachnoid haemorrhage (aSAH) and unruptured IAs in a nationwide population-based database of 1410 NF1 patients and its ten-fold matched control cohort (n = 14030) using national registry of hospital diagnoses between 1987 and 2014. RESULTS: One NF1 patient was identified among the 4543 IA patients. Three verified IA cases (one unruptured IA and two aSAH cases) were identified in the cohort of 1410 NF1 patients, with similar occurrences in the control cohort. CONCLUSIONS: We found no evidence in our population-based cohorts to support the conception that NF1 is associated with IAs. Our results indicate that the incidence of aSAH is not elevated in patients with NF1. Further studies are required to confirm that there is no association between NF1 and unruptured IAs.
format Online
Article
Text
id pubmed-5456355
institution National Center for Biotechnology Information
language English
publishDate 2017
publisher Public Library of Science
record_format MEDLINE/PubMed
spelling pubmed-54563552017-06-12 Neurofibromatosis type 1 is not associated with subarachnoid haemorrhage Kurtelius, Arttu Kallionpää, Roope A. Huttunen, Jukka Huttunen, Terhi J. Helin, Katariina Koivisto, Timo Frösen, Juhana von und zu Fraunberg, Mikael Peltonen, Sirkku Peltonen, Juha Jääskeläinen, Juha E. Lindgren, Antti E. PLoS One Research Article BACKGROUND: The prevalence of intracranial aneurysms (IAs) has been proposed to be elevated in the patients with neurofibromatosis type 1 (NF1). Our aims were to determine the prevalence of NF1 in a large Finnish population based cohort of IA patients and, on the other hand, the occurrences of subarachnoid haemorrhage and unruptured intracranial aneurysms in a nationwide population-based cohort of NF1 patients and its matched ten-fold control cohort. METHODS: The Kuopio IA Database (www.kuopioneurosurgery.fi) includes all ruptured and unruptured IA cases admitted to the Kuopio University Hospital (KUH) from its defined Eastern Finnish catchment population since 1980. In this registry-based study, we cross-linked the Kuopio IA database with the Finnish national registry covering all hospital diagnoses. The NF1 diagnoses of the 4543 patients with either saccular of fusiform IA were identified from 1969 to 2015 and verified from patient records. Our second approach was to analyze the occurrence of aneurysmal subarachnoid haemorrhage (aSAH) and unruptured IAs in a nationwide population-based database of 1410 NF1 patients and its ten-fold matched control cohort (n = 14030) using national registry of hospital diagnoses between 1987 and 2014. RESULTS: One NF1 patient was identified among the 4543 IA patients. Three verified IA cases (one unruptured IA and two aSAH cases) were identified in the cohort of 1410 NF1 patients, with similar occurrences in the control cohort. CONCLUSIONS: We found no evidence in our population-based cohorts to support the conception that NF1 is associated with IAs. Our results indicate that the incidence of aSAH is not elevated in patients with NF1. Further studies are required to confirm that there is no association between NF1 and unruptured IAs. Public Library of Science 2017-06-02 /pmc/articles/PMC5456355/ /pubmed/28575128 http://dx.doi.org/10.1371/journal.pone.0178711 Text en © 2017 Kurtelius et al http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Research Article
Kurtelius, Arttu
Kallionpää, Roope A.
Huttunen, Jukka
Huttunen, Terhi J.
Helin, Katariina
Koivisto, Timo
Frösen, Juhana
von und zu Fraunberg, Mikael
Peltonen, Sirkku
Peltonen, Juha
Jääskeläinen, Juha E.
Lindgren, Antti E.
Neurofibromatosis type 1 is not associated with subarachnoid haemorrhage
title Neurofibromatosis type 1 is not associated with subarachnoid haemorrhage
title_full Neurofibromatosis type 1 is not associated with subarachnoid haemorrhage
title_fullStr Neurofibromatosis type 1 is not associated with subarachnoid haemorrhage
title_full_unstemmed Neurofibromatosis type 1 is not associated with subarachnoid haemorrhage
title_short Neurofibromatosis type 1 is not associated with subarachnoid haemorrhage
title_sort neurofibromatosis type 1 is not associated with subarachnoid haemorrhage
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5456355/
https://www.ncbi.nlm.nih.gov/pubmed/28575128
http://dx.doi.org/10.1371/journal.pone.0178711
work_keys_str_mv AT kurteliusarttu neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT kallionpaaroopea neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT huttunenjukka neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT huttunenterhij neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT helinkatariina neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT koivistotimo neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT frosenjuhana neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT vonundzufraunbergmikael neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT peltonensirkku neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT peltonenjuha neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT jaaskelainenjuhae neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage
AT lindgrenanttie neurofibromatosistype1isnotassociatedwithsubarachnoidhaemorrhage