Widespread white matter changes in post-H1N1 patients with narcolepsy type 1 and first-degree relatives
To assess white matter involvement in H1N1-vaccinated hypocretin deficient patients with narcolepsy type 1 (NT1) compared with first-degree relatives (a potential risk group) and healthy controls. We compared four diffusion tensor imaging-based microstructural indices (fractional anisotropy [FA], me...
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creator | Juvodden, Hilde T Alnæs, Dag Lund, Martina J Agartz, Ingrid Andreassen, Ole A Dietrichs, Espen Thorsby, Per M Westlye, Lars T Knudsen, Stine |
description | To assess white matter involvement in H1N1-vaccinated hypocretin deficient patients with narcolepsy type 1 (NT1) compared with first-degree relatives (a potential risk group) and healthy controls.
We compared four diffusion tensor imaging-based microstructural indices (fractional anisotropy [FA], mean diffusivity [MD], radial diffusivity [RD], and axial diffusivity [AD]) in 57 patients with NT1 (39 females, mean age 21.8 years, 51/57 H1N1-vaccinated, 57/57 HLA-DQB1*06:02-positive, 54/54 hypocretin-deficient), 54 first-degree relatives (29 females, mean age 19.1 years, 37/54 H1N1-vaccinated, 32/54 HLA-DQB1*06:02-positive), and 55 healthy controls (38 females, mean age 22.3 years). We tested for differences between these groups, for parametric effects (controls > first-degree relatives > patients) and associations in patients (cerebrospinal fluid [CSF] hypocretin-1 and disease duration) and first-degree relatives (HLA-DQB1*06:02 and H1N1-vaccination). We employed tract-based spatial statistics and used permutation testing and threshold-free cluster enhancement for inference.
Patients with NT1 had a widespread, bilateral pattern of significantly lower FA compared with first-degree relatives and healthy controls. Additionally, patients with NT1 also exhibited significantly higher RD and lower AD in several focal white matter clusters. The parametric model showed that first-degree relatives had intermediate values. Full sample of patients with NT1 showed no significant associations with disease duration or CSF hypocretin-1.
Our study suggests widespread abnormal white matter involvement far beyond the already known focal hypothalamic pathology in NT1, possibly reflecting the combined effects of the loss of the widely projecting hypothalamic hypocretin neurons, and/or secondary effects of wake/sleep dysregulation. These findings demonstrate the importance of white matter pathology in NT1. |
doi_str_mv | 10.1093/sleep/zsy145 |
format | Article |
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We compared four diffusion tensor imaging-based microstructural indices (fractional anisotropy [FA], mean diffusivity [MD], radial diffusivity [RD], and axial diffusivity [AD]) in 57 patients with NT1 (39 females, mean age 21.8 years, 51/57 H1N1-vaccinated, 57/57 HLA-DQB1*06:02-positive, 54/54 hypocretin-deficient), 54 first-degree relatives (29 females, mean age 19.1 years, 37/54 H1N1-vaccinated, 32/54 HLA-DQB1*06:02-positive), and 55 healthy controls (38 females, mean age 22.3 years). We tested for differences between these groups, for parametric effects (controls > first-degree relatives > patients) and associations in patients (cerebrospinal fluid [CSF] hypocretin-1 and disease duration) and first-degree relatives (HLA-DQB1*06:02 and H1N1-vaccination). We employed tract-based spatial statistics and used permutation testing and threshold-free cluster enhancement for inference.
Patients with NT1 had a widespread, bilateral pattern of significantly lower FA compared with first-degree relatives and healthy controls. Additionally, patients with NT1 also exhibited significantly higher RD and lower AD in several focal white matter clusters. The parametric model showed that first-degree relatives had intermediate values. Full sample of patients with NT1 showed no significant associations with disease duration or CSF hypocretin-1.
Our study suggests widespread abnormal white matter involvement far beyond the already known focal hypothalamic pathology in NT1, possibly reflecting the combined effects of the loss of the widely projecting hypothalamic hypocretin neurons, and/or secondary effects of wake/sleep dysregulation. These findings demonstrate the importance of white matter pathology in NT1.</description><identifier>ISSN: 0161-8105</identifier><identifier>EISSN: 1550-9109</identifier><identifier>DOI: 10.1093/sleep/zsy145</identifier><identifier>PMID: 30016530</identifier><language>eng</language><publisher>United States: Oxford University Press</publisher><subject>Heredity ; Sleep disorders</subject><ispartof>Sleep (New York, N.Y.), 2018-10, Vol.41 (10)</ispartof><rights>Sleep Research Society 2018. Published by Oxford University Press on behalf of the Sleep Research Society. All rights reserved. For permissions, please e-mail journals.permissions@oup.com.</rights><rights>info:eu-repo/semantics/openAccess</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c381t-a41bbe4cc7c6dd86d6fa148198402ead7889d20fdfffd0e684fa9b70341f73293</citedby><cites>FETCH-LOGICAL-c381t-a41bbe4cc7c6dd86d6fa148198402ead7889d20fdfffd0e684fa9b70341f73293</cites><orcidid>0000-0001-6796-7177</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>230,314,780,784,885,26567,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/30016530$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Juvodden, Hilde T</creatorcontrib><creatorcontrib>Alnæs, Dag</creatorcontrib><creatorcontrib>Lund, Martina J</creatorcontrib><creatorcontrib>Agartz, Ingrid</creatorcontrib><creatorcontrib>Andreassen, Ole A</creatorcontrib><creatorcontrib>Dietrichs, Espen</creatorcontrib><creatorcontrib>Thorsby, Per M</creatorcontrib><creatorcontrib>Westlye, Lars T</creatorcontrib><creatorcontrib>Knudsen, Stine</creatorcontrib><title>Widespread white matter changes in post-H1N1 patients with narcolepsy type 1 and first-degree relatives</title><title>Sleep (New York, N.Y.)</title><addtitle>Sleep</addtitle><description>To assess white matter involvement in H1N1-vaccinated hypocretin deficient patients with narcolepsy type 1 (NT1) compared with first-degree relatives (a potential risk group) and healthy controls.
We compared four diffusion tensor imaging-based microstructural indices (fractional anisotropy [FA], mean diffusivity [MD], radial diffusivity [RD], and axial diffusivity [AD]) in 57 patients with NT1 (39 females, mean age 21.8 years, 51/57 H1N1-vaccinated, 57/57 HLA-DQB1*06:02-positive, 54/54 hypocretin-deficient), 54 first-degree relatives (29 females, mean age 19.1 years, 37/54 H1N1-vaccinated, 32/54 HLA-DQB1*06:02-positive), and 55 healthy controls (38 females, mean age 22.3 years). We tested for differences between these groups, for parametric effects (controls > first-degree relatives > patients) and associations in patients (cerebrospinal fluid [CSF] hypocretin-1 and disease duration) and first-degree relatives (HLA-DQB1*06:02 and H1N1-vaccination). We employed tract-based spatial statistics and used permutation testing and threshold-free cluster enhancement for inference.
Patients with NT1 had a widespread, bilateral pattern of significantly lower FA compared with first-degree relatives and healthy controls. Additionally, patients with NT1 also exhibited significantly higher RD and lower AD in several focal white matter clusters. The parametric model showed that first-degree relatives had intermediate values. Full sample of patients with NT1 showed no significant associations with disease duration or CSF hypocretin-1.
Our study suggests widespread abnormal white matter involvement far beyond the already known focal hypothalamic pathology in NT1, possibly reflecting the combined effects of the loss of the widely projecting hypothalamic hypocretin neurons, and/or secondary effects of wake/sleep dysregulation. These findings demonstrate the importance of white matter pathology in NT1.</description><subject>Heredity</subject><subject>Sleep disorders</subject><issn>0161-8105</issn><issn>1550-9109</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2018</creationdate><recordtype>article</recordtype><sourceid>8G5</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><sourceid>GNUQQ</sourceid><sourceid>GUQSH</sourceid><sourceid>M2O</sourceid><sourceid>3HK</sourceid><recordid>eNpdkUFvFDEMhSMEokvhxhkiceHA0HgymWSOVQUUqYILiGOUnTi7qWYzQ5ylWn49WbblwMm2_NnP8mPsJYj3IAZ5QRPicvGbDtCpR2wFSolmqJ3HbCWgh8aAUGfsGdGtqHU3yKfsTB5TJcWKbX5Ej7RkdJ7fbWNBvnOlYObj1qUNEo-JLzOV5hq-AF9ciZgK8btYtjy5PM4TLnTg5bAgB-6S5yHminvcZESecaojv5CesyfBTYQv7uM5-_7xw7er6-bm66fPV5c3zSgNlMZ1sF5jN4567L03ve-Dg87AYDrR1hu1MYNvRfAhBC-wN11ww1oL2UHQsh3kOXt92jvmSCUmm-bsLAijWqvbXh2JtydiyfPPPVKxu0gjTpNLOO_JtkKD0lpJVdE3_6G38z6ner9tZa9A1v_rSr17kJyJMga75Lhz-VBl7dEi-9cie7Ko4q_ul-7XO_T_4AdP5B9F5I1M</recordid><startdate>20181001</startdate><enddate>20181001</enddate><creator>Juvodden, Hilde T</creator><creator>Alnæs, Dag</creator><creator>Lund, Martina J</creator><creator>Agartz, Ingrid</creator><creator>Andreassen, Ole A</creator><creator>Dietrichs, Espen</creator><creator>Thorsby, Per M</creator><creator>Westlye, Lars T</creator><creator>Knudsen, Stine</creator><general>Oxford University Press</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>88G</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>8G5</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>GUQSH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>M2M</scope><scope>M2O</scope><scope>MBDVC</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PSYQQ</scope><scope>Q9U</scope><scope>7X8</scope><scope>3HK</scope><orcidid>https://orcid.org/0000-0001-6796-7177</orcidid></search><sort><creationdate>20181001</creationdate><title>Widespread white matter changes in post-H1N1 patients with narcolepsy type 1 and first-degree relatives</title><author>Juvodden, Hilde T ; Alnæs, Dag ; Lund, Martina J ; Agartz, Ingrid ; Andreassen, Ole A ; Dietrichs, Espen ; Thorsby, Per M ; Westlye, Lars T ; Knudsen, Stine</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c381t-a41bbe4cc7c6dd86d6fa148198402ead7889d20fdfffd0e684fa9b70341f73293</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2018</creationdate><topic>Heredity</topic><topic>Sleep disorders</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Juvodden, Hilde T</creatorcontrib><creatorcontrib>Alnæs, Dag</creatorcontrib><creatorcontrib>Lund, Martina J</creatorcontrib><creatorcontrib>Agartz, Ingrid</creatorcontrib><creatorcontrib>Andreassen, Ole A</creatorcontrib><creatorcontrib>Dietrichs, Espen</creatorcontrib><creatorcontrib>Thorsby, Per M</creatorcontrib><creatorcontrib>Westlye, Lars T</creatorcontrib><creatorcontrib>Knudsen, Stine</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>ProQuest - Health & Medical Complete保健、医学与药学数据库</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Psychology Database (Alumni)</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>Research Library (Alumni Edition)</collection><collection>ProQuest Central (Alumni)</collection><collection>ProQuest Central</collection><collection>ProQuest Central Essentials</collection><collection>AUTh Library subscriptions: ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>Research Library Prep</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>PML(ProQuest Medical Library)</collection><collection>Psychology Database (ProQuest)</collection><collection>ProQuest research library</collection><collection>Research Library (Corporate)</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest One Psychology</collection><collection>ProQuest Central Basic</collection><collection>MEDLINE - Academic</collection><collection>NORA - Norwegian Open Research Archives</collection><jtitle>Sleep (New York, N.Y.)</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Juvodden, Hilde T</au><au>Alnæs, Dag</au><au>Lund, Martina J</au><au>Agartz, Ingrid</au><au>Andreassen, Ole A</au><au>Dietrichs, Espen</au><au>Thorsby, Per M</au><au>Westlye, Lars T</au><au>Knudsen, Stine</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Widespread white matter changes in post-H1N1 patients with narcolepsy type 1 and first-degree relatives</atitle><jtitle>Sleep (New York, N.Y.)</jtitle><addtitle>Sleep</addtitle><date>2018-10-01</date><risdate>2018</risdate><volume>41</volume><issue>10</issue><issn>0161-8105</issn><eissn>1550-9109</eissn><abstract>To assess white matter involvement in H1N1-vaccinated hypocretin deficient patients with narcolepsy type 1 (NT1) compared with first-degree relatives (a potential risk group) and healthy controls.
We compared four diffusion tensor imaging-based microstructural indices (fractional anisotropy [FA], mean diffusivity [MD], radial diffusivity [RD], and axial diffusivity [AD]) in 57 patients with NT1 (39 females, mean age 21.8 years, 51/57 H1N1-vaccinated, 57/57 HLA-DQB1*06:02-positive, 54/54 hypocretin-deficient), 54 first-degree relatives (29 females, mean age 19.1 years, 37/54 H1N1-vaccinated, 32/54 HLA-DQB1*06:02-positive), and 55 healthy controls (38 females, mean age 22.3 years). We tested for differences between these groups, for parametric effects (controls > first-degree relatives > patients) and associations in patients (cerebrospinal fluid [CSF] hypocretin-1 and disease duration) and first-degree relatives (HLA-DQB1*06:02 and H1N1-vaccination). We employed tract-based spatial statistics and used permutation testing and threshold-free cluster enhancement for inference.
Patients with NT1 had a widespread, bilateral pattern of significantly lower FA compared with first-degree relatives and healthy controls. Additionally, patients with NT1 also exhibited significantly higher RD and lower AD in several focal white matter clusters. The parametric model showed that first-degree relatives had intermediate values. Full sample of patients with NT1 showed no significant associations with disease duration or CSF hypocretin-1.
Our study suggests widespread abnormal white matter involvement far beyond the already known focal hypothalamic pathology in NT1, possibly reflecting the combined effects of the loss of the widely projecting hypothalamic hypocretin neurons, and/or secondary effects of wake/sleep dysregulation. These findings demonstrate the importance of white matter pathology in NT1.</abstract><cop>United States</cop><pub>Oxford University Press</pub><pmid>30016530</pmid><doi>10.1093/sleep/zsy145</doi><orcidid>https://orcid.org/0000-0001-6796-7177</orcidid><oa>free_for_read</oa></addata></record> |
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subjects | Heredity Sleep disorders |
title | Widespread white matter changes in post-H1N1 patients with narcolepsy type 1 and first-degree relatives |
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