{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,23]],"date-time":"2026-06-23T02:30:03Z","timestamp":1782181803536,"version":"3.54.5"},"reference-count":57,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2014,6,4]],"date-time":"2014-06-04T00:00:00Z","timestamp":1401840000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"Sophies Minde Foundation"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Clinical Genetics"],"published-print":{"date-parts":[[2015,2]]},"abstract":"<jats:p>The purpose of this study was to explore the literature on the psychosocial aspects of Marfan syndrome (<jats:styled-content style=\"fixed-case\">MFS<\/jats:styled-content>), to critically appraise and to synthesize relevant literature. A mixed\u2010method systematic review was performed by searching the published literature databases using available medical, psychological, pedagogical and social databases and other sources. All studies that addressed psychosocial aspects of <jats:styled-content style=\"fixed-case\">MFS<\/jats:styled-content>, published in peer\u2010reviewed journals were assessed. Of 81 search results, 15 articles (four articles based on same study population) satisfied the eligibility criteria. All studies were cross\u2010sectional; no intervention or randomized controlled trial (<jats:styled-content style=\"fixed-case\">RCT<\/jats:styled-content>) studies were found. Most studies were of small sample sizes, had low response rate or participants without a verified diagnosis. Despite these limitations, all studies described, that <jats:styled-content style=\"fixed-case\">MFS<\/jats:styled-content> has a significant impact on the psychosocial aspects of people's lives: Decreased quality of life; challenges in education, work and family life, depression and anxiety. Some studies indicated that the subjective perception of discomfort did not necessarily match the medical severity of a disease. The research of the psychosocial aspects of <jats:styled-content style=\"fixed-case\">MFS<\/jats:styled-content> is limited in size and quality. More research is needed on the psychosocial aspects of <jats:styled-content style=\"fixed-case\">MFS<\/jats:styled-content> in samples with a verified diagnosis to develop evidence\u2010based knowledge and appropriate guidelines.<\/jats:p>","DOI":"10.1111\/cge.12422","type":"journal-article","created":{"date-parts":[[2014,5,9]],"date-time":"2014-05-09T16:38:41Z","timestamp":1399653521000},"page":"109-116","source":"Crossref","is-referenced-by-count":51,"title":["Systematic review of the psychosocial aspects of living with Marfan syndrome"],"prefix":"10.1111","volume":"87","author":[{"given":"G.","family":"Velvin","sequence":"first","affiliation":[{"name":"Sunnaas Rehabilitation Hospital TRS National Resource Centre for Rare Disorders  Nesoddtangen Akershus Norway"},{"name":"Norway Faculty of Social Sciences, Department of Social Work, Child Welfare and Social Policy Oslo and Akershus University College of Applied Sciences  Oslo Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"T.","family":"Bathen","sequence":"additional","affiliation":[{"name":"Sunnaas Rehabilitation Hospital TRS National Resource Centre for Rare Disorders  Nesoddtangen Akershus Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S.","family":"Rand\u2010Hendriksen","sequence":"additional","affiliation":[{"name":"Sunnaas Rehabilitation Hospital TRS National Resource Centre for Rare Disorders  Nesoddtangen Akershus Norway"},{"name":"Institute of Clinical Medicine, Faculty of Medicine University of Oslo  Oslo Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.\u00d8.","family":"Geirdal","sequence":"additional","affiliation":[{"name":"Norway Faculty of Social Sciences, Department of Social Work, Child Welfare and Social Policy Oslo and Akershus University College of Applied Sciences  Oslo Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2014,6,4]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.1998.tb03714.x"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.med.51.1.481"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19960424)62:4<417::AID-AJMG15>3.0.CO;2-R"},{"key":"e_1_2_7_5_1","volume-title":"Principles and practice of medical genetics","author":"Pyeritz R","year":"1996"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.2009.072785"},{"key":"e_1_2_7_7_1","first-page":"684","article-title":"The eye in the Marfan syndrome","volume":"79","author":"Maumenee IH","year":"1981","journal-title":"Trans Am Ophthalmol Soc"},{"key":"e_1_2_7_8_1","first-page":"3579","volume-title":"Emery and Rimoin's principles and practice of medical genetics","author":"Pyeritz E","year":"2007"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1399-0004.2001.600405.x"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2013.123"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/jor.20314"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1399-0004.2001.600406.x"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1097\/GIM.0b013e3181ea4c1c"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1399-0004.2002.620203.x"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1742-1241.2007.01407.x"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2005.00446.x"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1159\/000082265"},{"key":"e_1_2_7_18_1","unstructured":"The Campbell Collaboration Guidelines. How to make a Campbell Collaboration Review What is a Systematic Review Nordic Campbell Center.www.nc2.net fromhttp:\/\/www.sfi.dk\/graphics\/Campbell\/Dokumenter\/For_Forskere\/Guide_2_protocol_dec04.pdf. Accessed on March 22 2014."},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/jep.12017"},{"key":"e_1_2_7_20_1","unstructured":"The Campbell Collaboration c1c2Equity Checklists for Systematic Review Authors Whitehead 1991 fromhttp:\/\/www.campbellcollaboration.org\/artman2\/uploads\/1\/C2_Equity_Checklist.pdf. Accessed on March 20 2014."},{"key":"e_1_2_7_21_1","unstructured":"The Cochrane Collaboration. Cochrane Reviews. The Cochrane Collaboration. Version 5.1.0. Updated March 2011. Editors Higgins JPT Green S fromhttp:\/\/handbook.cochrane.org\/. Accessed on March 20 2014."},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijnurstu.2009.01.009"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11135\u2010011\u20109538\u20106"},{"key":"e_1_2_7_24_1","unstructured":"International Center for Allied Health EvidenceUniversity of South Australia University publications. CRICOS Provider no 0012B fromhttp:\/\/www.unisa.edu.au\/Research\/Sansom\u2010Institute\u2010for\u2010Health\u2010Research\/Research\u2010at\u2010the\u2010Sansom\/Research\u2010Concentrations\/Allied\u2010Health\u2010Evidence\/Resources\/CAT\/#Qualitative. Accessed on February 1 2014."},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.302.6785.1136"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.1177\/1524839909353023"},{"key":"e_1_2_7_27_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.315.7103.305"},{"key":"e_1_2_7_28_1","first-page":"106","volume-title":"Epidemiology in medicine","author":"Hennekens CH","year":"1997"},{"key":"e_1_2_7_29_1","doi-asserted-by":"publisher","DOI":"10.1177\/1524839910363537"},{"key":"e_1_2_7_30_1","doi-asserted-by":"crossref","first-page":"740","DOI":"10.1136\/bmj.315.7110.740","article-title":"How to read a paper: papers that go beyond numbers (qualitative research)","volume":"315","author":"Greenhalgh T","year":"1997","journal-title":"BMJ"},{"key":"e_1_2_7_31_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1440-1630.2007.00661.x"},{"key":"e_1_2_7_32_1","volume-title":"Supplementary guidance for inclusion of qualitative research in cochrane systematic reviews intervention","author":"Hannes K","year":"2011"},{"key":"e_1_2_7_33_1","doi-asserted-by":"publisher","DOI":"10.12968\/bjon.2007.16.12.23726"},{"key":"e_1_2_7_34_1","volume-title":"Handbook of qualitative research","author":"Denzin NK","year":"2005"},{"key":"e_1_2_7_35_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(01)05627-6"},{"key":"e_1_2_7_36_1","unstructured":"Systematic Reviews. CRD&s guidance for undertaking reviews in health care. Centre for Reviews and Dissemination.2009 fromhttp:\/\/www.york.ac.uk\/inst\/crd\/SysRev\/!SSL!\/WebHelp\/SysRev3.htm. Accessed on February 1 2014."},{"key":"e_1_2_7_37_1","doi-asserted-by":"publisher","DOI":"10.1177\/1558689810390217"},{"key":"e_1_2_7_38_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.36574"},{"key":"e_1_2_7_39_1","doi-asserted-by":"publisher","DOI":"10.1176\/appi.psy.49.3.243"},{"key":"e_1_2_7_40_1","doi-asserted-by":"publisher","DOI":"10.1097\/00004703-199006000-00005"},{"key":"e_1_2_7_41_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.35.5.405"},{"key":"e_1_2_7_42_1","doi-asserted-by":"publisher","DOI":"10.1177\/1049732308314853"},{"key":"e_1_2_7_43_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2008.01052.x"},{"key":"e_1_2_7_44_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1399-0004.2001.600507.x"},{"key":"e_1_2_7_45_1","unstructured":"Institute for Work & Health. Research Excellence Advancing Employee Health. \u201cCohort studies Case\u2010control studies and randomized controlled trials\u201d Source: At Work. issue 42. Fall 2005. Institute for Work & Health Toronto fromhttp:\/\/www.iwh.on.ca\/wrmb\/cohort\u2010studies\u2010case\u2010control\u2010studies\u2010and\u2010rcts. Accessed on April 20 2014."},{"key":"e_1_2_7_46_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.30525"},{"key":"e_1_2_7_47_1","doi-asserted-by":"publisher","DOI":"10.1080\/13548500600580824"},{"key":"e_1_2_7_48_1","unstructured":"LettsL WilkinsSLawM StewartD BoshJ WestmorlandM. Guidelines for Critical Review Form Qualitative Studies Version 2. Qualitative Review Form Guideline 2007.http:\/\/www.canchild.ca\/en\/canchildresources\/resources\/qualguide.pdf. Accessed on April 20 2014."},{"key":"e_1_2_7_49_1","volume-title":"Basic of qualitative research","author":"Strauss AM","year":"1998"},{"key":"e_1_2_7_50_1","volume-title":"Qualitative evaluation methods","author":"Patton MQ","year":"1980"},{"key":"e_1_2_7_51_1","volume-title":"Interpretation qualitative data","author":"Silverman D","year":"1993"},{"key":"e_1_2_7_52_1","unstructured":"RAND Corporation. 36 Item Short Form Survey from the RAND Medical Outcome Study RAND 2009 fromhttp:\/\/www.rand.org\/health\/surveys_tools\/mos\/mos_core_36item.html. Accessed on February 2 2014."},{"key":"e_1_2_7_53_1","unstructured":"Ferrand and Power Quality of life index Cardiac Version Copyright 1998. Ferrans CE & Powers MJ fromhttp:\/\/www.uic.edu\/orgs\/qli\/questionaires\/pdf\/cardiacversionIV\/Cardiac4english.pdf. Accessed on March 20 2014."},{"key":"e_1_2_7_54_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0378-5122(00)00142-0"},{"key":"e_1_2_7_55_1","doi-asserted-by":"publisher","DOI":"10.1586\/ern.09.34"},{"issue":"1","key":"e_1_2_7_56_1","first-page":"18","article-title":"Migration, culture bereavement and culture identity","volume":"4","author":"Bhugra D","year":"2005","journal-title":"World Psychiat"},{"key":"e_1_2_7_57_1","doi-asserted-by":"publisher","DOI":"10.1159\/000069734"},{"key":"e_1_2_7_58_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2710.2011.01316.x"}],"container-title":["Clinical Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fcge.12422","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cge.12422","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,3]],"date-time":"2023-10-03T17:18:17Z","timestamp":1696353497000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/cge.12422"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2014,6,4]]},"references-count":57,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2015,2]]}},"alternative-id":["10.1111\/cge.12422"],"URL":"https:\/\/doi.org\/10.1111\/cge.12422","archive":["Portico"],"relation":{},"ISSN":["0009-9163","1399-0004"],"issn-type":[{"value":"0009-9163","type":"print"},{"value":"1399-0004","type":"electronic"}],"subject":[],"published":{"date-parts":[[2014,6,4]]}}}