{"id":"https://openalex.org/W2921828045","doi":null,"title":"Identifying barriers to clinical trial participation with families affected by fragile X syndrome","display_name":"Identifying barriers to clinical trial participation with families affected by fragile X syndrome","publication_year":2017,"publication_date":"2017-12-01","ids":{"openalex":"https://openalex.org/W2921828045","mag":"2921828045"},"language":"en","primary_location":{"id":"pmh:oai:era.ed.ac.uk:1842/35531","doi":null,"is_oa":true,"landing_page_url":"http://hdl.handle.net/1842/35531","pdf_url":"http://hdl.handle.net/1842/35531","source":{"id":"https://openalex.org/S4377196353","display_name":"Edinburgh Research Archive (University of Edinburgh)","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I98677209","host_organization_name":"University of Edinburgh","host_organization_lineage":["https://openalex.org/I98677209"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"submittedVersion","is_accepted":false,"is_published":false,"raw_source_name":null,"raw_type":"Thesis or Dissertation"},"volume":null,"type":"dissertation","is_traditional":true,"indexed_in":[],"open_access":{"is_oa":true,"oa_status":"green","oa_url":"http://hdl.handle.net/1842/35531","any_repository_has_fulltext":true},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5030753822","display_name":"Sarah E. A. Eley","orcid":"https://orcid.org/0000-0001-8455-3670","observed_orcids":["https://orcid.org/0000-0001-8455-3670"]},"institutions":[],"countries":[],"is_corresponding":true,"raw_author_name":"Sarah Eley","raw_affiliation_strings":[],"raw_orcid":"https://orcid.org/0000-0001-8455-3670","affiliations":[]}],"institutions":[],"countries_distinct_count":0,"institutions_distinct_count":0,"corresponding_author_ids":["https://openalex.org/A5030753822"],"corresponding_institution_ids":[],"apc_list":null,"apc_paid":null,"fwci":null,"has_fulltext":true,"cited_by_count":0,"citation_normalized_percentile":null,"cited_by_percentile_year":null,"biblio":{"volume":null,"issue":null,"first_page":null,"last_page":null},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T10582","display_name":"Ethics in Clinical Research","score":0.9980000257492065,"subfield":{"id":"https://openalex.org/subfields/2739","display_name":"Public Health, Environmental and Occupational Health"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},"topics":[{"id":"https://openalex.org/T10582","display_name":"Ethics in Clinical Research","score":0.9980000257492065,"subfield":{"id":"https://openalex.org/subfields/2739","display_name":"Public Health, Environmental and Occupational Health"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T11772","display_name":"Genetics and Neurodevelopmental Disorders","score":0.0015999999595806003,"subfield":{"id":"https://openalex.org/subfields/1311","display_name":"Genetics"},"field":{"id":"https://openalex.org/fields/13","display_name":"Biochemistry, Genetics and Molecular Biology"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}},{"id":"https://openalex.org/T11632","display_name":"Family and Disability Support Research","score":0.0,"subfield":{"id":"https://openalex.org/subfields/3203","display_name":"Clinical Psychology"},"field":{"id":"https://openalex.org/fields/32","display_name":"Psychology"},"domain":{"id":"https://openalex.org/domains/2","display_name":"Social Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/fragile-x-syndrome","display_name":"fragile X syndrome","score":0.9559999704360962},{"id":"https://openalex.org/keywords/clinical-trial-enrollment","display_name":"clinical trial recruitment","score":0.8259999752044678},{"id":"https://openalex.org/keywords/barriers-to-research-participation","display_name":"barriers to research participation","score":0.9300000071525574},{"id":"https://openalex.org/keywords/family-caregivers","display_name":"family caregivers","score":0.5049999952316284},{"id":"https://openalex.org/keywords/research-participation-motivation","display_name":"research participation motivation","score":0.5799999833106995},{"id":"https://openalex.org/keywords/side-effect-concerns","display_name":"side effect concerns","score":0.6240000128746033},{"id":"https://openalex.org/keywords/travel-barriers","display_name":"travel barriers","score":0.8140000104904175},{"id":"https://openalex.org/keywords/financial-barriers","display_name":"financial barriers","score":0.7480000257492065},{"id":"https://openalex.org/keywords/autism-spectrum-disorder","display_name":"autism spectrum disorder","score":0.44699999690055847},{"id":"https://openalex.org/keywords/intellectual-and-developmental-disabilities","display_name":"intellectual and developmental disabilities","score":0.8759999871253967},{"id":"https://openalex.org/keywords/patient-and-public-involvement","display_name":"patient and public involvement","score":0.5460000038146973},{"id":"https://openalex.org/keywords/patient-centered-research","display_name":"patient-oriented research","score":0.3199999928474426},{"id":"https://openalex.org/keywords/informed-consent","display_name":"informed consent","score":0.39100000262260437}],"concepts":[{"id":"https://openalex.org/C2777630245","wikidata":"https://www.wikidata.org/wiki/Q221472","display_name":"Fragile X syndrome","level":2,"score":0.7336734533309937},{"id":"https://openalex.org/C15744967","wikidata":"https://www.wikidata.org/wiki/Q9418","display_name":"Psychology","level":0,"score":0.4028013348579407},{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.39868783950805664},{"id":"https://openalex.org/C70410870","wikidata":"https://www.wikidata.org/wiki/Q199906","display_name":"Clinical psychology","level":1,"score":0.3538539409637451},{"id":"https://openalex.org/C138496976","wikidata":"https://www.wikidata.org/wiki/Q175002","display_name":"Developmental psychology","level":1,"score":0.34693899750709534},{"id":"https://openalex.org/C512399662","wikidata":"https://www.wikidata.org/wiki/Q3505712","display_name":"Family medicine","level":1,"score":0.34086307883262634},{"id":"https://openalex.org/C118552586","wikidata":"https://www.wikidata.org/wiki/Q7867","display_name":"Psychiatry","level":1,"score":0.23796826601028442}],"mesh":[],"locations_count":2,"locations":[{"id":"pmh:oai:era.ed.ac.uk:1842/35531","doi":null,"is_oa":true,"landing_page_url":"http://hdl.handle.net/1842/35531","pdf_url":"http://hdl.handle.net/1842/35531","source":{"id":"https://openalex.org/S4377196353","display_name":"Edinburgh Research Archive (University of Edinburgh)","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I98677209","host_organization_name":"University of Edinburgh","host_organization_lineage":["https://openalex.org/I98677209"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"submittedVersion","is_accepted":false,"is_published":false,"raw_source_name":null,"raw_type":"Thesis or Dissertation"},{"id":"mag:2921828045","doi":null,"is_oa":false,"landing_page_url":"https://era.ed.ac.uk/handle/1842/35531","pdf_url":null,"source":null,"license":null,"license_id":null,"version":null,"is_accepted":false,"is_published":false,"raw_source_name":null,"raw_type":null}],"best_oa_location":{"id":"pmh:oai:era.ed.ac.uk:1842/35531","doi":null,"is_oa":true,"landing_page_url":"http://hdl.handle.net/1842/35531","pdf_url":"http://hdl.handle.net/1842/35531","source":{"id":"https://openalex.org/S4377196353","display_name":"Edinburgh Research Archive (University of Edinburgh)","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I98677209","host_organization_name":"University of Edinburgh","host_organization_lineage":["https://openalex.org/I98677209"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"submittedVersion","is_accepted":false,"is_published":false,"raw_source_name":null,"raw_type":"Thesis or Dissertation"},"sustainable_development_goals":[{"display_name":"Good health and well-being","score":1.0,"id":"https://metadata.un.org/sdg/3"}],"study_designs":[{"display_name":"Observational Study","id":"https://openalex.org/study-designs/observational-study"}],"awards":[],"funders":[{"id":"https://openalex.org/F4320320332","display_name":"University of Edinburgh","ror":"https://ror.org/01nrxwf90"}],"has_content":{"pdf":true,"grobid_xml":true},"content_urls":{"pdf":"https://content.openalex.org/works/W2921828045.pdf","grobid_xml":"https://content.openalex.org/works/W2921828045.grobid-xml"},"referenced_works_count":72,"referenced_works":["https://openalex.org/W30657002","https://openalex.org/W68065471","https://openalex.org/W181033957","https://openalex.org/W602710656","https://openalex.org/W622949211","https://openalex.org/W1486091068","https://openalex.org/W1544124124","https://openalex.org/W1547141139","https://openalex.org/W1547253197","https://openalex.org/W1575312775","https://openalex.org/W1604682389","https://openalex.org/W1636503892","https://openalex.org/W1726929985","https://openalex.org/W1971946761","https://openalex.org/W1976101979","https://openalex.org/W1979290264","https://openalex.org/W1994817684","https://openalex.org/W1997557106","https://openalex.org/W2001588996","https://openalex.org/W2004342279","https://openalex.org/W2005387522","https://openalex.org/W2009222294","https://openalex.org/W2012322293","https://openalex.org/W2021588093","https://openalex.org/W2021720741","https://openalex.org/W2023366466","https://openalex.org/W2031785254","https://openalex.org/W2035030950","https://openalex.org/W2037974201","https://openalex.org/W2040316692","https://openalex.org/W2042881656","https://openalex.org/W2043219730","https://openalex.org/W2045628508","https://openalex.org/W2048047187","https://openalex.org/W2051573474","https://openalex.org/W2056144694","https://openalex.org/W2059148325","https://openalex.org/W2060911896","https://openalex.org/W2063441624","https://openalex.org/W2065077390","https://openalex.org/W2069648006","https://openalex.org/W2071482305","https://openalex.org/W2073347350","https://openalex.org/W2076651184","https://openalex.org/W2079016689","https://openalex.org/W2084495849","https://openalex.org/W2091142866","https://openalex.org/W2093738396","https://openalex.org/W2099899772","https://openalex.org/W2101716121","https://openalex.org/W2103101481","https://openalex.org/W2105113140","https://openalex.org/W2116276594","https://openalex.org/W2126785094","https://openalex.org/W2127571361","https://openalex.org/W2136608375","https://openalex.org/W2138502971","https://openalex.org/W2143494630","https://openalex.org/W2144550671","https://openalex.org/W2147574101","https://openalex.org/W2148386452","https://openalex.org/W2149919973","https://openalex.org/W2156319664","https://openalex.org/W2157245091","https://openalex.org/W2159857276","https://openalex.org/W2162893002","https://openalex.org/W2168353812","https://openalex.org/W2171288469","https://openalex.org/W2172051343","https://openalex.org/W2237631658","https://openalex.org/W2493342998","https://openalex.org/W2796408370"],"related_works":["https://openalex.org/W3087680068","https://openalex.org/W2911619051","https://openalex.org/W2145511784","https://openalex.org/W1976050719","https://openalex.org/W2013514775","https://openalex.org/W4234590169","https://openalex.org/W2741177809","https://openalex.org/W3108820367","https://openalex.org/W4212953993","https://openalex.org/W2003274138"],"abstract_inverted_index":{"Fragile":[0,240,250],"x":[1],"syndrome":[2],"(FXS)":[3],"is":[4,32,57,414],"thought":[5],"to":[6,53,60,65,118,124,152,166,169,187,191,201,206,215,227,318,331,374,379,399,435],"be":[7,61,211],"the":[8,19,63,88,126,138,175,189,198,238,249,263,328,391,401],"most":[9,20,58],"common":[10,21,59],"inherited":[11],"form":[12],"of":[13,26,30,90,113,183,197,248,283,393],"intellectual":[14],"and":[15,18,38,86,103,177,231,259,269,302,344,370,446],"developmental":[16],"disability":[17],"known":[22],"single":[23],"gene":[24],"cause":[25],"autism.":[27],"The":[28,75,180,194,253,376],"prevalence":[29],"FXS":[31,48,80,114,286],"approximately":[33],"1.4":[34],"per":[35,40],"10,000":[36,41],"males":[37,69],"0.9":[39],"females.":[42,74],"Learning":[43],"difficulties":[44],"in":[45,84,155,213,299,320,324,386,396],"people":[46],"with":[47,68,174,285,290],"can":[49,420,433],"vary":[50],"from":[51,237],"mild":[52,64],"severe":[54],"however":[55],"it":[56,355],"within":[62],"moderate":[66],"range,":[67],"often":[70],"more":[71,443],"affected":[72],"than":[73],"current":[76],"medical":[77],"treatments":[78,122],"for":[79,95,98,105,218,341,425],"are":[81,129,150,309,428],"primarily":[82],"symptomatic":[83],"nature;":[85],"include":[87],"use":[89],"selective":[91],"serotonin":[92],"re-uptake":[93],"inhibitors":[94],"anxiety,":[96],"stimulants":[97],"attention":[99],"deficit":[100],"hyperactivity":[101],"disorder":[102],"antipsychotics":[104],"irritability":[106],"or":[107,287],"aggressive":[108],"behaviour.":[109],"However,":[110,327],"our":[111],"understanding":[112],"has":[115,145],"now":[116,130],"progressed":[117],"a":[119,246,321],"stage":[120],"where":[121],"selected":[123],"target":[125],"underlying":[127],"neurobiology":[128],"being":[131],"trialled.":[132],"Experience":[133],"conducting":[134],"research":[135,192,442],"studies":[136],"at":[137],"Patrick":[139,254,264],"Wild":[140,255,265],"Centre":[141,256,266],"into":[142],"this":[143,162,184,325],"condition":[144,176],"suggested":[146],"that":[147,307,358,382,411,418,422,431],"some":[148],"families":[149,398],"reluctant":[151],"take":[153],"part":[154],"clinical":[156,207,322],"trials":[157],"making":[158,440],"recruitment":[159,217],"challenging.":[160],"Therefore,":[161],"study":[163,185,199,305,402,409],"was":[164,186,200,225,275,356,383,390],"designed":[165],"understand":[167,400],"barriers":[168,190,205,330,360,381],"participation":[170,209,332,437],"specifically":[171],"amongst":[172],"patients":[173],"their":[178,441],"families.":[179],"primary":[181],"aim":[182,196],"identify":[188],"participation.":[193],"second":[195],"find":[202],"out":[203],"how":[204],"trial":[208,323],"could":[210],"overcome":[212,380],"order":[214],"maximise":[216],"future":[219],"studies.":[220],"A":[221,272],"mixed":[222],"method":[223],"design":[224],"used":[226],"collect":[228],"both":[229],"quantitative":[230,273],"qualitative":[232],"data.":[233],"Participants":[234],"were":[235,333,349,361],"identified":[236],"UK":[239],"X":[241,251],"Society":[242],"mailing":[243,257],"list":[244,258],"by":[245,277,288],"member":[247],"Society,":[252],"through":[260],"advertisements":[261],"on":[262],"website,":[267],"Facebook":[268],"Twitter":[270],"feeds.":[271],"questionnaire":[274],"completed":[276],"328":[278],"parents,":[279],"carers,":[280],"family":[281],"members":[282],"individuals":[284,289],"FXS.":[291],"Following":[292],"this,":[293],"three":[294],"focus":[295,353,388],"groups":[296,354],"took":[297],"place":[298],"Chelmsford,":[300],"Bristol":[301],"Edinburgh.":[303],"This":[304,408],"demonstrated":[306],"there":[308,413,427],"many":[310,372,429],"different":[311],"factors":[312],"which":[313],"may":[314,367],"negatively":[315],"influence":[316],"motivation":[317],"participate":[319],"group.":[326],"main":[329,377],"concerns":[334],"about":[335],"possible":[336],"side":[337],"effects,":[338],"travel,":[339],"requirement":[340],"blood":[342],"tests":[343],"financial":[345],"reasons.":[346],"When":[347],"these":[348,359],"explored":[350],"further":[351],"during":[352],"evident":[357],"not":[362],"as":[363,365,439],"clear":[364],"they":[366],"have":[368],"appeared":[369],"had":[371],"complexities":[373],"them.":[375],"way":[378],"repeatedly":[384],"discussed":[385],"each":[387],"group":[389],"importance":[392],"accessible":[394],"information":[395],"helping":[397],"including":[403],"any":[404],"potential":[405],"safety":[406],"issues.":[407],"highlights":[410],"while":[412],"no":[415],"one":[416],"thing":[417],"researchers":[419,432],"do":[421,434],"will":[423],"work":[424],"everyone,":[426],"things":[430],"improve":[436],"such":[438],"individual,":[444],"flexible":[445],"accommodating.":[447]},"counts_by_year":[],"updated_date":"2026-11-08T12:12:28.789561","created_date":"2019-03-22T00:00:00"}
