{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,9]],"date-time":"2026-03-09T09:36:34Z","timestamp":1773048994438,"version":"3.50.1"},"reference-count":29,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2018,10,19]],"date-time":"2018-10-19T00:00:00Z","timestamp":1539907200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100004191","name":"Novo Nordisk","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004191","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["European J of Haematology"],"published-print":{"date-parts":[[2018,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>To assess the reliability and validity of six patient\u2010reported outcomes (PRO) instruments for evaluating health\u2010related quality of life in adults with mild\u2010severe hemophilia B and caregivers of children with hemophilia B, including affected women\/girls.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Adults with hemophilia B and caregivers completed separate online surveys containing several PRO instruments, which were administered to adult participants only (EQ\u20105D\u20105L, Brief Pain Inventory v2 Short Form, Hemophilia Activities List, and International Physical Activities Questionnaire), both adults and caregivers (Patient Health Questionnaire [PHQ\u20109]), or caregivers only (Generalized Anxiety Disorder 7\u2010Item [GAD\u20107] scale). Construct validity and item\u2010total correlation were assessed using Pearson product\u2010moment correlation, internal consistency was assessed using Cronbach\u2019s alpha coefficient, and known\u2010group validity was assessed by comparisons to self\u2010reported characteristics based on the Kruskal\u2010Wallis test.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Patient\u2010reported outcomes instruments generally showed satisfactory reliability for adults (n\u00a0=\u00a0299) and caregivers (n\u00a0=\u00a0150). In adults, PRO instruments generally showed high construct validity. Most PRO instruments showed expected significant differences among known groups for adults and caregivers. PHQ\u20109 and GAD\u20107 did not show significant differences among caregiver age groups.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Patient\u2010reported outcomes instruments administered in B\u2010HERO\u2010S demonstrated reliability and validity in the broader population of adults with hemophilia B and caregivers when including all severities and genders.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ejh.13168","type":"journal-article","created":{"date-parts":[[2018,9,4]],"date-time":"2018-09-04T14:18:27Z","timestamp":1536070707000},"page":"781-790","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["Reliability and validity of patient\u2010reported outcome instruments in US adults with hemophilia B and caregivers in the B\u2010HERO\u2010S study"],"prefix":"10.1111","volume":"101","author":[{"given":"Tyler W.","family":"Buckner","sequence":"first","affiliation":[{"name":"University of Colorado School of Medicine Aurora Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"suffix":"Jr","given":"Robert","family":"Sidonio","sequence":"additional","affiliation":[{"name":"Emory University School of Medicine Atlanta Georgia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christine","family":"Guelcher","sequence":"additional","affiliation":[{"name":"Children\u2019s National Health System Washington District of Columbia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Craig M.","family":"Kessler","sequence":"additional","affiliation":[{"name":"Georgetown University Hospital Washington District of Columbia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michelle","family":"Witkop","sequence":"additional","affiliation":[{"name":"National Hemophilia Foundation New York New York"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"David","family":"Clark","sequence":"additional","affiliation":[{"name":"The Coalition for Hemophilia B New York New York"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wendy","family":"Owens","sequence":"additional","affiliation":[{"name":"Hemophilia Federation of America Washington District of Columbia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Moshe","family":"Fridman","sequence":"additional","affiliation":[{"name":"AMF Consulting Inc. Los Angeles California"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Neeraj N.","family":"Iyer","sequence":"additional","affiliation":[{"name":"Novo Nordisk Inc. Plainsboro New Jersey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4499-1526","authenticated-orcid":false,"given":"David L.","family":"Cooper","sequence":"additional","affiliation":[{"name":"Novo Nordisk Inc. Plainsboro New Jersey"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2018,10,19]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2012.02909.x"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.5435\/00124635-200407000-00004"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.seminhematol.2015.10.004"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejh.12497"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.2147\/PPA.S87659"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2012.05.005"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2005-09-3879"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12239"},{"key":"e_1_2_8_10_1","volume-title":"The HTC Population Profile","author":"Counts C","year":"2016"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejh.12854"},{"key":"e_1_2_8_12_1","unstructured":"Guidelines for Data Processing and Analysis of the International Physical Activity Questionnaire (IPAQ).Short and long forms 2005.https:\/\/www.ipaq.ki.se\/dloads\/IPAQ%20LS%20Scoring%20Protocols_Nov05.pdf. Accessed May 1 2018."},{"key":"e_1_2_8_13_1","unstructured":"KroenkeK.Instruction manual \u2010 instructions for Patient Health Questionnaire (PHQ) and GAD\u20107 measures.2010.phqscreeners.com. Accessed September 26 2017."},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1001\/archinte.166.10.1092"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-011-9903-x"},{"key":"e_1_2_8_16_1","volume-title":"The Brief Pain Inventory. User Guide","author":"Cleeland C","year":"2009"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2006.01186.x"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1525-1497.2001.016009606.x"},{"key":"e_1_2_8_19_1","volume-title":"Psychometric Theory","author":"Nunnally J","year":"1978"},{"key":"e_1_2_8_20_1","doi-asserted-by":"crossref","DOI":"10.1093\/oso\/9780198528470.001.0001","volume-title":"Health Measurement Scales","author":"Streiner DL","year":"2003"},{"key":"e_1_2_8_21_1","first-page":"118","volume-title":"Computing Test\u2010Reliability. A Handbook of Test Construction: Introduction to Psychometric Design","author":"Kline P","year":"1986"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1037\/0033-2909.112.1.155"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejh.12851"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejh.12852"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejh.13055"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.2147\/PPA.S141390"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.2147\/PPA.S141389"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.2147\/PPA.S141391"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.2147\/PPA.S141392"},{"key":"e_1_2_8_30_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpain.2011.03.003"}],"container-title":["European Journal of Haematology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fejh.13168","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/ejh.13168","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,7,9]],"date-time":"2024-07-09T22:48:35Z","timestamp":1720565315000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/ejh.13168"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,10,19]]},"references-count":29,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2018,12]]}},"alternative-id":["10.1111\/ejh.13168"],"URL":"https:\/\/doi.org\/10.1111\/ejh.13168","archive":["Portico"],"relation":{},"ISSN":["0902-4441","1600-0609"],"issn-type":[{"value":"0902-4441","type":"print"},{"value":"1600-0609","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,10,19]]},"assertion":[{"value":"2018-05-14","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-08-30","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-10-19","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}