{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,24]],"date-time":"2026-06-24T00:56:59Z","timestamp":1782262619467,"version":"3.54.5"},"reference-count":47,"publisher":"SAGE Publications","issue":"7","license":[{"start":{"date-parts":[[2018,2,26]],"date-time":"2018-02-26T00:00:00Z","timestamp":1519603200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["The Cleft Palate Craniofacial Journal"],"published-print":{"date-parts":[[2018,8]]},"abstract":"<jats:sec>\n                    <jats:title>Objective:<\/jats:title>\n                    <jats:p>The aim of this systematic review was to identify patient-reported outcome (PRO) instruments used in research with children\/youth with conditions associated with facial differences to identify the health concepts measured.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design:<\/jats:title>\n                    <jats:p>MEDLINE, EMBASE, CINAHL, and PsycINFO were searched from 2004 to 2016 to identify PRO instruments used in acne vulgaris, birthmarks, burns, ear anomalies, facial asymmetries, and facial paralysis patients. We performed a content analysis whereby the items were coded to identify concepts and categorized as positive or negative content or phrasing.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>A total of 7,835 articles were screened; 6 generic and 11 condition-specific PRO instruments were used in 96 publications. Condition-specific instruments were for acne (four), oral health (two), dermatology (one), facial asymmetries (two), microtia (one), and burns (one). The PRO instruments provided 554 items (295 generic; 259 condition specific) that were sorted into 4 domains, 11 subdomains, and 91 health concepts. The most common domain was psychological (n = 224 items). Of the identified items, 76% had negative content or phrasing (e.g., \u201cBecause of the way my face looks I wish I had never been born\u201d). Given the small number of items measuring facial appearance (n = 19) and function (n = 22), the PRO instruments reviewed lacked content validity for patients whose condition impacted facial function and\/or appearance.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions:<\/jats:title>\n                    <jats:p>Treatments can change facial appearance and function. This review draws attention to a problem with content validity in existing PRO instruments. Our team is now developing a new PRO instrument called FACE-Q Kids to address this problem.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1597\/16-148","type":"journal-article","created":{"date-parts":[[2016,12,21]],"date-time":"2016-12-21T11:20:26Z","timestamp":1482319226000},"page":"989-998","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":15,"title":["Content Validity of Patient-Reported Outcome Instruments used with Pediatric Patients with Facial Differences"],"prefix":"10.1177","volume":"55","author":[{"given":"Natasha M.","family":"Wickert","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karen W.Y.","family":"Wong Riff","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark","family":"Mansour","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christopher R.","family":"Forrest","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Timothy E.E.","family":"Goodacre","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrea L.","family":"Pusic","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne F.","family":"Klassen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2018,2,26]]},"reference":[{"key":"bibr1-16-148","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.jaad.2012.06.023","volume":"67","author":"Alikhan A.","year":"2012","journal-title":"J Am Acad Dermatol"},{"key":"bibr2-16-148","unstructured":"American Academy of Dermatology. Acne. Available at https:\/\/www.aad.org\/media-resources\/stats-and-facts\/conditions\/acne. Accessed March 1, 2015."},{"key":"bibr3-16-148","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.f167"},{"key":"bibr4-16-148","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-009-9540-9"},{"key":"bibr5-16-148","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0528.2007.00401.x"},{"key":"bibr6-16-148","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0528.2007.0002.x"},{"key":"bibr7-16-148","doi-asserted-by":"publisher","DOI":"10.1097\/PRS.0b013e31822b6a67"},{"key":"bibr8-16-148","doi-asserted-by":"publisher","DOI":"10.1006\/jado.2002.0470"},{"key":"bibr9-16-148","doi-asserted-by":"publisher","DOI":"10.1597\/03-097.2.1"},{"key":"bibr10-16-148","doi-asserted-by":"publisher","DOI":"10.1097\/00006534-199001000-00001"},{"key":"bibr11-16-148","doi-asserted-by":"publisher","DOI":"10.1097\/PRS.0b013e31823aedd9"},{"key":"bibr12-16-148","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-011-9851-5"},{"key":"bibr13-16-148","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaad.2010.05.045"},{"key":"bibr14-16-148","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19990326)89:1<1::AID-AJMG3>3.0.CO;2-8"},{"key":"bibr15-16-148","doi-asserted-by":"publisher","DOI":"10.1007\/BF00540020"},{"key":"bibr16-16-148","unstructured":"Government of Canada.\nCanadian Institutes of Health Research. Available at http:\/\/www.cihr-irsc.gc.ca\/e\/193.html. Accessed August 11, 2016."},{"key":"bibr17-16-148","doi-asserted-by":"publisher","DOI":"10.1177\/154405910208100705"},{"key":"bibr18-16-148","doi-asserted-by":"publisher","DOI":"10.1111\/j.1525-1470.2008.00626.x"},{"key":"bibr19-16-148","volume-title":"The CHQ: A User's Manual","author":"Landgraf J.M.","year":"1999"},{"key":"bibr20-16-148","doi-asserted-by":"publisher","DOI":"10.1159\/000246015"},{"key":"bibr21-16-148","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2133.1995.tb16953.x"},{"key":"bibr22-16-148","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.34352"},{"key":"bibr23-16-148","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2230.2001.00839.x"},{"key":"bibr24-16-148","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.b2535"},{"key":"bibr25-16-148","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-010-9606-8"},{"key":"bibr26-16-148","doi-asserted-by":"publisher","DOI":"10.14219\/jada.archive.2001.0055"},{"key":"bibr27-16-148","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2230.1992.tb02521.x"},{"key":"bibr28-16-148","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2011.06.014"},{"key":"bibr29-16-148","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2011.06.013"},{"key":"bibr30-16-148","doi-asserted-by":"publisher","DOI":"10.1597\/06-072.1"},{"key":"bibr31-16-148","unstructured":"PROMIS.\nDomain frameworks PROMIS Pediatric Self- and Proxy-Reported Health. Available at http:\/\/nihpromis.net\/(X(1)S(an1jgz0whyvrnjdy3x3m0d4f)\/measures\/domainframework2. Accessed March 1, 2015."},{"key":"bibr32-16-148","doi-asserted-by":"publisher","DOI":"10.2165\/00128071-200607030-00005"},{"key":"bibr33-16-148","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-007-9240-2"},{"key":"bibr34-16-148","unstructured":"Ravens-Sieberer U., Bullinger M. KINDL\n                      R\n                      questionnaire for measuring health-related quality of life in children and adolescents revised version manual. Available at http:\/\/kindl.org\/cms\/impressum. Accessed March 1, 2015."},{"key":"bibr35-16-148","doi-asserted-by":"publisher","DOI":"10.3109\/00016489.2012.658967"},{"key":"bibr36-16-148","doi-asserted-by":"publisher","DOI":"10.1177\/1359104509355021"},{"key":"bibr37-16-148","doi-asserted-by":"publisher","DOI":"10.1597\/09-050"},{"key":"bibr38-16-148","doi-asserted-by":"publisher","DOI":"10.1080\/13638490701217396"},{"key":"bibr39-16-148","doi-asserted-by":"publisher","DOI":"10.1097\/BCR.0b013e3181776ed9"},{"key":"bibr40-16-148","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-006-0063-3"},{"key":"bibr41-16-148","unstructured":"U.S. Department of Health, and Human Services. Guidance for industry patient-reported outcome measures: use in medical product development to support labeling claims. Available at http:\/\/www.ispor.org\/workpaper\/FDA%20PRO%20Guidance.pdf. 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