{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T12:17:04Z","timestamp":1788437824589,"version":"build-2803163510"},"reference-count":11,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2026,5,21]],"date-time":"2026-05-21T00:00:00Z","timestamp":1779321600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,5,21]],"date-time":"2026-05-21T00:00:00Z","timestamp":1779321600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100006093","name":"Patient-Centered Outcomes Research Institute","doi-asserted-by":"publisher","award":["EOSO\u201040455"],"award-info":[{"award-number":["EOSO\u201040455"]}],"id":[{"id":"10.13039\/100006093","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J. pediatr. gastroenterol. nutr."],"published-print":{"date-parts":[[2026,8]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>In rare diseases like progressive familial intrahepatic cholestasis (PFIC), lack of evidence results in treatment variability and strategies for families to engage care teams to understand how variability affects overall health are unknown. Patient\u2010centered comparative effectiveness research (CER) has been identified as an approach to address these challenges, but CER\u2010specific knowledge and infrastructure are lacking. We present the results from IMPACT (Identifying research targets by Merging Patient And Clinician Treatment information), a 2\u2010year capacity\u2010building project for patient\u2010centered CER.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Six learning modules were adapted from the Patient\u2010Centered Outcomes Research Institute to teach participants about CER and elicit feedback for patient\u2010specific CER priorities and processes in PFIC. Participants completed modules and engaged in focus groups (12 virtual and in\u2010person focus groups). Engagement quality was assessed using a validated patient engagement evaluation tool.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Participation in learning modules varied between 16 and 31 patient\/family participants (mean 22), and 5 and 8 clinicians\/researchers (mean 5.3). Focus group participation varied between 7 and 35 patient\/family participants (mean 13.1) and 3\u201335 clinicians\/researchers (mean 7.3). Collated data enabled develop of (1) the treatment experience app (TEA): a web\u2010based, community\u2010built information tool to display relevant, research\u2010based, lay language therapeutic approaches, augmented by patient experience information and (2) the IMPACT roadmap, a manual for future PFIC patient\u2010centered research. Engagement assessments demonstrate consistently strong and steadily improving levels of engagement.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>IMPACT successfully engaged key stakeholders to build shared understanding of patient\u2010centered CER, define PFIC\u2010specific CER targets, and develop strategies for equitable and sustainable patient\u2013researcher partnerships.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/jpn3.70455","type":"journal-article","created":{"date-parts":[[2026,5,21]],"date-time":"2026-05-21T10:50:13Z","timestamp":1779360613000},"page":"215-222","source":"Crossref","is-referenced-by-count":0,"title":["Identifying patient\u2010centered outcomes in progressive familial intrahepatic cholestasis: Results from IMPACT"],"prefix":"10.1002","volume":"83","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6979-8987","authenticated-orcid":false,"given":"James E.","family":"Squires","sequence":"first","affiliation":[{"name":"Division of Gastroenterology and Hepatology UPMC Children's Hospital of Pittsburgh Pittsburgh Pennsylvania USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gitta H.","family":"Lubke","sequence":"additional","affiliation":[{"name":"PFIC Network Stanton Kentucky USA"},{"name":"University of Notre Dame South Bend Indiana USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emily","family":"Ventura","sequence":"additional","affiliation":[{"name":"PFIC Network Stanton Kentucky USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexandra","family":"Perez","sequence":"additional","affiliation":[{"name":"PFIC Network Stanton Kentucky USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paula M.","family":"Hertel","sequence":"additional","affiliation":[{"name":"Division of Pediatric Gastroenterology and Hepatology Texas Children's Hospital Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emily R.","family":"Perito","sequence":"additional","affiliation":[{"name":"Division of Gastroenterology and Hepatology UCSF Benioff Children's Hospital San Francisco California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Bull","sequence":"additional","affiliation":[{"name":"Department of Medicine, Division of Gastroenterology and Hepatology, UCSF, Liver Center Institute for Human Genetics San Francisco California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Melissa","family":"Kochanowsky","sequence":"additional","affiliation":[{"name":"PFIC Network Stanton Kentucky USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,5,21]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cld.2022.03.003"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.4254\/wjh.v11.i5.450"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejmg.2021.104317"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.3748\/wjg.v26.i47.7470"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jhep.2024.04.006"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/jpn3.70366"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajt.2024.01.030"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1097\/MPG.0000000000002974"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/petr.13528"},{"key":"e_1_2_7_11_1","unstructured":"Patient\u2010Centered Outcomes Research Institute. Research fundamentals: preparing you to successfully contribute to research; 2025.https:\/\/www.pcori.org\/engagement-research\/engagement-resources\/research-fundamentals-preparing-you-successfully-contribute-research"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jclinepi.2021.11.034"}],"container-title":["Journal of Pediatric Gastroenterology and Nutrition"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/jpn3.70455","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/jpn3.70455","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/jpn3.70455","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T11:52:46Z","timestamp":1788436366000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/jpn3.70455"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,5,21]]},"references-count":11,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2026,8]]}},"alternative-id":["10.1002\/jpn3.70455"],"URL":"https:\/\/doi.org\/10.1002\/jpn3.70455","archive":["Portico"],"relation":{},"ISSN":["0277-2116","1536-4801"],"issn-type":[{"value":"0277-2116","type":"print"},{"value":"1536-4801","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,5,21]]}}}