{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,28]],"date-time":"2026-08-28T20:13:52Z","timestamp":1787948032662,"version":"build-2784847793"},"reference-count":31,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2019,5,17]],"date-time":"2019-05-17T00:00:00Z","timestamp":1558051200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100000024","name":"Canadian Institutes of Health Research","doi-asserted-by":"publisher","award":["PEG \u2010151805"],"award-info":[{"award-number":["PEG \u2010151805"]}],"id":[{"id":"10.13039\/501100000024","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100009520","name":"Rick Hansen Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100009520","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["American J of Med Genetics Pt C"],"published-print":{"date-parts":[[2019,9]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Arthrogryposis multiplex congenita (AMC) is considered a rare disorder resulting in multiple congenital contractures in two or more areas. Considerable literature is available on managing the contractures during an affected child's development but little information is available to those managing these ongoing issues in adulthood. Due to the heterogeneity etiological factors and presentation of AMC, and the small sample sizes of previous studies, it has been difficult to generalize results to the adult population. This current study presents the several steps taken to create an international AMC database for adults to populate with their own data over time. The methods included a scoping review of the literature for valid and reliable outcome measures used for AMC, a Delphi methodology to create the database with a team of clinicians, researchers and patients, a Beta testing of the database, and a final launch of the Adult AMC Registry. This registry includes 48 nonstandardized questions and 12 standardized questionnaires. It takes 35\u201345\u2009min for a participant to complete. A shorter version will be created for participants to complete for years 2 and 3, followed by this longer version every 4\u2009years. The protocol for referring English\u2010speaking patients and access to the registry is provided. Data will be reviewed every year to ensure quality. The registry will be maintained for a minimum of 10\u2009years and data will be comprehensively analyzed every 5\u2009years. Our goal is to have 500 adults with AMC from around the world as participants.<\/jats:p>","DOI":"10.1002\/ajmg.c.31706","type":"journal-article","created":{"date-parts":[[2019,5,17]],"date-time":"2019-05-17T14:03:02Z","timestamp":1558101782000},"page":"454-460","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Development of an online registry for adults with arthrogryposis multiplex congenita: A protocol paper"],"prefix":"10.1002","volume":"181","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8901-2301","authenticated-orcid":false,"given":"Bonita","family":"Sawatzky","sequence":"first","affiliation":[{"name":"Department of Orthopaedics University of British Columbia  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8567-7173","authenticated-orcid":false,"given":"N.","family":"Dahan\u2010Oliel","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation Therapy, Shriner's Hospital for Children  Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ann\u2010Marie","family":"Davison","sequence":"additional","affiliation":[{"name":"Kwantlen Polytechnic University  Surrey British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5062-491X","authenticated-orcid":false,"given":"Judith","family":"Hall","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics University of British Columbia  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Harold","family":"Van Bosse","sequence":"additional","affiliation":[{"name":"Shriner's Hospital for Children  Philadelphia Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"W. Ben","family":"Mortenson","sequence":"additional","affiliation":[{"name":"Occupational Science and Occupational Therapy University of British Columbia  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"Registry Team","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2019,5,17]]},"reference":[{"key":"e_1_2_10_2_1","unstructured":"AltiokH FlanaganA KrzakiJ HassaniS.(2018)Long\u2010term follow up on quality of life and function of adults with arthrogryposis. Paper presented at the 3rd International Symposium on Arthrogryposis Philadelphia USA September 20\u201323."},{"issue":"5","key":"e_1_2_10_3_1","first-page":"1038","article-title":"Development of the QuickDASH: Comparison of three item\u2010reduction approaches","volume":"87","author":"Beaton D. 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