{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,5]],"date-time":"2026-08-05T08:47:22Z","timestamp":1785919642231,"version":"3.56.0"},"reference-count":61,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,8,5]],"date-time":"2026-08-05T00:00:00Z","timestamp":1785888000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,8,5]],"date-time":"2026-08-05T00:00:00Z","timestamp":1785888000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["American J of Med Genetics Pt C"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Arthrogryposis Multiplex Congenita (AMC), a rare congenital condition characterized by joint contractures in two or more body regions, is often accompanied by pain. Understanding pain and its relationship to health outcomes among adults with AMC is critical for effective pain management. The objectives of this survey study were to characterize the pain experience in adults with AMC and determine the extent to which pain intensity relates to health, while considering potential sex differences. Participants (\n                    <jats:italic>n<\/jats:italic>\n                    \u2009=\u200973, 67.3% female) reported average pain intensity, severity, and extent per body diagrams, and completed the Pain Catastrophizing Scale and 29\u2010item Patient\u2010Reported Outcomes Measurement Information System. In the preceding 7\u2009days, 82.7% of participants reported pain; females were more likely to report pain. Pain intensity was significantly greater in females than males. Beyond covariates of sex and AMC severity, higher pain intensity was associated with worse physical function and ability to participate in social roles, as well as greater sleep disturbance. Female sex and an interaction term (sex\u2009\u00d7\u2009pain intensity) were significantly associated with greater fatigue. Findings highlight the importance of considering sex\u2010specific differences in pain and the relationship between pain intensity and fatigue among adults with AMC.\n                  <\/jats:p>","DOI":"10.1002\/ajmg.c.70021","type":"journal-article","created":{"date-parts":[[2026,8,5]],"date-time":"2026-08-05T08:08:12Z","timestamp":1785917292000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Pain and Health: Sex\u2010Specific Differences From a Survey Study of Adults With Arthrogryposis Multiplex Congenita"],"prefix":"10.1002","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8151-1341","authenticated-orcid":false,"given":"Jaclyn Megan","family":"Sions","sequence":"first","affiliation":[{"name":"Department of Physical Therapy University of Delaware  Newark Delaware USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8878-1857","authenticated-orcid":false,"given":"Maureen","family":"Donohoe","sequence":"additional","affiliation":[{"name":"Department of Physical Therapy University of Delaware  Newark Delaware USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-1056-0793","authenticated-orcid":false,"given":"Claire Elizabeth","family":"Vallery","sequence":"additional","affiliation":[{"name":"Department of Physical Therapy University of Delaware  Newark Delaware USA"},{"name":"Dankmeyer Inc  Linthicum Heights Maryland USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3050-1071","authenticated-orcid":false,"given":"Samantha Jeanne","family":"Stauffer","sequence":"additional","affiliation":[{"name":"Department of Physical Therapy University of Delaware  Newark Delaware USA"},{"name":"Independence Prosthetics\u2010Orthotics Inc  Newark Delaware USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8385-8218","authenticated-orcid":false,"given":"Ryan Todd","family":"Pohlig","sequence":"additional","affiliation":[{"name":"Department of Epidemiology University of Delaware  Newark Delaware USA"},{"name":"Biostatistics Core University of Delaware  Newark Delaware USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,8,5]]},"reference":[{"key":"e_1_2_12_2_1","doi-asserted-by":"publisher","DOI":"10.3390\/medicina60081305"},{"key":"e_1_2_12_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31717"},{"key":"e_1_2_12_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.apmr.2015.03.024"},{"key":"e_1_2_12_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpsychires.2011.09.004"},{"key":"e_1_2_12_6_1","doi-asserted-by":"publisher","DOI":"10.1037\/emo0001228"},{"key":"e_1_2_12_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jclinepi.2010.04.011"},{"key":"e_1_2_12_8_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41598\u2010024\u201067561\u20101"},{"key":"e_1_2_12_9_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.mlr.0000163658.65008.ec"},{"key":"e_1_2_12_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpain.2018.07.009"},{"key":"e_1_2_12_11_1","doi-asserted-by":"publisher","DOI":"10.1097\/BPO.0000000000003007"},{"key":"e_1_2_12_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31725"},{"key":"e_1_2_12_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31721"},{"key":"e_1_2_12_14_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000005418"},{"key":"e_1_2_12_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpain.2024.01.339"},{"key":"e_1_2_12_16_1","doi-asserted-by":"publisher","DOI":"10.1177\/0309364618774059"},{"key":"e_1_2_12_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpain.2015.08.010"},{"key":"e_1_2_12_18_1","doi-asserted-by":"publisher","DOI":"10.3390\/children10050768"},{"key":"e_1_2_12_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12160\u2010016\u20109860\u20102"},{"key":"e_1_2_12_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/sms.12611"},{"key":"e_1_2_12_21_1","doi-asserted-by":"publisher","DOI":"10.1682\/JRRD.2015.12.0228"},{"key":"e_1_2_12_22_1","first-page":"12","volume-title":"Introduction to Mediation, Moderation, and Conditional Process Analysis: A Regression\u2010Based Approach","author":"Hayes A. 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