{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,8]],"date-time":"2026-01-08T20:04:43Z","timestamp":1767902683926,"version":"3.49.0"},"reference-count":52,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2016,11,14]],"date-time":"2016-11-14T00:00:00Z","timestamp":1479081600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"NIH","award":["K23 NS070933 T32 NS070201 T32 NS070201-12"],"award-info":[{"award-number":["K23 NS070933 T32 NS070201 T32 NS070201-12"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2017,2]]},"abstract":"<jats:sec><jats:label\/><jats:p>The clinical manifestations of Marfan syndrome frequently cause pain. This study aimed to characterize pain in a cohort of adults with Marfan syndrome and investigate demographic, physical, and psychological factors associated with pain and pain\u2010related disability. Two hundred and forty\u2010five participants (73% female, 89% non\u2010Hispanic white, 90% North American) completed an online questionnaire assessing clinical features of Marfan syndrome, pain severity, pain\u2010related disability, physical and mental health, depressive symptoms, pain catastrophizing, and insomnia. Eighty\u2010nine percent of respondents reported having pain with 28% of individuals reporting pain as a presenting symptom of Marfan syndrome. Almost half of individuals reported that pain has spread from its initial site. Participants in our study reported poor physical and mental health functioning, moderate pain\u2010related disability, and mild levels of depressive symptoms, sleep disturbances, and pain catastrophizing. Those who identified pain as an initial symptom of Marfan syndrome and those who reported that pain had spread from its initial site reported greater psychological burden compared with those without pain as an initial symptom or pain spreading. Physical health is the largest predictor of pain severity and pain\u2010related disability. While pain catastrophizing and worse mental health functioning are significant correlates of pain severity and pain\u2010related disability, respectively. Pain is a significant and persistent problem in Marfan syndrome and is associated with profound disability and psychological burden. Further studies are indicated to better characterize the directionality of pain, pain\u2010related disability, and psychological burden in Marfan syndrome. \u00a9 2016 Wiley Periodicals, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/ajmg.a.38051","type":"journal-article","created":{"date-parts":[[2016,11,16]],"date-time":"2016-11-16T16:41:35Z","timestamp":1479314495000},"page":"315-323","source":"Crossref","is-referenced-by-count":38,"title":["Characterization of pain, disability, and psychological burden in Marfan syndrome"],"prefix":"10.1002","volume":"173","author":[{"given":"Traci J.","family":"Speed","sequence":"first","affiliation":[{"name":"Department of Psychiatry and Behavioral Sciences Johns Hopkins University School of Medicine Baltimore Maryland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vani A.","family":"Mathur","sequence":"additional","affiliation":[{"name":"Department of Psychology, Texas A&amp;M University College Station Texas"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Matthew","family":"Hand","sequence":"additional","affiliation":[{"name":"Department of Psychiatry and Behavioral Sciences Johns Hopkins University School of Medicine Baltimore Maryland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bryt","family":"Christensen","sequence":"additional","affiliation":[{"name":"Division Pain Medicine, Department of Anesthesiology and Critical Care Medicine Johns Hopkins University School of Medicine Baltimore Maryland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Paul D.","family":"Sponseller","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery Johns Hopkins University School of Medicine Baltimore Maryland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kayode A.","family":"Williams","sequence":"additional","affiliation":[{"name":"Division Pain Medicine, Department of Anesthesiology and Critical Care Medicine Johns Hopkins University School of Medicine Baltimore Maryland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Claudia M.","family":"Campbell","sequence":"additional","affiliation":[{"name":"Department of Psychiatry and Behavioral Sciences Johns Hopkins University School of Medicine Baltimore Maryland"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2016,11,14]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1007\/s002560100323"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.36574"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/0272-7358(88)90050-5"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pain.2007.03.024"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0005-7967(03)00029-9"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1037\/0022-006X.71.1.81"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.2217\/pmt.12.7"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1214\/ss\/1009213286"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1159\/000082265"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.ejhg.5201851"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/4.suppl_1.1799"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1080\/10790268.2010.11689700"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.21865"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrrheum.2011.2"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1097\/00007632-200011010-00025"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0304-3959(01)00349-9"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.30525"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0895-4356(98)00109-7"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/34.2.126"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1742-1241.2007.01407.x"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpainsymman.2010.08.016"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1249\/JSR.0b013e3181d4066c"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1093\/oxfordjournals.pubmed.a024606"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/0304-3959(93)90148-I"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpain.2010.08.009"},{"key":"e_1_2_7_27_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.2009.072785"},{"key":"e_1_2_7_28_1","doi-asserted-by":"publisher","DOI":"10.3109\/10582452.2010.538821"},{"key":"e_1_2_7_29_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.spinee.2012.10.021"},{"key":"e_1_2_7_30_1","doi-asserted-by":"publisher","DOI":"10.1097\/AJP.0000000000000202"},{"key":"e_1_2_7_31_1","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCULATIONAHA.108.783753"},{"key":"e_1_2_7_32_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1399-0004.2001.600406.x"},{"key":"e_1_2_7_33_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1399-0004.2002.620203.x"},{"key":"e_1_2_7_34_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1399-0004.2001.600405.x"},{"key":"e_1_2_7_35_1","doi-asserted-by":"publisher","DOI":"10.1586\/ern.09.34"},{"key":"e_1_2_7_36_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gde.2007.04.006"},{"key":"e_1_2_7_37_1","doi-asserted-by":"publisher","DOI":"10.1097\/GIM.0b013e3181ea4c1c"},{"key":"e_1_2_7_38_1","first-page":"34","article-title":"Quantifying health status and function in Marfan syndrome","volume":"25","author":"Rao SS","year":"2016","journal-title":"J Surg Orthop Adv"},{"key":"e_1_2_7_39_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1526-4637.2008.00498.x"},{"key":"e_1_2_7_40_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0002-9149(00)80066-1"},{"key":"e_1_2_7_41_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pain.2009.02.011"},{"key":"e_1_2_7_42_1","doi-asserted-by":"publisher","DOI":"10.1037\/1040-3590.7.4.524"},{"key":"e_1_2_7_43_1","doi-asserted-by":"publisher","DOI":"10.1097\/00002508-200103000-00008"},{"key":"e_1_2_7_44_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.35.5.405"},{"key":"e_1_2_7_45_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.12422"},{"key":"e_1_2_7_46_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.37370"},{"key":"e_1_2_7_47_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.12699"},{"key":"e_1_2_7_48_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1399-0004.2001.600507.x"},{"key":"e_1_2_7_49_1","doi-asserted-by":"publisher","DOI":"10.2147\/TACG.S60472"},{"key":"e_1_2_7_50_1","doi-asserted-by":"publisher","DOI":"10.1097\/00005650-199603000-00003"},{"key":"e_1_2_7_51_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00246-008-9346-5"},{"key":"e_1_2_7_52_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10865-007-9138-z"},{"key":"e_1_2_7_53_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2648.2010.05394.x"}],"container-title":["American Journal of Medical Genetics Part A"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fajmg.a.38051","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmg.a.38051","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,14]],"date-time":"2023-09-14T18:48:27Z","timestamp":1694717307000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ajmg.a.38051"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,11,14]]},"references-count":52,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2017,2]]}},"alternative-id":["10.1002\/ajmg.a.38051"],"URL":"https:\/\/doi.org\/10.1002\/ajmg.a.38051","archive":["Portico"],"relation":{},"ISSN":["1552-4825","1552-4833"],"issn-type":[{"value":"1552-4825","type":"print"},{"value":"1552-4833","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,11,14]]}}}